ORIGINAL

Niger J Paed 2014; 41 (3):229 –233

Atimati AO

Osarogiagbon OW

Prevalence of BCG scar among

BCG-vaccinated children in a

southern Nigeria tertiary

hospital

DOI:http://dx.doi.org/10.4314/njp.v41i3,15

Accepted: 6th April 2013

Abstract: Background: The bur-

information were obtained using a

proforma. The anthropometric

measurements of the children were

obtained and the children were

examined for presence of a BCG

scar.

den of tuberculosis is high in Nige-

ria as in other developing coun-

tries. The administration of BCG

vaccine to neonates is essential in

the control of tuberculosis. A scar

usually develops 6 – 8 weeks later

at the site of vaccination, which

can be used clinically as a proof of

vaccination. Not all vaccinated

infants however, develop a BCG

scar.

Objectives: To determine the

prevalence of scar formation post-

vaccination and to unravel, if pre-

sent, any factors responsible for

scar failure.

Atimati AO⁽

)

Osarogiagbon OW

Department of Child Health,

University of Benin Teaching Hospital,

Benin. Edo State, Nigeria

Results: Two hundred and six sub-

Email: tonyatimati@yahoo.com

jects (96.3%) had

a

post-

vaccination BCG scar. About 72%

of the subjects were vaccinated

within the first week of life. The

age at vaccination was signifi-

cantly affected by gestational

maturation (P=0.003) and birth

weight (P=0.0001). Gestational

maturation is a strong predictor of

BCG scar formation post-

vaccination (P = 0.007)

Conclusion: There is high preva-

lence of BCG scar formation in

this study and gestational matura-

tion is a strong predictor of BCG

scar formation.

Methods: Two hundred and four-

teen children were consecutively

recruited from those who presented

for immunization in the University

of Benin Teaching Hospital, Benin.

The bio-data and other relevant

2

Introduction

adult pulmonary form of the disease . In various clinical

trials the estimates of effectiveness have ranged from

3

Tuberculosis is an infectious disease which is prevalent

in developing countries. In 2011, there were an esti-

mated 8.7million new cases of Tuberculosis in the gen-

80% protection to no benefit . Despite its limitations, the

BCG vaccine is the only currently available vaccine for

the prevention of tuberculosis. Overall, more than 80%

of all neonates and infants in countries where the vac-

cine is part of the national childhood immunization pro-

eral population (13% co-infected with HIV) and

1

.4million people died from the disease . There were an

4

estimated 0.5million cases and 64000 deaths among

children in 2011.Africa and Asia have the highest bur-

den of tuberculosis . The African Region has approxi-

grammereceive the vaccine . The coverage however,

varies from country to country. The estimated BCG cov-

erage for the year 2011, reported by the World Health

Organization, revealed levels ranging from 54% in

Ethiopia and 60% in Nigeria, to 99.5% in India and

1

mately one-quarter of the world’s cases, and the highest

rates of cases and deaths relative to population. Nigeria

is one of the 22 countries with a high burden of tubercu-

losis, with an inciden₁ce of about between 90,000 to

5

China .

3

30,000 cases per year .

The BaccilleCalmette-Guerin contains a live attenuated

strain of Mycobacterium bovis which is administered

intra-dermally over the left deltoid muscle. After a

As part of control measures to reduce the burden of tu-

berculosis especially in children, the World Health Or-

ganization recommends vaccination with BacilleCal-

mette-Guerin vaccine at birth or first contact with health

period of 6 – 8 weeks post-vaccination a swelling

appears which increases in size and ruptures leaving

behind a life-long puckered scar after healing. The pres-

ence of a BCG scar and the tuberculin skin test are util-

ized in clinical settings to determine those who have

been immunized with the BCG vaccine. The tuberculin

skin test is usually positive in people who have received

the vaccine. Considering the fact that the tuberculin test

2

services, especially in developing countries. The World

Health Organization has emphasized this policy in re-

cent years, because of consistent evidence that BCG

protects against serious childhood forms of tuberculosis,

even where it may not protect to a high degree against

2

30

is also positive in those with the disease and those ex-

posed to non-tuberculous mycobacteria infection, it is

not specific for identifying those who have received the

vaccine. Moreover, the result is often negative in im-

munocompromised (HIV, disseminated tuberculosis,

malignant conditions) individuals, even those who had

previously been vaccinated, due to cutaneous anergy.

and 42 completed weeks of gestation as term while

those delivered after 42 completed weeks were classified

as post-term. The weight of the subjects was_Rmeasured

with an infant weighing scale, Way master made in

England, calibrated to the nearest 50gm; the length was

assessed with an infantiometer while the head circumfer-

ence was measured with a non-elastic measuring tape.

The left upper arm around the deltoid was examined for

presence of a BCG scar. The subjects were classified

nutritionally using the WHO weight for age z-score

growth charts. Subjects with z-score of less than –3 were

classified as severely under-nourished; between –3 and –

2 as moderately under-nourished; between –2 and +2 as

In the absence of a vaccination card the BCG scar may

thus be the only option left to clinically determine vacci-

nation status. It is however, noted that not all vaccinated

6

children develop a scar. Different studies world-wide

have reported varying prevalence rates of the presence

of BCG scar in vaccinated children. A study in Karachi,

9

normal; while above+2 as overweight. The data col-

involving 250 infants, reported presence of scar in

lected was recorded in Microsoft Excel spreadsheet and

transported to SPSS version 19 for analysis. Univariate

analysis was conducted for all variables to assess their

distribution. Continuous variables were summarized

using means and standard deviations while categorical

variables were summarized using proportions.

Chi-square test was used to determine association

between categorical variables. P-value of less than 0.05

was considered statistically significant.

6

8

0.4% of the infants. However in this study, the age at

vaccination, gestational age and other characteristics of

the children were not evaluated in order to find the pos-

sible reason for the absence of scar formation. A study

in Northern Nigeria reported a 95.1% prevalence of scar

7

formation. This study however, evaluated only 41 chil-

dren. Another study in Northern Nigeria evaluated 296

children between the ages of 3 – 59months receiving

immunization in a Teaching Hospital and two Primary

Health Centers. Only 55.7% of the vaccinated children

8

had a BCG scar. This study is aimed at evaluating in-

Results

fants in Southern Nigeria to determine the prevalence of

scar formation and to unravel, if present, any factors that

may be associated with BCG scar formation.

A total of two hundred and fourteen subjects comprising

117 (54.7%) males and 97 (45.3%) females were re-

cruited for the study. The mean age of the subjects was

4

.33 ± 2.54 months. The ages of the subjects ranged

from 6weeks to 15months. The age group of 6weeks – 6

months formed the bulk (90.7%) of the study popula-

tion. The general characteristics of the study population

are as shown in table 1.

Materials and method

This is a cross-sectional study carried out between June

and September, 2012 at the University of Benin Teach-

ing Hospital, Benin-city, Edo State. The Hospital offers

curative and preventive services to patients from Edo

State and the neighbouring States of Delta, Ondo,

Bayelsa, Ekiti and Kogi. Immunization services take

place in the General Practice Clinic and the Institute of

Child Health, on a daily basis from Monday to Friday,

except on public holidays. The immunization units of

the General Practice Clinic and Institute of Child Health

vaccinate about 1000 children respectively annually.

Ethical clearance for the study was obtained from the

Ethical Committee of the University of Benin Teaching

Hospital. A verbal consent was obtained from the par-

ents and caregivers of the subjects after explaining the

objectives and the harmless nature of the study.

Table 1: General characteristics of the study population

Characteristic

n

%

Gender

Male

Female

117

97

54.7

45.3

Age (in months)

1

7

≥

.5 – 6

– 12

13

94

16

4

90.7

7.5

1.9

Age at vaccination (days)

1 – 7

151

35

9

71.9

16.7

4.3

8

1

2

– 14

5 – 21

2 – 28

3

1.4

≥

29

12

5.7

Gestational Maturation

Pre-term

Term

15

186

9

7.1

88.6

4.3

The subjects were consecutively recruited from children

attending the General Practice Clinic, who had been

previously vaccinated with BCG and have presently

come for subsequent vaccines in the National Pro-

gramme on Immunization schedule. Information on the

bio data such as age, sex, and gestational age were ob-

tained using a proforma. Information on birth weight,

age at receipt of BCG vaccine and place of vaccination

were also obtained. Subjects delivered before 37 com-

pleted weeks of gestation from the mothers last men-

strual cycle were classified as preterm; those between 37

Post-term

Birth weight category

Low birth weight

Normal birth weight

High birth weight

Place of vaccination

Private Hospitals

UBTH

Other Public Hospitals

Nutritional status

Overweight

13

137

21

7.6

80.1

12.3

21

180

11

9.9

84.9

5.2

11

190

8

5.2

88.8

3.7

Normal

Underweight

Severe malnutrition

5

2.3

2

31

Majority of the subjects (71.9%) were vaccinated within

the first week of life_t_hwhile 16.7% were vaccinated be-

Table 3: Association between BCG scar formation and some

variables.

th

Variables Presence of scar Absence of scar

n(%) n(%)

P-value

0.057

tween the 8 and 14 day of life. Twelve (5.7%) sub-

jects were vaccinated after one month of life. Among

Gender

Male

those vaccinated within the first week o_n_df life 9.3% and

110(94)

96(99)

7(6)

st

1

0.6% were vaccinated on the 1 and 2 day of life re-

Female

1(1)

spectivel_ty_h. Majority of them (44.4%) were vaccinated

on the 7 day of life. A greater proportion (26.7%) of

pre-term infants were vaccinated after 4weeks of age in

comparison to the term (9.3%) and post-term (0%) sub-

jects as shown in table 2. This difference was statisti-

cally significant (P=0.003). Similarly, a greater propor-

tion (30.8%) of the subjects with low birth weight were

vaccinated after 4weeks in comparison to normal birth

weight (0.7%) and high birth weight (4.8%) babies. This

difference was also statistically significant (P=0.0001).

The above findings indicate that prematurity and low

birth weight are significantly associated with late pres-

entation of the study population for BCG vaccination.

Gestational Maturity

Pre-term

Term

Post-term

15(100)

180(96.8)

7(77.8)

0(0)

6(3.2)

2(22.2)

0.011

0.509

0.415

0.619

0.789

Age at vaccination (days)

1

≥

– 14

5 – 28

29

180(96.8)

11(91.7)

12(100)

6(3.2)

1(8.3)

0(0)

Birth weight categories

Normal

134(95)

7(5)

0 (0)

0(0)

Low birth weight 13(100)

High birth weight 21(100)

Place of vaccination

UBTH

173(96.1)

7(3.9)

1 (9.1)

0(0)

Other GovtHosp 10 (90.9)

Private Hospitals 21(100)

Nutritional status

Table 2: Association between age of vaccination and

gestational maturity and birth weight

Over-weight

Normal

Underweight

11(100)

182(95.8)

8(100)

0(0)

8(4.2)

0(0)

2

Age at vaccination (days)

χ

P-value

0

n(%)

– 14

15 – 28

n(%)

≥29

n(%)

Severe mal-nutrition 5(100)

0(0)

Gestational maturity

Pre-term

Term

Post term

9(60)

165(90.7) 9(4.9)

9(100)

2(13.3)

4(26.7)

8(4.4)

0(0)

16.28 0.003

35.93 0.0001

Discussion

0(0)

Birth weight category

The WHO recommendations for routine use in EPI

schedule and available data on BCG vaccine effective-

ness indicate that the vaccine should be administered as

soon as possible after birth and before 1 month of age

LBW

NBW

HBW

7(53.8)

130(94.9) 6(4.4)

18(85.7) 2(9.5)

2(15.4)

4(30.8)

1(0.7)

1(4.8)

1

for maximumprotection. In this study, 71.9% of the

subjects were vaccinated within the first week of life,

while 5.7% were vaccinated after one month of age.

Previous studies showed variable rates at reception of

BCG vaccination. A study from Sri Lanka reported 99%

Evaluation of the nutritional status of the study popula-

tion, as shown in table 3, showed normal nutrition in

1

2

90 (88.8%) subjects; 5.2% were overweight while

.3% had severe malnutrition.

1

2

reception of BCG within the first week of life . The

very high rate of vaccination within the first week of life

in the Sri Lankan study may be due to high awareness of

the need for BCG immunization which₁ is reflected in the

Presence of scar post-vaccination was observed in 206

of the subjects, giving a prevalence of 96.3%. There was

absence of scar in 8 (3.7%).Evaluation of the factors

related to scar formation showed a statistically signifi-

cant difference (p = 0.011) among subjects in the vari-

ous gestational age groups as shown in table 3. Absent

scar formation was highest among the post-term (22.2%)

in comparison to the term (3.2%) and pre-term (0%)

subjects. The presence of BCG scar was not signifi-

cantly associated with the place of vaccination; chrono-

logical age at vaccination, nutritional status, birth

weight, and gender of the subjects.

1

high BCG coverage of almost 100% as against 49.7%

1

2

in Nig₇ eria where this study was carried out. A similar

study from the Northern part of Nigeria reported a

lower percentage (36.2%) of BCG vaccination within

the first week of life. This difference might be due to a

lower BCG coverage in the Northern part of Nigeria

compared to Southern Nigeria, where our study was

carried out, as shown in the National Demographic

1

2

Health Survey in Nigeria .

It was observed from this study that birth weight and

gestational age significantly influenced the age of BCG

vaccination. These two factors are closely related as pre-

term neonates will most likely have a low birth weight.

Weight is usually a limitation in the commencement of

immunization in Nigeria since, from observation in most

immunization centres, a minimum weight of 2kg is in-

sisted upon by health workers before administration of

BCG. The same practice of late vaccination of Low birth

2

32

1

5

weight infants have also been reported in Guinea-

and centre of vaccination. Santiago et al , similarly, did

not find any association between scar formation and sex,

birth weight, age at vaccination and nutritional status.

There was however, a significant association between

development of BCG scar and gestational age in our

study, as a higher proportion of the post-term infants

showed absence of scar post-vaccination. The possible

reason for this finding is not quite apparent as previous

studies relating gestational age and scar formation post-

vaccination are at variance. Preterm neonates are more

likely to show absent scar formation compared to term

and post-term neonates due to₈ poor immune response as

1

3

Bissau . According to the World Health Organization,

pre-term infants in developing countries should be vac-

cinated with BCG at a post-conceptional age of

4

0weeks. Since establishing the correct gestational age

is a challenge in most developing countries, the birth

weight rather than gestational maturity is utilized in de-

fining when BCG is administered. This has varied impli-

cations for the low birth weight infant, since failure to

vaccinate children with BCG at birth has been reported

to contribute to lower BCG vaccination coverage among

1

3

low birth weight children. Early vaccination of Low

Birth Weight infants with BCG has also been reported to

reduce mortality rate by 17% in a randomized control

1

reported by Sedaghatianet al in the United Arab Emir-

1

9

ates. A study in India among preterm babies delivered

less than 35weeks gestation and vaccinated at birth and

at 38 – 40 weeks post-conception did not show any sta-

tistically significant difference in scar formation. The

small number of post-term infants in our study may af-

fect the interpretation of this finding and thus affect the

deductions made.

1

4

trial in Guinea-Bissau . Late reception of BCG in the

few patients (1%) reported in Sri Lanka was ascribed to

illness which resulted in the children being admitted in

1

the Special Care Baby Unit . The gestational age and

birth weight may be contributory as both are common

reasons for admission into the neonatal unit.

The prevalence of scar formation from our study indi-

cate that the presence of a BCG scar can be utilized as a

reliable clinical evidence of BCG vaccination, in the

absence of immunization card, as most of the studied

population (96.3%) developed a scar post-vaccination.

This observation is comparable to the findings from Pe-

Limitations of the study

Mothers’ information on birth weight and gestational

age was utilized in absence of information from the case

file. The accuracy of this information might not be com-

pletely reliable.

1

4

15

ru and India where the prevalence of scar formation

was 99% and 90.2% respectively. This observation is,

8

however, at variance with the study of Mustapha et al in

Conclusion

Northern Nigeria where the prevalence of scar formation

was 55.7%. This difference might be accounted for by

the different age groups in Mustapha’s study and these

other studies. Mustapha et al studied children between

the ages of 3 – 59months as against 6weeks – 15months

in our study, with children between the ages of 6weeks –

This study shows a high prevalence of BCG scar forma-

tion post vaccination in early childhood and gestational

age is a strong predictor of BCG scar formation post-

vaccination.

6

months forming the bulk (90.7%) of the study popula-

Author’s contributions

tion. The studies from India and Peru similarly studied

younger children vaccinated from birth to 3months of

age and were followed up until 6months.There has been

documented evidence of waning of BCG scar post-

vaccination in children followed up from infancy to

AOA: Conceptualization, methodology, planning and

data collection, analysis and writing of the manu-

script.

OOW: Methodology, planning and data collection,

analysis, proof reading of the

Conflict of Interest: None

Funding: None

manuscript.

1

7

fourteen years of age . The possibility therefore, of dis-

appearance of the BCG scar in the older children among

the subjects in Mustapha’s study could have contributed

to the lower prevalence of scar formation reported.

Other factors which include use of a non-potent vaccine,

faulty vaccination techniques and lack of maturation of

the immune system are documented factors that may

Acknowledgement

The authors gratefully acknowledge the contributions of

Dr. W.E Sadoh and Dr. D.O Nwaneri for their assistance

in the statistical analysis and Dr. A.E Sadoh for helping

to proof read the manuscript. Our appreciation also goes

to Dr. S. Awoyomi for his assistance in the data

collection.

15

contribute to failure of scar following vaccination .It is

difficult to ascertain if these factors contributed to the

difference in the prevalence of scar formation.

Presence of BCG scar was not significantly affected by

sex, birth weight, age at vaccination, nutritional status

References

2

.

Fine PE, Carneiro IA, Milstien JB,

3. Kernodle D. “Decrease in the ef-

fectiveness of BacilleCalmette-

Guerin Vaccine against Pulmonary

Tuberculosis”. Clin Infect Dis

2010; 177

1

.

World Health Organization.

Global tuberculosis report 2012.

World Health Organization 2012.

Available at http://www.who.int/

tb/publications/global_report/

gtbr/2_main.pdf.

Clements CJ. Issues relating to the use

of BCG in Immunization programs: a

discussion document. Geneva, Switzer-

land: Department of Vaccines and

Biologicals, World Health Organiza-

tion, 1999: 1 – 45.

2

33

4

5

.

World Health Organization. BCG

vaccine, Geneva, Switzerland.

World Health Organization 2013.

Available at http://www.who.int/

bilogicals/areas/vaccine/bcg/en

World Health Organization. Re-

ported estimates of BCG coverage.

World Health Organization2013.

Available at http: //

10. World Health Organization. EPI

Schedule report. World Health

Organization 2006. http://

www.who.int/immunization/

sage/3_EMRO_1_EPI_Schedule_

Report.pdf

15. Santiago EM, Lawson E, Gillen-

water K, Kalangi S, Lescano AG,

Du Quella G, Cummings K,

Cabrera L, Torres C, Gilman RH.

A prospective study of Bacillus

Calmette-Guerin scar formation

and Tuberculin skin test reactivity

in infants in Lima, Peru. Pediatrics

2003;112;e298. http://

pediatrics.aappublications.org./

content/112/4/e298.full.html.

16. Surekha RH, Vijayalakshmi V,

Sunil k, Lakshmi KA, Suman LG,

Murthy KJR. Cell mediated immu-

nity in children with scar-failure

following BCG vaccination. Ind-

Ped 1998;35:123 – 7.

17. Channabasava R, Mohan VM,

Suryanarayana , Murthy MSK,

Shashidhara AN. Waning of BCG

scar and its implications. Ind J

Tub. 1993;40:137–44.

18. Sedaghatian MR, Karduoni K. Tu-

berculin response in preterm in-

fants after BCG vaccination at

birth. Arch Dis Child 1993; 69:

309 – 11.

19. Thayyil-Sudham S, Kumar A,

Singh M, Paul VK, Deorari AK.

Safety and effectiveness of BCG

vaccination in preterm babies.

Arch Dis Child Fetal Neonatal Ed.

1999 July 81(1); F64 – F66.

11. Srisaravanapavanathan N, Dis-

sanayake NN, Sarathchandra J.

BCG vaccination scars of children

under five years in a tertiary care

hospital. Sri Lanka J Child healt

2008; 37: 81 – 4.

12. National Population Commission

(NPC) Nigeria and ICF Macro.

2009. National Health Demo-

graphic Health Survey 2008.

Abuja, Nigeria: National Popula-

tion Commission and ICF Macro.

13. Roth A, Jensen H, Garly

ML,Djana Q, Martins CL, Sode-

mann M, Rodrigues A, Aaby P.

Should low birth weight infants

receive BCG at birth? Community

survey from Guinea-Bissau. PIDJ

2004; 23: 544-50.

14. Aaby P, Roth A, Ravn H, Napirna

BM, Rodrigues A, Lisse IM,

Stensballe L, Diness BR, Lausch

KR, Lund N, Biering-Sorensen S,

Whittle H, Benn CS. Randomized

trial of BCG vaccination at birth to

Low Birth Weight children: bene-

ficial non-specific effects in the

neonatal period. JID

www.apps.who.int/

immunization_monitoring/

globalsummary/timeseries/

tscoveragebcg.html.

Sherjil A, Iqbal J. Absence of scar

formation in infants after BCG

vaccination. Prof. Med J 2006;13

6

7

.

(4):637-41.

Wammanda RD, Gambo MJ, Ab-

dulkadir I. Age at BCG admini-

stration during routine immuniza-

tion. J Comm Med Prim Healt

Care 2004;16(1):33-5.

Mustapha MG, Garba MA, Rabasa

AI, Farouk AG. Prevalence of

BCG scar formation among BCG

vaccinated apparently healthy U-5

children and its correlation with

Mantoux skin test induration in

Maiduguri, Nigeria. Niger Med J

8

9

.

2

008;49(4):84-7.

WHO Global Database on Child

Growth and Mal-nutrition. World

Health Organization Geneva,

1

997. http://

www.whqlibdoc.who.int/hq/1997/

WHO_NUT_97.4.pdf.

2011;204:245 – 52.

ORIGINAL

Niger J Paed 2014; 41 (3):234 –238

Onubogu UC

Anochie IC

Empiric antibiotic prescription

among febrile under-five Children

in the University of Port Harcourt

Teaching Hospital, Rivers State,

Nigeria.

DOI:http://dx.doi.org/10.4314/njp.v41i3,16

Accepted: 6th April 2014

Abstract: Background: More than

7% of febrile infants and young

studied. Two hundred and eighty

three (78.2%) febrile children re-

ceived empiric antibiotic prescrip-

tions. The most frequent antibiotic

prescribed was amoxicillin 80

(28.3%). Children aged 1-

12months received the highest

number of prescriptions 113

(80.7%). There was no significant

relationship between age, tempera-

ture level, weight for age, number

of symptoms and frequency of

antibiotic prescription (p>0.05).

Upper respiratory tract infection

(83.7 %) and diarrhea (55.9%)

were significantly associated with

empiric antibiotic prescription

(P=0.05 and 0.002 respectively).

9

Onubogu UC (

Anochie IC

)

children have self-limiting viral

infection and therefore, would not

require antibiotics. Over prescrip-

tion of antibiotics increases antibi-

otics exposure and development of

resistance among patients. There is

need to evaluate empiric antibiotic

prescription in order to limit its use

to only febrile children with bacte-

rial infection.

Aim and Objectives: The aim of

this study was to determine the

prevalence of empiric antibiotic

prescription among febrile under-

five, post neonatal children pre-

senting in the children outpatient

clinic of the University of Port

Harcourt teaching hospital.

Method: Febrile Children aged 29

days to <60 months who presented

in the outpatient clinic were re-

cruited from September 2010 to

January 2011. Their weight, bio-

data, symptoms, Physician’s diag-

nosis, and names of antibiotic pre-

scribed were entered into a prede-

termined proforma and analysed.

Result: A total of 362 children with

male to female ratio of 1.03:1 were

Department of Paediatrics,

Braithwaite Memorial Specialist

Hospital, Port Harcourt, Rivers State.

Email: utchayonubogu@yahoo.co.uk

Conclusion:

Empiric antibiotic

prescription for febrile under-five

children is a common practice in

UPTH. Physicians should there-

fore reduce the frequency of anti-

biotics prescription in febrile chil-

dren unless there is clinical

evidence of bacterial infection.

Key words: Empiric Antibiotics,

Fever, post neonatal under-five,

Nigeria

5

Introduction

signs, stiff neck or any sign of severe malaria . The

National institute for health and clinical excellence

(NICE) guide lines for management of febrile children

in the United Kingdom recommends empiric antibiotic

be given to children with suspected serious bacterial

Acute febrile illness among infants or young children is

a common clinical scenario, accounting for up to 30% of

paediatric clinic consultation . More than 97% of non-

1

6

toxic but febrile infants and young children have self-

limiting viral infection therefore would not require anti-

infection. Considering that the prevalence of bacterial

infections among febrile children in ambulatory clinic

setting is 1.1% in the United States of America, few

febrile children₇would actually require empiric antibiot-

ics prescription .

2

biotics . In Uganda, antibiotic was prescribed empiri-

cally to 59.5% of febrile, under-five children while in

Netherland, it was prescribed to 26.5% of febrile chil-

dren aged 1month to 6years .

3

,4

It is important to monitor antibiotic usage in order to

8

The Integrated management for childhood illnesses

protect their efficacy . This is because improper antibi-

(

Malaria endemic areas recommended use of antibiotics

when any of the following is present: General danger

IMCI) guideline for management of febrile children in

otic usage, increases antibiotic exposure among humans

and animals. This directly increases antibiotic resistance

by promoting emergence of resistant bacteria strains.

9

,10

2

35

If this continues unchecked it would ultimately cause

increased mortality from treatable diseases. In manage-

ment of the febrile child, there is a need to evaluate

empiric antibiotic treatment in order to limit its use to

Empiric antibiotic prescription.

Their nutritional status was determined using the Gomez

classification .

1

3

1

only children at risk of bacterial infection .

Statistical analysis was done using EP Info version 3.5.

Chi- squared test and Fishers Exact test were used to test

for significant associations between proportions. Com-

parison of means was done with the student’s t test. A p

value of 0.05 or less was considered statistically signifi-

cant

The aim of this study was to determine the prevalence of

empiric antibiotic prescription among febrile under-five,

post neonatal age children presenting in the children

outpatient clinic of the University of Port Harcourt

teaching hospital. We also set out to identify the factors

on which physicians base their decision to prescribe

empiric antibiotics and to identify the pattern of antibiot-

ics prescription. It is hoped that consideration of find-

ings from this study may lead to better founded and con-

sequently, diminished empiric antibiotic prescriptions.

This will ultimately help to protect available antibiotics

from the emergence of resistant bacteria strains, threat-

ening to render them ineffective.

Result

Three hundred and sixty two children who met the in-

clusion criteria were enrolled into the study. There were

184 (50.8%) males and 178 (49.2%) females, giving a

male to female (M:F) ratio of 1.03: 1. They were aged 1

to < 60 months (mean 21.1 ± 15.4 months). The mean

age of the male subjects was 20.8 ± 15.07 months, while

that of the females was 21.4 ± 15.8 months (p=0.15).

The median age for all the subjects was 18 months and

the modal age was 24months. Two hundred and fifty

eight (71.3%) children were aged ≤ 24months. The

mean temperature of the study population was 38.2 ±

Subjects and Methods

This was a prospective study that was carried out in the

Children’s outpatient clinic (CHOP) of the University of

Port Harcourt Teaching Hospital (UPTH) between

o

0.6 C (range 37.5 - 40.8 C). One hundred and ninety

children had axillary temperatures within the range of

o

September 2010 and January 2011. The University of

Port Harcourt Teaching hospital is a tertiary hospital

located in Southern part of Nigeria. The children outpa-

tient clinic runs both general and specialist paediatric

services. The general paediatric clinic is covered mostly

by Resident doctors and House officers with access to

review cases with the Consultant Paediatrician. Ethical

clearance for the study was obtained from the Ethics

Committee of the University of Port Harcourt Teaching

Hospital. Written informed consent was obtained from

parents or guardian. The minimum sample size of 362

was calculated using a bacteraemia prevalence rate of

37.5 - 38 C. Seven had temperature >40 C( Table 1).

Table 1: Temperature and age distribution of the study population

o

Temp ( C)

Age in months (%)

Total (%)

1-12

>12-24

>24-36

>36-

>48-

<60

4

8

3

7.5-38.0

85(44.7)

31(35.6)

54(28.4)

29(33.3)

31(16.3)

12(13.8)

10

10(5.3)

190(100)

87(100)

(5.3)

>38.0-38.5

5(5.7)

10

(11.5)

>

38.5-39.0

39.0-39.5

>39.5-40.0

>40.0

13(34.2)

9(30.0)

2(20.0)

0(0)

16(42.1)

11(36.7)

5(50.0)

3(42.9)

6(15.8)

8(26.7)

0(0)

1(2.6)

1(3.3)

0(0)

1

2(5.3)

1(3.3)

3(30.0)

2(28.6)

38(100)

30(100)

10(100)

7(100)

1(14.3)

(14.3)

3

8.2% among febrile infants in a children emergency

Total (%)

140

118

58(16.0)

18

28(7.7)

362(100)

12

ward in Nigeria . All the children that presented to the

clinic and met the inclusion criteria within the study

period were consecutively recruited. The criteria for

inclusion into the study was age one month to < 5years,

and axillary temperature ≥ 37.5°C. Children that had

initially received antibiotic were excluded. The tem-

perature and weight of all the children were measured

and recorded in a structured data collection form. Each

child was given a code prior to their scheduled consulta-

tion by the physician. In order not to influence the pa-

tients’ prescription, the attending physicians were not

informed that their prescriptions were being recorded.

After being attended by the physician, they were seen in

a separate room where data was collected by interview-

ing the caregivers. Sociodermographic information

regarding age, sex, address was obtained. Clinical infor-

mation including the number of symptoms the subject

presented with, Physician’s diagnosis, number and

names of antibiotic prescribed if any was obtained from

the patient files. The questionnaire was filled by the in-

vestigator. Any antibiotic prescribed prior to laboratory

evidence of any bacterial infection was defined as

(38.7)

(32.6)

(5.0)

Two hundred and sixty seven (_n7_d3.8%) children had

st

rd

normal nutritional status. 1 , 2 and 3 degree malnutri-

tion was seen in 72(19.9%), 21(5.8%) and 2(0.6%)

respectively. The mean weight of the study population

was 11.2 ± 4.5kg. Two hundred and eighty three

(

78.2%) febrile children received prescription for em-

piric antibiotics from the consulting physician. The most

frequent antibiotic prescribed was amoxicillin in 28.3%

of children (Fig 1). Antibiotics prescribed less than five

times were grouped under others and they include am-

picllox, ceftazidime, ceftriaxone, cephalexin, ciproflox-

acin, erythromycin, septrin, ampicillin/ sulbactam and

chloramphenicol. Most children 257(90.8%) received

one antibiotic while two antibiotics were prescribed in

2

5(8.8%) and 3 in a single prescription.

2

36

Fig 1: Names of Empiric antibiotics and frequency

Table 4: Nutritional status and empiric antibiotic prescription

9

8

7

6

5

4

3

2

1

0

8

0

Nutritional status

Gomez)

Empiric

antibiotic

Yes(%)

Empiric

antibiotic

No(%)

Total

6

9

6

(

2

6

1

9

11

Normal

210(78.7)

54(75.0)

18(85.7)

1(50.0)

57(21.3)

18(25.0)

3(14.3)

267(100)

72(100)

21(100)

2(100)

7

5

st

1 degree malnutrition

nd

2

3

degree malnutrition

rd

1(50.0)

Total

283(78.2)

79(21.8)

2(100)

2

χ =2.0, df=3, P=0.55

Children aged 1-12months received the highest number

of prescriptions (80.7%) and frequency of antibiotic

prescription decreased with age (Table 2). Although the

difference was not statistically significant (P=0.76).

Table 5: Number of symptoms excluding fever and antibiotic

prescription

No of symp-

Empiric anti-

biotic

No(%)

Total

toms excluding biotic

Table 2: Age distribution of children given empiric antibiotics

fever

Yes(%)

Age

Empiric

antibiotics

No (%)

Total

0

1

2

3

4

≥5

Total

16(72.7)

55(76.4)

108(77.1)

70(83.3)

29(76.3)

5(83.3)

6(27.3)

17(23.6)

32(22.9)

14(16.7)

9(23.7)

1(16.7)

79(21.8)

22(100)

72(100)

140(100)

84(100)

38(100)

6(100)

(months) antibiotics

Yes (%)

1

-12

113(80.7)

93(78.8)

44(75.9)

13(72.2)

20(71.4)

283(78.2)

27(19.3)

25(21.2)

14(24.1)

5(27.8)

8(28.6)

79(21.8)

140(100)

118(100)

58(100)

18(100)

28(100)

362(100)

>12-24

>24-36

>36-48

>48-<60

283(78.2)

362(100)

2

Total

χ =2.08, df=5, P=0.83

2

χ =1.86 df=4 P=0.76

Febrile Children who had upper respiratory tract infec-

tion (URTI) presenting only as either cough or catarrh or

both were given empiric antibiotic prescription in 83.7%

of their consultations. Those who had diarrhoea received

prescription for empiric antibiotics in 55.9% of their

consultations (Table 6). Diagnosis that occurred in less

than 9%(32) of the study population was not evaluated

because of the small sample size. URTI and diarrhoea

were significantly associated with increased antibiotic

prescription (P=0.05 and 0.002 respectively).

o

All children with temperature >39.5-40.0 C received

empiric antibiotics (Table 3). There was however, no

significant relationship between temperature level and

the frequency of empiric antibiotic prescription (P=0.2)

Table 3: Relationship between temperature and empiric

antibiotic prescription

Temperature range Empiric anti-

Empiric

antibiotic

No(%)

Total

o

(

C)

biotic

Yes(%)

Table 6: Clinical diagnosis and frequency of antibiotic

prescription

3

7.5-38.0

143(75.3)

71(81.6)

31(81.6)

24(80.0)

10(100)

4(57.1)

47(24.7)

16(18.4)

7(18.4)

6(20.0)

0(0)

190(100)

87(100)

38(100)

30(100)

10(100)

7(100)

>38.0-38.5

>38.5-39.0

>39.0-39.5

>39.5-40.0

>40.0

Clinical

Empiric antibiotic Empiric antibi-

P value

diagnosis

Yes(%)

otics

No(%)

URTI

118(83.7)

41(87.2)

33(86.8)

19(55.9)

23(16.3)

6(12.8)

5(13.2)

15(44.1)

0.05

0.1

0.2

3(42.9)

Tonsillitis

Pneumonia

Diarrhoea

Total

283(78.2)

79(21.8)

362(100)

2

0.002

χ =6.4, df=5, P=0.2

Children with second degree malnutrition received the

highest number of prescriptions for empiric antibiotics

(

Table 4). Only two children however had third degree

Discussion

malnutrition. Nutritional status was not significantly

related with the frequency of prescriptions (P=0.55)

An overall frequency of 78.2% for empiric antibiotic

prescription among febrile under-five children is high.

This high frequency of antibiotic prescription is similar

to 72.2% reported among similar age group in Tanza-

nia, another African country . African countries with

developing economies could have similar challenges in

their health sector. In such settings, challenges with

laboratory services ranges from delay in laboratory re-

sults to complete absence of laboratories. Results of

laboratory investigations to confirm infection usually

takes more than 24 hours. This necessitates a second

Children that presented with 3 and ≥5 symptoms

received the highest number of empiric antibiotics

prescription (Table 5), while 72.75% of children that

presented with fever and no other symptom where given

empiric antibiotic prescription. The number of symp-

toms the patient presented with was not significantly

related to the frequency of empiric antibiotic

prescription.

1

4

2

37

visit or often the patient is lost to follow up. This situa-

tion makes the physician to opt for empiric antibiotic

prescription. A study done in the Netherland among

febrile children reported a lower antibiotic prescription

shown in Table 1. Although hyperpyrexia has been

documented to be associated with higher risk for sepsis,

guidelines on management of febrile children recom-

mends that, height of body temperature alone should not

be used to identify children with high risk for bacterial

infection bu₆t_,₇t_,h₁₆e age of the child should also be taken

4

rate of 26.5% . In the Netherland study a practice

guidel₅ine for the management of febrile children was

1

used. This guideline does not recommend routine use

into account

.

of antibiotics in children with fever without an apparent

source. This adherence to the guideline could have con-

tributed to the lower frequency of antibiotic prescription.

Also the mean temperature of the Netherlands study was

Febrile Malnourished children could receive higher anti-

biotic prescriptions as they have a higher risk of bacte-

2

3

rial infectio_nn_d . This was the case in our study as chil-

dren with 2 degree malnutrition received the most anti-

biotics. The number of symptoms did not significantly

affect the rate of antibiotic prescriptions. This finding is

not surprising as increased symptoms could have meant

more system involvement in the on-going pathology but

does not differentiate between viral and bacterial

aetiology.

o

lower than our study (37.9 vs 38.2 C respectively). The

lower temperature could mean that the study population

in the Netherlands study was at a lower risk of bacterial

infection than our study population. Previous studies

have demonstrated that high temperatures in association

with young age increases the likelihood of bacterial in-

6

,16

.

fection

Amoxicillin and cefuroxime were the most frequently

prescribed antibiotics in this study. Amoxicillin, a nar-

row spectrum penicillin has been reported in Tanzania,

Nigeria and America as on₁₄e_,₁₇o_,₁f₈ the common antibiotics

In our study 83.7% of children who had common cold

were prescribed antibiotics. This directly contravenes

WHO and IMCI treatment guidelines, which discour-

ages₅_,₂t₄he use of antibiotics in children with common

cold . In Tanzania 68.9% of children with common

cold were prescribed antibiotics, while in USA 29.6%

of children received antibiotic for acu₄t_,e₁₈respiratory tract

used in paediatric practice

. The high use of cefu-

roxime, a broad spectrum cephalosporin in our study

shows a growing pattern of clinicians choosing more

expensive and broader spectrum antibiotics in their prac-

tice. Similar trend has also been reported in USA and it

raises serious concerns about the overuse of broad-

spectrum antibiotics, particularly for patients for whom

1

infections when it was not indicated . Antibiotics do

not reduce the severity or duration of illness in viral in-

fections. Thus their use in viral illness exposes a patient

to the risks of side effects from a medication without

any benefit. Antibiotics_,₂a₄re also not recommended for

18

antibiotic therapy is not indicated at all . The recom-

mended principle for rational antibiotic prescription in-

cludes: Choosing a drug that has efficacy in treating or

preventing the disease but leaves other bacteria in the

body intact₁₉a_,₂n₀d one that is available, convenient and

5

acute watery diarrhoea . Our study however reported

55.9% antibiotic prescription rate in management of

diarrheal diseases. Similar high frequency of antibiotic

prescriptio₁₄n_,₂₅_,h₂₆as been reported in other low income

countries.

inexpensive

.

The risk of bacterial infection is higher in youn₁ger chil-

2

dren due to immaturity of the immune system. knowl-

edge of this fact may encourage physicians to prescribe

empiric antibiotics more often in this age group. How-

ever, even among febrile young children there is an ur-

gent need for classification based on risk for bacterial

infection using clinical guidelines. In Europe and Amer-

Conclusion

In conclusion, empiric antibiotic prescription for febrile

under-five children is very high in UPTH. This finding

has also been reported in studies conducted in other low

income countries. Such high rate of empiric antibiotic

prescription would lead to increased development of

resistant strain of bacteria to the present antibiotics and

threatens the end of antibiotic era. There is a need to

protect available antibiotics by rational prescription only

when they are indicated. The use of available practise

guidelines in the management of febrile children would

help reduce inappropriate antibiotic prescription in feb-

rile children. The campaign to protect these antibiotics

needs to be actively brought to low income countries.

These countries have a higher prevalence of infectious

diseases and as such would have greater mortality

should these drugs be rendered inactive.

6

,15,22

. These

ica these guidelines are already available

guidelines use both clinical and laboratory parameters in

risk assessment for bacterial infection. This is based on

the assumption that results of these laboratory tests are

available to the physician during the index consultation.

In a resource poor setting however, this is not the case,

so an assessment for risk of bacterial infection in a feb-

rile child is often made without the use of any laboratory

results.

The highest antibiotic prescription rate was found

among children with temperature range of 39.5-40°C.

The combination of young age and hyperpyrexia may

have contributed to the 100% antibiotic prescription

seen in this group of children. This is because in our

study, children with temperature range of 39.5-40°C

were much younger (70% < 24mths) while those with

temperature >40°C were older (57% > 24mths), as

We recommend implementation of current evidence

based practice that advocates for prior documentation of

evidence of bacterial infection by laboratory testing

before antibiotic prescription for febrile children. The

2

38

febrile child at urgent need for empiric antibiotic should

however receive it while laboratory testing to document

bacterial infection is being done. We acknowledge the

need for locally useful clinical detector/screening tools

which could be used in the absence of sophisticated

laboratory methods to identify the febrile child at risk of

serious bacterial infection. There is no doubt that classi-

fying children based on their risk assessment for bacte-

rial infection prior to commencing antibiotics will,

identify the small group of children that need urgent

commencement of empiric antibiotics and at the same

time, limit the irrational use of antibiotics. This prac-

tice may reduce development of antibiotic resistance and

reduce the cost of healthcare.

Conflict of interest: None

Funding: None

References

1

.

Eskerud JR, Laerum E, Fagerthun

H, Lunde PKM, Naess AA. Fever

in general practice Frequency and

diagnoses. Fam Pract. 1992;263–

10. Hay AD, Thomas MM, Montgom-

19. Iyalomhe GBS, Iyalomhe SI,

Eholor RE. Antibiotic prescription

and resistance: A contemporary

literature review. Int J Med Med

HYPERLINK "http://

www.academicjournals.org/

journal/IJMMS" Sci. 2011; 3(14):

376-380.

20. World Health Orgarnization, The

Rational Use of Drugs - Report of

the Conference of Experts, Nairobi

25-29 November 1985, World

Health Organization, Geneva.

21. Baker MD. Evaluation and man-

agement of infants with fe-

ver. Pediatr Clin North Am 1999;

46(6):1061-72.

22. Baraff LJ, Bass JW, Fleisher GR,

Klein JO, McCracken GH

ery A, Wetherell M, Lovering A,

McNulty C. The relationship be-

tween primary care antibiotic pre-

scribing and bacterial resistance in

adults in the community: a con-

trolled observational study using

individual patient data. J Antim-

icrob Chemother. 2005;56: 146-

153.

11. Finkelstein JA, Christiansen CL,

Platt R, Fever in Pediatric Primary

Care: Occurrence, Management,

and Outcomes. Pediatrics 2000;

105(2): 260 -266.

2

69.

2

.

Jaskiewicz JA, McCarthy CA,

Richardson AC, et al. Febrile

infants at low risk for serious bac-

terial infection—an appraisal of

the Rochester criteria and implica-

tions for management. Febrile

Infant Collaborative Study Group.

Pediatrics.1994;94:390–6.

Batwala V, Magnussen P, Nu-

waha F. Antibiotic use among

patients with febrile illness in a

low malaria endemicity setting in

Uganda. Malar J 2011;10:377.

Elshout G, Marijke K, Johannes C,

et al. Antibiotic Prescription in

Febrile Children: A Cohort Study

during Out-of-Hours Primary

Care. J Am Board Fam Med 2012;

3

4

.

12. Ayoola OO, Adeyemo AA,

Osinusi K. Concurrent bacterae-

mia and malaria in febrile Nige-

rian infants. Trop Doc 2005; 35

(1):34-6.

Jr.,Powell KR, et al. Practice

guideline for the management of

infants and children 0 to 36 months

of age with fever without source.

Agency for Health Care Policy and

Research. Ann Emerg Med

13. Gomez F, Ramos GR, Frenk S,

Cravioto MJ, Chavez R, Vasquez

J. Mortality in second and third

degree malnutrition. J trop pedi-

atr Afr child health 1956; 2:

14. Gwimile JJ, Shekalaghe SA, Ka-

panda GN, Kisanga ER. Antibiotic

prescribing practice in manage-

ment of cough and/or diarrhoea in

Moshi Municipality, Northern

Tanzania: cross-sectional descrip-

tive study. Pan Afr Med J 2012;

12:103.

2

5: 810-818.

5

6

.

World Health Organization. Inter-

national Management of childhood

Illnesses chart book_nf_dor primary

health Care Level. 2 ed. 2004; 7-

1993;22:1198–210.

23. Schaible UE, Kaufmann SH. Mal-

nutrition and Infection: Complex

Mechanisms and Global Impacts.

Plos Med 2007; 4(5):115.

1

7.

Richardson M, Lakhanpaul M.

Assessment and initial manage-

ment of feverish illness in children

younger than 5 years: summary of

NICE guidance. BMJ

24. World Health Organization. Pocket

book of hospital care for children

guidelines for the management of

common illnesses with limited

resources. 2007; 72-81, 109-130

25. Hoan le T, Chuc NT, Ottosson E,

Allebeck P. Drug use among chil-

dren under 5 with respiratory ill-

ness and/or diarrhoea in a rural

district of Vietnam. Pharmacoepi-

demiol Drug Saf. 2009 ;18(6):448-

53.[PMID:19326362]

26. Siddiqi S, Hamid S, Sauerborn R

et al. Prescription practices of pub-

lic and private health care provid-

ers in Attock District of Pakistan.

Int J Health Plann Manage. 2002;

17: 23-40.

15. Berger MY, Boomsma LJ, Albeda

FW, et al. The standard of the

Dutch College of General Practi-

tioners on children with fever.

Huisarts en Wetenschap

2

007;334:1163–4.

7

.

Stanley R, Pagon Z, Bachur

R. Hyperpyrexia among infants

younger than 3 months. Pediatr

Emerg Care 2005; 21(5):291-4.

2008;51:287–96.

[PMID:15874809]

16. Lee GM, Harper MB. Risk of

bacteraemia for febrile young

children in the post-Haemophilus

influenzae type B era. Arch Pedi-

atr Adolesc Med 1998; 152

8

.

Morris K Battle against antibiotic

resistance is being lost. Lancet

Infect Dis.2007; 7(8):509.

Magee JT, Pritchard EL, Fitzger-

ald KA et al. Antibiotic prescrib-

ing and antibiotic resistance in

community practice: retrospective

study, 1996–8. BMJ 1999; 319:

9

.

(7):624-8.

17. Oshikoya KA, Chukwura HA,

Ojo OI. Evaluation of outpatient

paediatric drug prescriptions in a

teaching hospital in Nigeria for

rational prescribing. Paediatr

Perinat Drug Ther 2006;7:183-8

1

239–40.

1

8. Hersh AL, Shapiro DJ, Pavia AT,

Shah SS. Antibiotic Prescribing in

Ambulatory Pediatrics in the

united states. Pediatrics. 2011;128

(6):1053-1061.

CASE REPORT

Niger J Paed 2014; 41 (3): 239 –243

Amuabunos AE

Eregie CO

Omoigberale AI

Effiong V

Conjoined twins in Edo state of

Nigeria; a report of the first

surviving set

DOI:http://dx.doi.org/10.4314/njp.v41i3,17

Accepted: 8th March 2014

Abstract:

The term conjoined

delivered in 2009 and survived a

separation surgery. A third set of

female thoracoomphalopagus was

delivered in another institution

same year and referred to our unit

but they only survived for 48

hours.

The first surviving twins were om-

phalopagus, sharing a single liver,

and common bile duct emptying

into a common duodenum. The

stomach, as well as the jejunum

was normal and unshared. Surgical

separation of the liver was done

and biliary reconstruction proce-

dure performed for twin II. A three

-year follow up showed good out-

come.

twins refers to babies who are

physically joined at some point. It

is a rare condition with an esti-

mated incidence of 1 per 200,000

live births. We report our experi-

ence with conjoined twins over a

twelve year period in tertiary hos-

pital in Nigeria and a case of the

first set of conjoined twin survivors

in Benin City, Nigeria. Over the

last twelve years (1999-2011),

three cases of conjoined twin have

been recorded in our teaching hos-

pital. A set of thoracoomphalo-

pagus twins (females) were deliv-

ered in 1999 and they survived for

only 36hrs. Another set of female

omphalopagus twins were

Amuabunos AE

Eregie CO

(

)

Omoigberale AI

Department of Child Health,

Effiong V

Neonatal Unit

University of Benin Teaching hospital,

Ugbowo, Benin City,

PMB 1111, Edo State, Nigeria.

Email: amua4@yahoo.com

Introduction

nancy. Overall the condition is rare with an estimated

incidence of 1 per 200,000 live births . Though there are

4

The care of conjoined twins continues to pose a daunt-

ing medical challenge that includes adequate care of

pregnancy, well planned delivery, critical care in early

neonatal life, advance surgical intervention and last but

more live born females conjoined twin with a female to

male ration of 3:1, however this condition occur more in

male foetuses as evidenced by its higher rates in male

stillborn. Male conjoined twins are also more likely to

die shortly after birth, implying that female conjoined

foetuses have better chance at survival than their male

counterparts.

The site of union forms the basis of the terms used for

classifying conjoined twins: Thoracoomphalopagus

(joined at the chest, abdomen or both) – 74%

1

,2

not the least is the ethical issues . We report our experi-

ence with conjoined twins over a twelve year period in a

tertiary hospital in Nigeria and a case of the first set of

conjoined twin survivors in Benin City, Nigeria. The

incidence of conjoined twins in Nigeria is unknown;

however an article published in 2001 suggests that over

the preceding 60 years there were 12 published cases

Thoracopagus or xiphopagus ( joined at the chest) - 40%

Omphalopagus (joined at the abdomen) - 34%

3

nationwide, excluding our own cases. The cases seen in

our center have so far not been reported perhaps because

this center had not recorded any survival of conjoined

twins since its existence three and half decades ago.

Therefore under reporting may be due to the poor prog-

nosis and stigmatization associated with this condition.

Pygopagus (joined at the buttocks) – 18%

Ischiopagus (joined at the ischium) – 6%

Craniopagus (joined at the head) – 2%

A rare type occurs when one incompletely formed

(parasitic) twin is dependent on the well-formed one.

This is known as heteropagus twinning. The term

“pagus” is a Greek word which means “that which is

fixed”

The term Siamese twins comes from Eng and Chang

Bunker (1811-1874), the famous conjoined twins from

Thailand (previously known as Siam). They were thora-

copagus twins and were exhibited in circus shows

around the world before settling in the United States,

where they married two sisters and had nearly two dozen

children. They were successful businessmen and lived

up to 63 years. There have been several other reports of

Conjoined twining is one of the most fascinating human

malformations but it is not exclusive to our specie as it

has also been reported in other mammals, reptiles, birds

4

and fishes. The term conjoined twins refers to babies

who are physically joined at some point. It results from

incomplete splitting of monozygotic (identical) twins

after 12 days of embryogenesis. Some authors recently

had postulated that it actually results from “fusion” of

4

stem cells of already separated embryo . Conjoined

twinning occurs sporadically with no risk in future preg-

2

40

conjoined twins in different parts of the globe. In Nige-

ria the earlies_tt_hreport of conjoined twins were born in

Sokoto on 20 December 1935 to a 25 year old para 3

sporadic, howbeit scanty, reports of conjoined twin from

the country, but a three year survival follow up of the

survivors is generally scarce. Since the first documenta-

tion in 1935 till date, 18 cases have so far been reported

as summarized in table 1. There are, nonetheless, some

cases of conjoined twins that were found in the Nigerian

news reports that never made the medical literature,

Table 2

5

woman at home. They shared only abdominal wall and

skin, but no shared internal organs. They were readily

separated at the General Hospital in Sokoto by a British

5

missionary doctor. Twenty years later the Kano ompha-

lopagus twins Tamonotanye and Waiboko, were sepa-

6

rated in London by Ian Ard. Since then there have been

Table 1: Summary of conjoined twin reported from Nigeria

Place of delivery Author(s)

Type of Conjoin Twins No of pairs Place of surgery

Outcome

Twin I Twin II

Sokoto

Kano

Port Harcourt

McLaren, 1936

Aird, 1945

Holgate and

Omphalopagus

1

Sokoto General Hospital

Hammersmith, UK

Enugu General Hospital

S

D

S

Ikpeme, 1956

Stigglebout, 1958

Gupta, 1966

Kaduna

Ibadan

Thoracopagus

Pygopagus

Thoracopagus

1

-

Hammersmith, UK

UCH, Ibadan

Still Born

S

D

Still Born

D

Omokhodion et al,

2

001

Warri

Zaria

Bankole et al, 1972

Mabogunje, 1978

Ischiopagus

Omphalopagus

Thoracopagus

Dicephalus

1

UCH, Ibadan

ABUTH, Zaria

D

Still Born

S

D

Still Born

D

1

980

Sathiakumar et al,

990

1

Pygopagus

1

NDU Sule

Anambra

Mabogunje and

Lawrie, 1978

Iroku and Anah,

Heteropagus

Pygopagus

1

S

-

UNTH, Enugu

D

1

990

Lagos

Ile-Ife

*2003

Adejuyigbe et al,

005

Thoraco-abdomino pagus

Ischiopagus

John Hopkins, Baltimor

OAUTHC, Ile-Ife

S

2

Enugu

Maiduguri

Ekenze et al, 2009

Auwal et al, 2011

Omphalopagus

Ischiopagus

1

Germany

UMTH, Maiduguri

S

*Total

18

*Total excluded the conjoined twins seen at the UBTH between 1999 and 2011

Table 2: News reports of Nigerian conjoined twins

Gender

Date

Birth place

Extent of joining

Place of surgery Website

Chest

??

2005

Owerri

Abdomen

www.allafrica.com/stories/200503040379.html

Pelvic girdle

Genitalia

F

2013

2012

Oturkpo

Kano

Abdomen

Pyopagus

www.newsinnigeria.org/2013/18

www.punchng.com

India

Heart

Chest to abdomen

Upper intestines

Thoracoomphalopagus

One heart

F

M

2013

Nasarawa

Enugu

www.news2.onlinenigeria.com

www.nigerianuniversitynews.com/2013/06

Joined genitals

F

2011

Jos

Parasite twin – no head Ibadan

Chest down

www.enownow.com

3

legs

One liver

One intestine

www.dailytrust.info/index.php/city-news/2010

-abuja

M

F

2013

2004

Abuja

www.nigerianmonitor.com

www.business,highbeam.com/3548/article-IGI

Abakaliki

??

-121544947

Surgical separation of conjoined twins that results in the

death of one, or both, of the twins raises complex moral,

ethical and legal issues. Where organs such as brain or

heart are shared there is a great risk of one or both twins

dying if attempt is made at separation. Indeed, any

shared organ is often not shared equally and the question

often arises as to who should be left with what. Of par-

ticular concern is the potential for homicide charges

against doctors. The parents of the Manchester twins,

2

Mary and Jodie born in 2000, refused to grant permis-

sion for surgery, despite the judges’ ruling in favour of

surgery. A circumstance, where Mary was sacrificed at

surgery₇, was argued by some as “a murder Mary to save

Jodie”.

2

41

Over the last twelve years (1999-2011), three sets of live

conjoined twins were documented in our teaching hospi-

tal. The hospital has an average annual delivery rate of

Twins I and II weighed 4.7kg and 4.8kg respectively

after surgery. At 2 years postnatal age they weighed 8.5

and 8.6kg respectively while at 3 years they weighed

13.4kg and 13.6kg. Their psychomotor development

was compatible with their age at 18 months using the

Bailey Developmental Scale. Thorough clinical and

laboratory re-evaluation at age three, paying particular

attention to the cardiovascular, digestive and renal sys-

tems of the twins yielded normal findings.

1

,600 and serves as a major referral center for a popula-

tion of approximately six million people. There were

two thoracoomphalopagus and one omphalopagus twins.

One set of the thoracoomphalopagus twins was deliv-

ered elsewhere. Both sets of thoracoomphalopagus twins

died within 48 hours following birth.

Case

Fig 1: The

conjoined

twins at two

weeks of life

th

The surviving twins were delivered on the 9 of Sep-

tember 2009 at 02:48hrs to a young couple at the Uni-

versity of Benin Teaching Hospital, in Benin City, Edo

State which is in the South-South part of Nigeria. Their

mother, a 29 year old lady regis_t_htered this first pregnancy

at our health facility at the 19 week of gestation. Ob-

stetric ultrasound scan done at her antenatal booking

revealed that she had a set of omphalopagus twins. The

antenatal period remained otherwise unremarkable until

Fig 2: After surgery

th

the 34 week of gestation when she went into spontane-

ous preterm labour. She was delivered of live female

omphalopagus twins by an emergency Caesarian Section

Twin I

(

Fig 1). The twins were both small-for-dates. They had a

combined birth weight of 3.4kg but the Apgar Scores

were good. They were joined at the level of the xiphis-

ternum to a point just above the umbilicus. There was a

small exomphalos with separate umbilical cords. They

both had mild respiratory distress syndrome which

resolved within 72hrs following delivery. On the third

day of life they developed jaundice requiring photother-

apy: the highest bilirubin levels were 13.2 for twin 1 and

Twin II

1

3.5mg/dl or twin 2. They were treated for Escherichia

coli sepsis with ciprofloxacin and gentamycin guided by

the antibacterial sensitivity. By the third week of life

they had shown evidence of full recovery and had re-

gained their combined birth weight. From the fourth

week they were on full milk feeds and had satisfactory

growth.

Thoraco-abdominal CT scan revealed that they both

shared a single liver and proximal part of the gut. Twin I

had dextrocardia without any functional abnormality.

Extensive evaluation of the other systems was normal.

Discussion

They remained in our newborn unit, until the age of nine

months. Their combined weight was 9.8 kg, and a sepa-

ration surgery was then performed at the Narayana hos-

pital in Bangalore, India. Findings at surgery included a

single liver that was “fused” in the midline with separate

blood supplies. There was a common gallbladder and

bile duct that emptied into a common duodenum which

extended up to about 20cm in length. Each twin had her

own stomach and jejunum. The liver was divided and

biliary reconstruction procedure done for twin II. The

duodenum was shared between the two by resection and

re-anastomosis. The twins required initial_t_hmechanical

ventilation and were weaned off by the 4 day. Post

surgery they remained stable and were transferred back

to the UBTH (fig 2). Physical therapy was instituted to

enable them “catch-up” with their motor development

that was hitherto made difficult whilst conjoined.

This case is a report of the first surviving conjoined

twins in a decade of conjoined twins history in this cen-

ter. Overall reporting of conjoined twins is low in the

country. Review of available literature showed that 18

cases have been reported across the country in the last

7

6 years from 1935 to 2012. Five cases, 28%, were re-

8

ported from a single center in the North, Zaria,₃ while

9

the other cases were reported from Ibadan, Ife,

10

Enugu and few tertiary health centers in other parts of

the country. The higher report from Zaria may be due to

the heightened interest of the workers. None so far has

been reported from Benin and some other tertiary cen-

ters across the country to enable a more countrywide

data review. In contrast, 22 cases were reported from a

single institu₁ti₁on in Philippines, over a 30 year period

(

1974-2006). An institution in Sao Paula, Brazil re-

ported 14 cases over a 25 year period further reflecting

2

42

1

2

possible underreporting in Nigeria. It is hoped that this

report will be an important contribution to the few exist-

ing publications in the country. Although a small num-

ber of centers in the country have reported survival of

conjoined twins, of note is that information on follow up

morbidity and mortality were generally lacking. Our

surviving conjoined twins were followed up for catch-up

growth, psychomotor development, presence of organ

dysfunction and possible late complications of the sur-

gery. All these parameters turned out to be normal at the

age of three years. Due to the complexity of the surgical

separation, a follow up to evaluate long term survival

and quality of life is useful reviewing surgical interven-

tion in the future. In the recent₃case of separation of het-

favourable outcome following surgery. In contrast to

reports from other parts of the world, the author is un-

aware of any report of adult conjoined twins in the coun-

try. This situation may not be surprising as these babies

might have been deprived of care and left to die largely

because of stigma, poverty and ignorance. The current

case required a lot of psycho-social support for the

young parents who had initially abandoned these babies.

On the contrary the Biddenden Maids, born in England

in 1100, were famous and lived for 34 years. The

Siamese twins were also wealthy and famous in the

United States. We suggested that providing national

awareness, special government support and opening

national conjoined twins’ registry will go a long way in

improving the outcome of these babies, especially when

surgery pose a survival risk to one or both twins.

1

eropagus twins in Maiduguri, the twins had major re-

constructive surgery, consequently, long term follow up

of these twins will be complementary to our knowledge.

All three sets of conjoined twins seen at our center were

females, which is in keeping with the female preponder-

ance noted in the literature. Four out of the six babies

suffered early neonatal deaths while 2 (index twins) sur-

vived at 3-year follow up. Due to paucity of data it is

difficult to say, with any degree of accuracy, what the

still birth rate or neonatal death rate for conjoined twins

in the country is. Three of the reported 18 pairs where

still born, all the reported live born had surgery, 11 in

Nigeria and 4 abroad (Table I). Six out of the eight ba-

bies (75%) operated abroad survived (one baby was sac-

rificed to save the other twin). Twelve out of 22 babies

Conclusion

There is under reporting of conjoined twins in Nigeria

compared to other parts of the world. Experience from

available literature showed that these can be largely pre-

vented by demystifying the condition, providing more

awareness and support for the families. These measures

will go a long way to improving reporting as well as

enhancing the survival of these babies. Secondly paedia-

tricians and surgeons in Nigeria might want to review

their decisions to separate when the risk to one or both

twins is greater than the risk without the procedure.

(

54%) operated in Nigeria survived. Success rate was

fairer with ischio/pygopagus twins (58.3%) and poor

with thoraco/omphalopagus twins, especially when in-

ternal organs are shared.

Authors’ contribution

Amuabunos AE, Eregie CO, Omoigberale AI

Effiong V: All managed the patient and reviewed the

manuscript

A careful review of the current case with surgeons in our

institution and consult with other surgeons within and

outside the country informed the choice of having the

surgery done abroad to improved the chances of survival

of both twins considering the shared organs. This re-

flects the need to build on the already existing capacity

to handle such cases.

Conflicts of interest: None

Funding: None

While some have questioned the decision to separate

conjoined twins “when two are born as one”, having the

Acknowledgement

2

twins separated may seem justified if it is adjudged that

one or both twins would die without separation. This is

the case in some heteropagus twin situations in which

the parasite twin may die and/or cause the host twin seri-

ous physiologic embarrassment due to vascular, biliary

or enteric anastomosis. Even though our omphalopagus

twins are likely to survive into adulthood, the decision to

separate was preferential because of the expected

The authors acknowledge with gratitude the assistance

of the management of the Narayana Hospital in Banga-

lore, India and the surgical team especially Professor

Asley D’Cruz. We also thank the surgical team at the

university of Benin Teaching Hospital under the leader-

ship of Professor Evbounwan for their expert and pro-

fessional contribution to the care of the babies.

References

1

.

Davis C. Separating conjoined

twins: a medical and criminal law

dilemma. J Law Med. 2010;17

3. Omokhodion SI, Ladipo JK, Ode-

4. Spencer R. Conjoined twins: theo-

retical embryologicbasis. Teratol-

ogy 1992;45:591–602.

5. McLaren DW. Separation of con-

joined twins. Brit Med J 1936;

ii:971

bode T O, et al The Ibadan con-

joined twins: a report of omphalo-

pagus twins and a review of cases

reported in Nigeria over 60 years.

Ann Trop Paediatr. 2001;21

(3):263

(4):594-607.

2

.

Gillett G. When two are born as

one: the ethics of separating con-

joined twins. J Law Med. 2009;17

6. Aird I. The Conjoined Twins of

Kano. Brit. Med. J 1954 ;1: 831

(2):184-9.

2

43

7

.

Paris JJ, Elias-Jones AC. "Do we

murder Mary to save Jodie?" An

ethical analysis of the separation

of the Manchester conjoined

10. Ekenze SO, Ibeziako SN, Adimora

GN, et al. Ruptured omphalocele

in thoracoomphalopagus conjoined

twins. Int Surg. 2009;94(3):221-3.

11. Saguil E, Almonte J, Baltazar W,

et al Conjoined twins in the Philip-

pines: experience of a single insti-

tution. Pediatr Surg Int. 2009 ;25

(9):775-80

12. Berezowski AT, Duarte G, Rodri-

gues R, et al Conjoined twins: an

experience of a tertiary hospital in

Southeast Brazil. Rev Bras Ginecol

Obstet. 2010;32(2):61-5.

13. Abubakar AM, Ahidjo A, Chinda

JY, et al The epigastric het-

eropagus conjoined twins. J Pedi-

atr Surg. 2011;46(2):417-20

twins. Postgrad Med J. 2001;77

(911):593-8.

8

.

Mabogunje OA, Lawrie JH. Con-

joined twins in West Africa. Arch

Dis Child 1980; 55:626–30.

9

.

Adejuyigbe O, Sowande OA, Ola-

banji JK, et al. Successful separa-

tion of two pairs of conjoined

twins in Ile Ife, Nigeria: case re-

ports. East Afr Med J. 2005;82

(1):50-4.

CASE REPORT

Niger J Paed 2014; 41 (3): 244 –246

Okocha EC

Ulasi T

Aneke JC

Ajuba IC

Okwummuo EP

Unusual presentations of

childhood acute lymphoblastic

leukaemia: A case report

DOI:http://dx.doi.org/10.4314/njp.v41i3,18

Accepted: 6th April 2014

Abstract: Childhood acute lym-

following a bone marrow aspira-

tion study that revealed abnormal

cellularity consistent with L1

morphological subtype. Unfortu-

nately, the child was discharged

against medical advice before de-

finitive therapy could be com-

menced.

phoblastic leukaemia, (ALL) is

increasingly reported to present in

an atypical fashion which may

have significant implications for

treatment outcomes and survival.

This case report presents a

Nigerian child who’s clinical and

radiological features together with

effusion cytological findings were

suggestive of metastatic neuroblas-

toma. However, a definitive

(

)

Aneke JC

Okocha EC, Ajuba IC, Okwummuo EP

Department of Haematology,

Ulasi T

Department of Paediatrics,

Nnamdi Azikiwe University Teaching

Hospital, PMB, 5025, Nnewi,

Anambra State, Nigeria.

Email: anekejc@ymail.com.

Key words: Atypical presenta-

tions, metastatic neuroblastoma,

childhood acute lymphoblastic

leukaemia.

diagnosis of ALL was established

Introduction

Case Report

Acute lymphoblastic leukaemia (ALL) is a malignant

haematological condition that arises from an acquired

somatic mutation in a lymphoid progenitor cell . This

mutation may occur at various points in the development

of the lymphoid progenitor. Malignant proliferation and

accumulation of lymphoid blasts in the bone marrow

and some extramedullary sites such as the liver, spleen,

skin, testes (in males) and even the central nervous sys-

tem (CNS) is the hallmark of this disease.

A 4- year- old male Nigerian was seen at the Nnamdi

Azikiwe University Teaching Hospital, Nnewi, with a

14- week history of recurrent fever, multiple facial

swellings with enlargement of the head and protrusion

of the eyes, (fig. 1). There was also a history of signifi-

cant weight loss and nasal discharge that occasionally

was blood tinged.

1

ALL accounts for up to 30% of childhood cancers in

Caucasians , thus it is among the most common paediat-

ric malignancies.

2

Fig1: Showing head and

anterior chest wall swellings

Clinically, ALL may have diverse patterns of presenta-

tion; typically its clinical presentation is related to bone

marrow failure and extra medullary effects of the dis-

ease. As such common symptoms range from those aris-

ing secondary to cytopaenias (including anaemia, leuco-

paenia and thrombocytopaenia) to those due to organ/

system infiltration such as lymphadenopathy and hepa-

tosplenomegaly. Increasingly, unusual presentation of

childhood ALL is being documented in literature and

cases presenting with back pain and vertebral compres-

General examination was significant for marked weight

loss, moderate mucosal pallor, significant generalized

lymph node enlargement and bipedal pitting oedema.

Three discrete masses were noted on the left part of the

frontal bone, left part of the jaw and anterior chest wall,

measuring 10cm, 6cm and 9cm in their longest diame-

ters respectively. These swellings were globular, firm to

hard in consistency, non mobile, non tender and were

neither attached to overlying skin nor showed any differ-

ential warmth.

3

4

5

sion , stroke , absence of blasts in the peripheral blood ,

6

obstructive jaundice , and isolated masseter muscle

7

involvement have been variously reported.

We report here a case of ALL presenting with atypical

features, in a Nigerian child to highlight challenges of

diagnosis.

Bilateral parietal bossing along with coronal sutural

diathesis was also noted. The anterior fontannel was

2

45

patent and normotensive, measuring 2cm x 2cm. He had

no signs of meningeal irritation and muscle tone and

power were normal globally.

terminated as child was discharged against medical ad-

vice.

His abdomen was uniformly distended with palpably

enlarged, firm and tender liver, 10cm below the right

costal margin. Ascites was present and demonstrable.

Chest examination was significant for reduced chest

expansion and stony dull percussion notes over the right

hemi thorax with absence of breath sounds in both the

right mid and lower zones.

A provisional clinical diagnosis of neuroblastoma me-

tastatic to the right hemi thorax and the head region was

considered.

Discussion

Atypical presentations of ALL have reportedly consti-

tuted an enormous challenge, in terms of diagnosis, es-

pecially in resource poor settings. However, advances in

diagnostic protocols, especially in the realm of immuno-

phenotyping and relevant molecular diagnostics have

greatly enhanced diagnostic precision in such atypical

3

-6

cases . While such levels of diagnostic accuracy is de-

sirable, the application of basic cytological techniques

for the analysis of appropriate specimens has continued

to provide valuable information in resource poor set-

tings. In this patient, bone marrow study was able to

establish a diagnosis of ALL.

An abdomino-pelvic ultrasonographic examination

showed a right sided supra renal mass while a chest ra-

diograph demonstrated right sided pleural effusion, the

cytology of which revealed hypercellular smears show-

ing sheets of medium sized cells with high nucleo-

cytoplasmic ratio, in a dirty background. The neoplastic

cells have coarse chromatin pattern. Overall features

were suggestive of a malignant (round) blue cell tumour,

probably neuroblastoma. These findings reinforced me-

tastatic neuroblastoma as the most probable diagnosis.

The patient was seronegative for HIV 1 and 2, while

haemoglobin electrophoresis confirmed AA haemoglo-

bin phenotype. Complete blood count was significant for

severe anaemia (Haematocrit was 0.17L/L), moderate

Our patient presented with head enlargement, facial and

anterior chest wall swellings. Typically children with

ALL tend to present with extra medullary organ enlarge-

ment owing to infiltration by lymphoblasts and while the

liver, spleen and testes are the most frequently affected

organs, other less common extra medullary sites have

been reported in the literature. Coronal sutural diathesis

as well the patent anterior frontannel observed in this

patient may be an indication of CNS infiltration by lym-

phoblasts; a CT scan however was not done to confirm

9

leucocytosis (white cell count of 20.8 x 10 /L) and mild

9

7

thrombocytopaenia (platelet count of 76 x 10 /L). Blood

this. Wimperis et al in 1992 described two children with

film and bone marrow cytology were however in keep-

ing with ALL, L1 morphological type (figs 2 and 3).

Flow cytometric analysis of peripheral blood cells

showed positivity for CD 45, an extended immunophe-

notypic profile as well as cranial computed tomography

ALL in whom isolated masseter muscle involvement

was the only presenting feature of the disease. Accurate

diagnoses of the cases were hinged on the use of im-

munophenotyping and immunogenotyping. Indeed, such

extended panel of diagnostic tools have proved useful in

establishing diagnoses in similar atypical cases of ALL

(

CT) scan could not be done because of the non

3

4

availability of funds.

presenting as vertebral compression , stroke , absen₆ce of

5

blasts in peripheral blood and obstructive jaundice .

Fig 2: Bone marrow

film, showing L1

lymphoblasts

The initial diagnosis in this child was metastatic neuro-

blastoma based on his age and the clinical presentation

of multiple masses in the head region and chest and re-

enforced by the ultrasonographic finding of a supra renal

mass together with the pleural effusion cytology report.

However, in this patient, peripheral blood and bone mar-

row cytology were both in keeping with ALL of the L1

morphological type. Besides, the demonstration of CD

4

5 lineage antigen supported the haematopoietic origin

of this malig₈nant condition. In a recent case study, D’an-

gelo et al, reported ALL co-existing with neuroblas-

toma in a 3 year old girl, as different disease entities. We

had entertained the possibility of this phenomenon at a

stage in the management of this child prior to bone mar-

row investigations; in point of fact, biopsied tissue sam-

ple of one of the masses had been scheduled but later

considered unnecessary.

Fig 3: Peripheral

blood film, showing

lymphoblasts

Advances in ALL treatment have ushered in an indi-

vidualized, tailored and risk adapted approach, utilizing

a myriad of chemotherapy options with or without

haematopoietic stem cell transplantation. Treatment

Supportive treatment including blood product transfu-

sions were commenced while work up including biopsy

of the masses was planned as a prelude to definitive che-

motherapy for ALL. However, further care was abruptly

2

46

stratification is commonly based on the biologic features

of individual d₉isease . Utilizing the risk adapted proto-

diagnostic tests is essential in making early and accurate

diagnoses in cases of ALL with atypical presentations.

2

col, Pui et al, suggested that prophylactic cranial irra-

diation, which has been a component of the standard

treatment of childhood ALL, may safely be omitted.

Unfortunately our patient did not stay long enough in

our care to receive any definitive treatment; he was not

followed up to the community.

Conflict of Interest: None

Funding: None

Limitation of this report

A biopsy and histology of the body masses, including

that on the adrenals (preferably via ultrasound guide)

might have been a more definite way to rule out the pos-

sibility of neuroblastoma co-existing with ALL in this

child, this was however not done.

Conclusion

Childhood ALL may present in a rather atypical manner.

A high index of suspicion, complimented by appropriate

References

1

.

Provan D, Singer CRJ, Baglin T,

Dokal I. Oxford Handbook of

Clinical Haematology. Oxford

University Press, New York. 3

Ed. 2009.p132.

Lo Nigro L. Biology of childhood

acute lymphoblastic leukaemia.

J PediatrHematol Oncol.

5. Cogulu O, Karapinar DY, Karaca

8. D’angelo, Grigoli A, Sementa AR,

Tropia S, Alaggio R, Arico M.

Simultaneous diagnosis of acute

lymphoblastic leukaemia and pe-

ripheral neuroblastic tumour in a

child. J PediatrHematol Oncol.

2012;34:75-5.

9. Piu CH, Campana D, Pei D, Bow-

man WP, Sandlund JT, Kaste SC

et al. Treating childhood acute

lymphoblastic leukaemia without

cranial irradiation. N Engl J Med.

2009;360:2730-41.

E, Aydinok Y, Ozkinay F. Un-

usual course of an acute lym-

phoblastic leukaemia case with i

(9q) as a sole cytogenetic abnor-

mality. Leuk Res. 2006; 30:1461-

3.

6. Alvaro F, Jain M, Morris LL, Rice

MS. Childhood acute lymphoblas-

tic leukaemia presenting with

jaundice. J Paediatr Child Health.

1996;32:466-8.

7. Wimperis JZ, Brandt LJ, O’Con-

nor S, Marcus R, Broadbent V.

Unusual presentation of common

acute lymphoblastic leukaemia

antigen-positive extra medullary

disease in childhood. Two patients

with isolated masseter muscle

involvement. Cancer.1992

rd

2

3

.

2

013;35:245-52.

Hafiz MG, Islam A, Siddique R.

Back pain and vertebral compres-

sion: an unusual presentation of

childhood acute lymphoblastic

leukaemia. Mymensingh Med J

2

010; 19:130-6.

4

.

Ege MJ, Meyer LH, Debatin KM,

Stahnke K. Co-incidence of recur-

rent hemiparesis and detection of

acute lymphoblastic leukaemia in

a 4 year old girl: one or two dis-

ease. KlinPediatr. 2009;221:386-

15;70:8897-901.

9

.

CASE REPORT

Niger J Paed 2014; 41 (3): 247 –250

Paul NI

Ugwu RO

Diphtheria in a 13 year old

adolescent girl: Management

challenges

DOI:http://dx.doi.org/10.4314/njp.v41i3,19

Accepted: 10th April 2013

Abstract: Background: Diphthe-

ria is an acute toxic infection which

is associated with a high morbidity

and mortality and can pose man-

agement challenges especially in

the absence of proper diagnostic

and therapeutic facilities.

Case report: A.S. was a 13 year

old girl who presented with fever

of five days duration, dysphagia

and neck swelling of 4 days dura-

tion and sore throat and hoarse

voice of 3days duration. Her ill-

ness started a day after returning

from a 4-day holiday youth camp.

She received only oral polio vac-

cine immunization in childhood.

Significant physical examination

findings included a swollen neck, a

greyish membrane covering the

soft palate and uvula with haemor-

rhagic spots. The pharynx, anterior

nares and the nasal turbinates were

inflamed and erythematous.

A working diagnosis of respiratory

diphtheria was made. Throat swab

microscopy showed club shaped

Gram positive baccilli. Appropri-

ate culture medium for C. diphthe-

ria was not available.

She received intravenous crystal-

line penicillin and metronidazole

and lateroral erythromyc_ti_hn in an

isolated ward. On the 6 day of

admission she developed cardiac

and neurologic complications–

bradycardia (PR=40bpm),

hypotension (BP=70/40mmHg),

drooling of saliva and paraparesis.

Electrocardiography confirmed a

comple_tt_he heart block. She died on

the 11 day of admission while

efforts were being made to raise

funds for a cardiac pace maker.

Conclusion: Management of this

vaccine preventable disease re-

quires a high index of suspicion

and diphtheria antitoxin should be

made readily available.

Paul NI⁽

Ugwu RO

)

Department of Paediatrics & Child

Health

Faculty of Clinical Sciences

University of Port Harcourt,

Port Harcourt Nigeria.

Email: nsypaul@yahoo.co.uk

6

Introduction

>95% across the region in the past 10 years . In Nigeria

also, reported cases of diphtheria has been declining

7

Diphtheria is an acute toxic infection caused by Coryne-

bacterium species, typically Corynebacterium diphthe-

riae and_,₂r_,₃arely toxigenic strains of Corynebacterium

even with just low to moderate coverage with DPT3.

Accordingly, there has been no reported case from

Our centre in the past 10 years.

1

ulcerans . The classic disease affects the upper respi-

ratory tract with the formation of an adherent gray-white

pseudomembrane in the infected place followed by sys-

temic symptoms caused by elaboration of an exotoxin

However, recently there are pockets of sporadic cases

being reported in Nigeria. Sadoh et al reported nine

8

cases of diphtheria in children who were aged between

11months and 10years in the University of Benin Teach-

ing Hospital (UBTH) between 2008 and 2010, while

1

,4

produced by the bacillus . The disease progresses rap-

idly with a case fatality rate as high as >20% in acute

disease states if there is no sufficient diagnostic proce-

9

Oyeyemi et al reported ten cases of diphtheria in chil-

1

dure and therapy option . Therefore it requires a high

dren aged 3-13years in the Federal Medical Centre

Katsina on two clusters of diphtheria outbreak between

2009 and 2010 involving three contiguous local govern-

ment area in Katsina State. In this case we report a 13

year old girl who died from probable diphtheria myocar-

ditis and the diagnostic and management challenges

encountered.

index of suspicion. The most dominant factor causing

death is myocarditis and diphtheria myocarditis inci-

dences related to nasopharyngeal d₅iphtheria is 10-20%

with a death rate as high as 50-60% .

The emergence of immunization program changed the

epidemiology of the disease and reduced its prevalence

worldwide. In the Western world, diphtheria is near

Case Report

6

eradication level in most countries . Also, in many Afri-

can countries with a high diphtheria immunization cov-

erage rate, the incidence of diphtheria has decreased by

AS was a 13 year old girl who presented at the Children

Out Patient Clinic of the University of Port Harcourt

2

48

Teaching Hospital with complaints of fever of five

days duration, dysphagia and neck swelling of four days

duration, sore throat and hoarse voice of three days

duration. Her illness started a day after returning from a

four-day holiday youth camp. She received amoxicillin

capsules before presentation. She had never been immu-

nized except for Oral Polio Vaccines which she received

on National Immunization Days (NIDs).

Fig 2: ECG tracing of

AS showing complete

dissociation of the p

wave and QRS com-

plex which are wid-

ened (172ms), idio-

ventricular rhythm

with rate of 25/mm

and a giant T wave

inversion

Physical examination revealed a lethargic child in pain-

ful distress with a bull neck, hoarse voice, and drooling

saliva. Throat inspection showed a thick greyish mem-

brane covering most part of the soft palate and hanging

down over the uvula with areas of haemorrhagic spots.

The pharynx was erythematous, the anterior nares and

the nasal turbinates were inflamed and plugged with

blood crusts. (Fig 1) She had a good volume and regular

pulse with a rate of 82 beats per minute, a blood

Discussion

Diphtheria is an acute toxic infection caused by Coryne-

bacterium diphtheriae, an aerobic, non-encapsulated,

Gram positive bacillus. C. diphtheriae is an exclusive

1

pressure of 100/70mmhg and normal heart sounds. She

had no neurological deficits.

inhabitant of human mucous membranes and skin. It

spreads primarily by airborne respiratory droplets, direct

contact with respiratory secretions or exudates from

infected skin lesion. Incidence peaks during the dry sea-

son with majority of the cases occurring in unimmu-

nized children below 15 years of age. Diphtheria occurs

by entry of C. diphtheriae into the nose or mouth. After

a 2-4 day incubation period, toxins are secreted which

leads to toxin-mediated tissue necrosis. This coupled

with local inflammatory response produces patchy exu-

dates which later forms fibrinous exudates and a tough

Fig 1: Greyish

adherent membrane

in the soft palate and

uvula, and the haem-

orrhagic exudates in

the nostrils

4

adherent membrane. Respiratory embarrassment may

follow extension of disease into larynx or tracheobron-

chial tree.

A diagnosis of probable respiratory diphtheria was

made. Microscopy of the throat swab and swab of the

anterior nares showed club shaped Gram positive rods.

Culture using Tellurite salt agar could not be done as

this was not available. She was reviewed by the Otorhi-

nolaryngologist while the State Disease Surveillance and

Notification (DSN) unit was notified.

Our patient never had DPT vaccine and hadjust returned

from a crowded youth camp. These are strong risk fac-

tors for respiratory diphtheria. She also presented with

features typical of probable respiratory diphtheria like

sore throat and dysphagia, progressive neck swelling,

haemorrhagic and inflamed nasal turbinates and an ad-

herent greyish white membrane hanging down the phar-

ynx. The early presentation and short duration of these

symptoms confirms the short incubation period and

rapid progression of the disease as this child at presenta-

tion within five days of disease onset was already very

ill and lethargic.

She was nursed in an isolation room, received intrave-

nous crystalline penicillin at 0.4MU/kg/day in 4 divided

doses, intravenous Metronidazole at 8mg/kg/dose every

8

hours, intra venous fluid, oral toileting with saline wa-

ter and bed rest. All close contacts were counseled espe-

cially on the need to immunize all under-5 children

whose last DPT dose was more than 12 months ago and

were placed on Tablets Erythromycin – 500mg qds for

two weeks.

Complications remain the greatest cause of morbidity

and mortality following infection with diphtheria. Com-

plications secondary to the elaborated diphtheria toxin

are the most common. Toxic cardiomyopathy most com-

monly occur in the second week of the disease but can

appear _,a₁₀s early as the first or as late as the sixth week of

th

By the 6 day of admission, she developed cardiovascu-

lar complications – bradycardia (PR=40bpm) and

hypotension (BP=70/40Hg). She received 20mls/kg of

normal saline over 30minutes, intravenous hydrocorti-

sone and Atropine with no apparent clinical i_t_hmprove-

ment. Her condition deteriorated and by the 7 day of

admission her pulse rate dropped further down to 24bpm

and the power in the lower limbs was reduced to grade

two. A diagnosis of Diphtheria Toxic cardiomyopathy

1

illness . Toxic cardiomyopathy occurs in 10–25% of

patients with respiratory diphtheria and is responsible

1

for 50–60% of deaths . Neurologic complications appear

after a variable latent period, are predominantly bilateral

and are motor rather than sensory and usually resolve

completely. Paralysis of the soft palate is common and

generally appears in the third week. Our patient devel-

oped features of myocarditis by the second week of dis-

ease onset and bilateral motor weakness of the lower

limbs by the third week which is in line with disease

(

Heart Block) and neuropathy (Para paresis) was made.

An electrocardiogram confirmed a Complete Heart

Block. (Fig 2) Parents were counselled on the need for

an urgent pacemaker. Efforts were ongoing to raise fund

for a pacemaker before she died on the 11 day of

admission.

th

2

49

progression. This early onset of cardiac manifestation is

associated with rapid disease progression and is a poor

prognostic feature as was the case of our patient. Drool-

ing of saliva and hoarse voice in this patient may be due

to sore throat and dysphagia or to paralysis of the soft

palate.

respond to it. This was probably because the disease has

reached an advanced stage before presentation and

elaborated toxins may have fixed to tissues which are

not affected by antibiotics. Management of complica-

tions was also challenging. Our case developed both

cardiac and neurologic complications both of which may

have contributed to the mortality. Although she was

diagnosed of having complete heart block, lack of funds

and unavailability of the pacemaker made this manage-

ment option not available

A

diagnosis of diphtheria may be described as

“probable” or “confirmed”. It is probable if the case

meets the clinical description or confirmed if a probable

case is laboratory confirmed or linked epidemiologically

to a laboratory confirmed case. A clinical description is

an illness characterized by laryngitis or pharyngitis or

tonsillitis, and an adherent membrane on the tonsils,

pharynx and/or nose. However, persons with positive C.

diphtheriae cultures and not meeting the clinical de-

scription (i.e. asymptomatic carriers) should not be re-

ported as probable or confirmed diphtheria cases. Our

patient met the criteria for probable diphtheria but could

not be confirmed. Our centre and many others in Nigeria

lack the appropriate capacity and skills for the isolation

of this organism and this does have several deleterious

effects on the management and surveillance of this

vaccine preventable disease.

Primary prevention in form of active immunization as

DPT vaccine at 6,10, 14 weeks of age and booster dose

at 15-18 months and again between 4-6 years of age is

recommended. The National Programme on Immuniza-

tion (NPI) presently does not provide booster doses but

a high coverage rate in infancy provides significant dis-

ease protection. Unfortunately, our case received neither

the primary vaccine nor booster dose. This buttresses the

need to reinforce and ensure full coverage of primary

immunization by checking immunization cards as a re-

quirement for school enrolment.

All household contacts and those who have had intimate

physical contact with a patient are closely monitored for

illness through the 7-day incubation period. Antibiotic

prophylaxis is given, regardless of immunization status

using erythromycin (40-50 mg/kg/day) for 7-14 days or

A lot of challenges were encountered in the management

of this patient. Once diphtheria is suspected, manage-

ment entails isolation of the patient, use of specific anti-

toxin and antibiotics, management of complications,

supportive care and chemoprophylaxis for close contacts

of patient. The first management challenge was lack of

specific diphtheria antitoxin. The use of specific anti-

toxin is vital in halting disease progression. Antitoxin

can neutralize circulating toxin or toxin that is absorbed

to cells but is ineffective once cell penetration has oc-

curred. Specific antitoxin is the main stay of therapy and

should be administered as early as possible by intrave-

nous route and in a dosage sufficient to neutralize the

free toxin. Unfortunately as important as this is in the

treatment of patient with diphtheria this anti toxin is

unavailable in the country. Only a probable diagnosis

could be made in this case as it could not be bacte-

riologically confirmed by the appropriate culture

1

a single injection of benzathine penicillin. This was

done for all close contacts of our patient including the

managing team. Unfortunately, it was not possible to

trace the asymptomatic carrier from whom our patient

contracted the disease, neither was it possible to trace

other adolescents that participated in the youth camp for

possible development of symptoms.

Conclusion

In conclusion, diphtheria, a vaccine preventable disease

(VPD) is a disease with rapid progression and requires a

high index of suspicion. Facilities necessary for the di-

agnosis and treatment of this disease especially diphthe-

ria specific antitoxin should be made readily available in

Nigeria. Parents and caregivers of children should utilize

the opportunity of free immunizations to vaccinate their

children, for indeed, prevention is better than cure.

medium. This challenge may not be limited to our cen-

tre as many other centres contacted to assist with the

culture also admitted not having the culture medium.

Antibiotics are indicated to clear the causative organism

and thereby halt toxin production, and prevent transmis-

sion of organisms to contacts. Our patient received intra

venous crystalline penicilline and metronidazole which

have very good coverage for diphtheria but did not

Conflict of interest: None

Funding: None

References

2

.

Seto Y, Komiya T, Iwaki M,

3. De Zoysa A, Hawkey PM, Engler

K, George R, Mann G, Reilly W.

Characterization of toxigenic

Corynebacterium ulcerans strains

isolated from humans and domes-

tic cats in the United Kingdom. J

Clin Microbiol. 2005;43:4377–81

1

.

Stephen B E. Diphtheria In: Behr-

man RE, Kliegman RM, Jenson

HB, Stanton BF. (editors) Nelson

Textbook of Pediatrics, 18th edi-

tion. Philadelphia: W.B. Saunders

Company, 2007. 1153-1157.

Kohda T, Mukamoto M, Takaha-

shi M. Properties of corynephage

attachment site and molecular

epidemiology of Corynebacterium

ulcerans isolated from humans and

animals in Japan. Jpn J Infect Dis.

2

008;61:116–22

2

50

4

5

.

Vitek CR, Wharton M. Diphtheria

toxoid. In: Plotkin S, Orenstein W,

Offic P, (editors). Vaccines. Am-

sterdam: Elsevier Inc.; 2008: 139–

6. WHO World Health Organization

Immunization, Vaccines And

Biologicals. Vaccine preventable

diseases Vaccines monitoring

system 2013 Global Summary

Reference Time Series DIPH-

THERIA

7. Oyeyemi BO, Suleiman AO,

Suleiman BM, AjetomobiA, Ibra-

him A. A report on two clusters of

diphtheria outbreak involving

three contiguous local Govern-

ment Area in Katsina state_n_d, Nige-

ria. Proceedings of the 42 An-

nual General and Scientific Con-

ference of the Paediatric Associa-

tion of Nigeria (Panconf), 2011.

Jan 11-15 Abuja, Nigeria. P 12

8. Sadoh AE, Okhakhu A, Omuemu

V, Lofor PVO, Osarogiagon W,

Oviawe O, Diphtheria in Nigeria:

Is ther_ne_dresurgence. Proceedings of

the 42 Annual General and Sci-

entific Conference of the Paediat-

ric Association of Nigeria

5

6.

Ralph DF, Barbara WS, Pratip

KN. Diphtheria In: Feigin RD,

Cherey JD. (eds) Textbook of

Pediatric Infections Diseases, 6th

edition. Philadelphia: W.B. Saun-

ders company, 2009. 1393-1395.

(Panconf), 2011. Jan 11-15 Abuja,

Nigeria. Pg 40-41

CASE REPORT

Niger J Paed 2014; 41 (3): 251 –253

Garba BI

Adelakun MB

Aminu MS

Onazi SO

Musa A

Incidental finding of dextrocardia

with situs inversus totalis in a day

old neonate: Case report and

review of the literature

Sule MB

DOI:http://dx.doi.org/10.4314/njp.v41i3,20

Accepted: 10th April 2013

Abstract: Dextrocardia with situs

inversus are rare congenital anoma-

lies which can be asymptomatic

and compatible with normal life.

They are characterized by mirror

images of all intra-thoracic and

intra-abdominal viscera. Our aim is

to report an incidental finding of

dextrocardia with situs inversus in

a neonate with neonatal sepsis. A

day-old male term neonate pre-

sented with features of infection.

Physical examination revealed car-

diac apex on the 4th right intercos-

tal space, along the mid-clavicular

line. Chest radiograph and abdomi-

nal ultrasound confirmed the diag-

nosis of dextrocardia with situs

inversus. Bilateral cervical ribs

were also seen on chest radio-

graph. He was managed with anti-

b i o t i c s a n d d i s c h a r g e d .

Newborn babies should have a

thorough physical examination

after delivery before discharge to

enable early diagnosis of congeni-

tal anomalies for appropriate refer-

ral.

Garba BI (

)

Adelakun MB

Department of Paediatrics,

Aminu MS

Department of Medicine

Yariman Bakura Specialist Hospital,

Tudun Wada round about, PMB 1010,

Gusau, Zamfara State.

Email: bgilah@yahoo.com

Onazi SO

Department of Paediatrics

Federal Medical Centre, Gusau.

Key words: Dextrocardia,

neonate, neonatal sepsis.

Musa A

Department of Paediatrics

Ahmadu Bello University Teaching

Hospital, Zaria.

Sule MB

Department of Radiology

Usmanu Danfodio University Teaching

Hospital, Sokoto.

Introduction

Case report

Dextrocardia (also called looping defect) is an abnormal

congenital positioning of heart on the right side . Situs

A 24 hour old male term neonate presented with com-

plaints of refusal to suck, fever, convulsion and bloody

stool of few hours duration. He had associated abdomi-

nal distension, but no vomiting or bleeding from any

other site. Pregnancy was supervised, uneventful, no

maternal risk factors of sepsis. Delivered at a general

hospital and cried immediately after birth. He was not

examined by a paediatrician after delivery as none is

available at the hospital.

1

inversus totalis also called situs transversus, is a con-

genital condition in which major visceral organs are

reversed or mirrored from normal positions. Many peo-

ple with situs inversus are unaware of their unusual con-

genital anomaly until they seek medical attention for

1

unrelated conditions . Individuals with dextrocardia and

situs inversus totalis may have associated congenital

2

heart malformations ,₃ primary ciliary dyskinesia or

splenic malformations.

On examination he was not febrile and not pale. Cardio-

vascular system examination revea_t_hled full volume

pulses, regular with apex beat at 4 right intercostal

space mid clavicular line. He had normal heart rate with

first and second heart sounds. Abdomen was full, soft,

not tender and no organ was palpable. Rectal examina-

tion revealed finger stained with bloody stool.

We describe a case of dextrocardia with situs inversus

totalis in a one day old neonate with neonatal sepsis, the

first case to be reported in Gusau, Zamfara State, Nige-

ria.

2

52

2

Chest X-ray showed normal heart size with apex located

to the right in keeping with dextrocardia. Hepatic

shadow was noted on the left and possible splenic

shadow on the right. There were cervical ribs bilaterally.

Abdominal scan demonstrated liver on the left while the

spleen was on the right. Demonstrable bowel loops were

slightly distended but otherwise normal.

Full blood count showed leucocytosis, random blood

sugar and serum electrolytes were normal. Blood culture

did not yield any growth. Cerebrospinal fluid analysis

was in keeping with bacterial meningitis and gram nega-

tive cocobacilli were seen on microscopy. However,

cerebrospinal fluid culture yielded no growth.

tus. This is due to the fact that dextrocardia with situs

inversus is merely a mirror image of the normal situs

2

solitus, hence any associated cardiac malformations are

usually mirror images of similar malformations in

people with the normal situs solitus. In isolated dextro-

cardia, in which the heart is on the right side without

inversion of the abdominal viscera, malformations of the

heart are almost always invariably present. It has been

postulated that even though the factors responsible for

situs inversus are not clear autosomal recessive gene,

2

maternal diabe₅t_,e₆s, cocaine use and conjoined twinning

are implicated.

A case of dextrocardia with situs inversus occurring

early in life has only been reported in a three day old

neonate . Some cases of dextrocardia have been reported

Fig 1: Radiograph

showing the cardiac

apex pointing to the

right and the hepatic

shadow on the left.

7

in Nigerian children and adults which were mostly inci-

7

dental findings. Ekpe al reported on dextrocardia with

situs inversus co existing with neonatal intestinal ob-

struction in a three day old neonate. A 14 year old child

was incidentally found to have dextrocardia with situs

inversu₈s when he was evaluated for chronic sinusitis at

Enugu.

9

Danbauchi and Alhassan . in Zaria reported two cases

of dextrocardia with situs inversus; a 35-year-old man

that presented for the first time with respiratory symp-

toms but no cardiac symptoms and a 14-year-old who

presented with cardiac symptoms.

Echocardiography was not done as it is not available in

our hospital

Diagnosis of early onset neonatal sepsis with meningitis

was made with background Dextrocardia and situs soli-

tus inversus. He was managed on nil per os, antibiotics,

anticonvulsants and intravenous fluid; however blood

tr_ta_hnsfusion was not required. Bloody stool stopped on

Dextrocardia with situs inversus have also been reported

in cadav₀ers in medic₁a₁l schools during dissection in

1

Nigeria and India. An unusual occurrence of dextro-

cardia with situs inversus have been reported in two

generations of families in India; affecting a fath₂er and

his two sons following consanguineous marriage.

5

day of admission and he remained stable and was

th

discharged by 10 day. Echocardiography in another

centre was not done by the parents as requested and

baby was lost to follow up at age of 3 months despite

adequate counselling of parents.

Conclusion

An incidental finding of dextrocardia with situs inversus

in a newborn is reported and the need for clinicians to

have high index of suspicion is highlighted due to its

asymptomatic nature. Clinicians should look for this

anomaly when reporting or viewing chest x-rays. New-

born babies should have a thorough physical examina-

tion after delivery before discharge to enable early diag-

nosis of congenital anomalies for appropriate referral.

Discussion

Dextrocardia with complete situs inversus is rare,

usually discovered incidentally in otherwise normal sub-

2

jects . Mirror-image dextrocardia with situs₁i_,₂nversus

occurs in 1 in 10, 000 of the general population.

Most neonates delivered in the hospitals are not exam-

ined especially by paediatricians as is the case of this

index baby, to detect such cases in neonatal period. It

may be discovered in infancy because of associated

anomalies but often remains asymptomatic and discov-

Authors contributions

Garba BI and Aminu MS: Conceptualised the case

report.

4

ered by chance in adult life. Many people with this con-

dition are unaware of their unusual anatomy until they

seek medical attention for an unrelated condition. This

Onazi SO, Musa A, Adelakun MB and Sule MB :

Literature review.

1

anomaly may not be diagnosed until late life in some

cases and it is associated w₃ith primary ciliary dyskinesia

and splenic malformations.

Sule MB Ultrasound.

Garba BI and Aminu MS: Manuscript writing

Conflict of interest: None

Funding: None

It has been shown that the incidence of congenital heart

malformations is higher in patients with dextrocardia

and situs inversus than in patients with normal situs soli-

2

53

References

1

.

Tabry IF, Calabrese J, Zammar H

et al. Case report: off-pump total

myocardial revascularization for

dextrocardia and situs inversus.

Heart Surg Forum 2001; 4: 251–

5. Agirbashi M, Hamid R, Jennings

HS, Tiller GE. Situs inversus and

hypertrophic cardiomyopathy in

identical twins. Am J Genetics

2000;91:327-30.

9. Danbauchi SS, Alhassan MA. Case

report: dextrocardia with situs

inversus; two cases presenting

differently. Niger Postgrad Med J

2002; 9: 248–52.

3

.

6. Distefano G, Romeo MG, Grasso

S, Mazonne D, Sciacca P, Mollica

F. Dextrocardia with and without

situsviscerum in 2 siblings. Am J

Med 1987;27:929-34.

7. Ekpe EE, Uwah U, Nyong EE.

Dextrocardia with situs inversus

co existing with neonatal intestinal

obstruction. Port Harcourt Med J

10. Ofusori DA, Okwuonu CU, Ude

RA, Adesanya OA. Dextrocardia

and situsinversustotalis in a Nige-

rian cadaver: a case report of a rare

anomaly. Int J Morphol 2009;27

(3):837-40.

11. Dabbiru R,NarreddySR, Kup-

piliVMM. Dextrocardia with situs

inversus- a case report. Int Jour

Anat Variat 2011;4:88-9.

2

.

Chib P, Grover DN, Shahi BN.

Unusual occurrence of dextrocar-

dia with situs inversus in succeed-

ing generations of a family. J Med

Genetics 1977;14:30-2.

3

.

Nawaz A, Matta H, Hamchou M,

Jacobez A, Trad O, Al Salem AH.

Situsinversusabdominus in association

with congenital duodenal obstruction:

a report of two cases and review of

literature. Pediatr SurgInt 2005; 21:

2

008;2(2):177-80.

8

.

Maduebuchi CJ, Amuche UF,

Uzodinma EC. Situsinversustotalis

in a child with chronic sinusitis.

Open J Paed 2013;3:236-8.

5

89–92.

4

.

Uchenna DI, Jesuorobo DE, An-

yalechi JI. Dextrocardia with situs-

inversustotalis in an adult Nige-

rian: a case report. Am J Med Med-

Sci 2012;2(3):59-61.