Management challenges and outcome of congenital giant intra lingual fibroma in a 2 year old boy.html

Niger J Paed 2015; 42 (1): 76 –77

CASE REPORT

Osifo OD

Management challenges and

Akpoduado DD

outcome of congenital giant

intra-lingual fibroma in a 2-year

old boy.

DOI:http://dx.doi.org/10.4314/njp.v42i1,18

Accepted:

Abstract: Oral fibromas are often

culty was diagnosed in a two-year-

acquired painless benign slowly

old boy. The case is reported to

Osifo OD

(

)

growing tumour usually seen on

draw attention to this rarely con-

Paediatric Surgery Unit, Department of

the buccal mucosa along the plane

genital tumour found in an unusual

Surgery,

of occlusion of the maxillary and

location and highlights the chal-

lenges of surgical management in

Akpoduado DD

mandibular teeth. Congenital intra

Department of Anaesthesia,

-lingual fibroma is rare and not

a financially constrained African

University of Benin Teaching Hospital,

much has been reported on it in

setting.

Benin City, Nigeria.

literature. A giant intra-lingual

Email: Davidosifo4@Gmail.Com,

fibroma which was present at

Key

words:

Intra-lingual, Fi-

Leadekso@Yahoo.Com

birth that completely filled and

broma, Management challenges,

protrudes

out

the

mouth

Outcome.

thereby resulting in feeding diffi-

Introduction

Fig 1: A two-yr-old

boy with huge intra-

Intra-oral tumours are very common pathology in chil-

lingual mass noticed as

dren and the majority are congenital benign lesions

a small lump at birth.

which can be located in any region of the mouth. The

floor of the mouth is the commonest site for the fre-

quently diagnosed paediatric oral tumours such as

ranula, haemagioma, lymphangioma, dermoid cyst, mu-

cous retention cyst and calculi.

1-3

A purely intra-lingual

tumour is very rare in children and there are very few

documentation on tumour occurrence in this location.

Ingestion of liquid feeds was accomplished by the par-

Similarly, although fibroma can be seen in any part of

ents insinuating a straw between the cheek and the mas-

the body, including oral cavity, an intra-lingual location

sive tongue. Financial constraints and ignorance resulted

is rare.

4,5

They are painless benign slowly growing tu-

in the parents seeking help from quacks and traditional

mour that hardly produce life threatening symptoms. A

health care providers without improvement.

giant intra-lingual fibroma present from birth that grew

However, apart from the lingual mass the child was

and could not be contained within oral cavity but com-

clinically stable, looking well fed and active on arrival.

pletely filled and protrudes out of the mouth thereby

The tongue was grossly enlarged with a firm non tender

resulting in feeding and anaesthetic difficulties was di-

and non mobile smooth mass within its substance.

agnosed in a two-year-old boy.

Haematocrit was 11.5mg%, other blood parameters and

The case is reported to draw attention to this rarely con-

chemistry were within normal ranges and lung fields

genital tumour found in an unusual location and high-

were clear on plain chest radiograph. He was booked for

light the challenges of surgical management in a finan-

excision biopsy under general anaesthesia but the anaes-

cially constrained African setting.

thetist assessment showed that there was no space to

insinuate laryngoscope for endotracheal intubation to

Case

secure the airway during surgery. The parents were

counselled for tracheostomy but declined consent due to

Master M.W, a two-year-old boy, is the last of three

fear of its outcome and inability to pay the additional

children in a monogamous family. He was brought to

bills.

the hospital on account of swelling of the tongue which

was noticed as a small intra-lingual lump at birth. The

Consequently, the mass was excised with anaesthesia

swelling increased gradually and at the age of one year

provided using intravenous

ketamine 2mg/kg, mida-

had filled and protruded out of the month as the oral

zolam 1mg/kg, atropine 1mg/kg to dry up secretion and

cavity could no longer contain the massive tongue (fig.

simultaneous local infiltration of the tongue with 1%

1). There was difficulties closing the mouth, talking and

xylocaine in adrenaline 1:1000 6mg/kg. He was placed

eating which was associated with drooling of saliva.

on a left lateral position with continuous suctioning of

the operating field during the operation which lasted 25

operations performed with parenteral ketamine and si-

minutes. A 6 x 8 cm intra-lingual mass weighing 250g,

multaneous local infiltration of operation site with xylo-

which was histopathologically confirmed as a lingual

caine in adrenaline. These operations were performed in

fibroma, was well circumscribed and easily enucleated

children older than a month with lesions located outside

(figs. 2). Post operative course was uneventful following

the mouth, and the duration of each operation lasted less

thrice daily oral warm saline lavage, prophylatic men-

than one hour. In neonates and older children with intra-

tronidazole and paracetamol which provided adequate

oral lesions, however, securing the airway to prevent

analgesia. Oral feed was commenced and tolerated 4

aspiration during operation is of paramout importance.

hours after operation and he was discharged to out pa-

Endotracheal intubation, nasotracheal intubation, laryn-

tient follow up on the evening of surgery. Redundant

geal mass, or creation of tracheostomy where these are

not possible, are advised in such cases

9,10

lingual tissues resolved within two weeks of follow up

. The huge intra

and he has been doing well.

-lingual mass which filled the mouth could not allow

space for endotracheal intabution or placement of laryn-

Fig 2: The resected congeni-

geal mass in this child. Coupled with financial con-

tal intra-lingual mass which

straints and ignorance which resulted in parents refussal

weighed 250g and was

to consent to tracheostomy posed major challenges in

histopathologically con-

surgical management of index case.

firmed as fibroma.

As a last resort, anaesthesia was provided using intrave-

nous ketamine, midazolam, atropine which reduced oral

secretion, and simultaneous local infiltration of the

tongue with xylocaine in adrenaline . Aspiration during

the operation was further prevented by placing the child

on a left lateral position with continuous suctioning of

Discussion

the operating field throughout the operation. The suc-

Fibromas are said to represent reactive focal fibrous

cessful outcome recorded in excising this huge intra-

hyperplasia due to trauma or local irritation which can-

lingual mass with this method may have been possible

not account for the occurrence of this lesion in this case.

because the mass was well circumscribed and easily

It was reported

5-7

to be seen most often on the buccal

enucleated within 25 minutes. A less well defined mass

mucosa along the plane of occlusion of the maxillary

with poorly developed plane for disection that can result

and mandibular teeth unlike in this patient where it was

in longer duration of surgery may not have given com-

parably good outcome as recorded in this case .

found within and grossly enlarged the tongue. Also in

the index case, the fibroma was present at birth and di-

agnosed in a two years child which supports a congeni-

Congenital huge intra-lingual fibroma which completely

tal rather than acquire origin. Oral fibromas which are

filled and protrudes out of the mouth thereby resulting in

either sessile or pedunculated are often submucosal in

feeding and endotracheal intubation difficulties was suc-

location. They are painless and are usually seen in older

cessfully excised with excellent outcome in a two-year-

patients following repeated abrasions from chewing and

old boy. The case is reported to draw attention to this

chemical irritation of oral mucosa .

6-8

rarely congenital tumour found in an unusual location

Earlier authors recorded excellent results in paediatric

and highlighted the challenges of surgical management

in a financially constrained African setting.

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