CASE PRESENTATION
We report a case of a 6-month old girl referred to the Nairobi hospital
with a massive, progressively growing congenital mass in the oral
cavity. A thorough medical history revealed no co-morbidities with all
vital signs within normal limits. Upon physical examination, the patient
was found to have difficulty in breathing, difficulty feeding and
inability to close the mouth. Intra oral examination revealed a large
swelling involving more than two thirds of the tongue, most of which lay
outside the oral cavity (figure 1) . The lesion was a deep
red-purple colour, soft on palpation, non-tender and normal in
temperature with no appreciable thrills but blanched on application of
pressure. Examination of the surrounding structures was unremarkable.
Magnetic Resonance Imaging (MRI) with gadolinium revealed a
well-circumscribed soft tissue mass measuring 3.9cm x 4.1cm x 5.8cm. It
involved most of the tongue except for the base and extended to both
lateral borders, while inferiorly, involved the entire thickness of the
tongue to its ventral surface. The mass was hypointense on T1-weighting
and was found to be hyperintense on T2- weighting. Multiple low signal
foci were noted which were compatible with flow voids (Figure 2) .
Digital subtraction angiography revealed the feeder vessels to be
anomalous branches of the lingual arteries. These findings, coupled with
blood collection in the interior of the lesion, informed the diagnostic
hypothesis of a congenital lingual hemangioma.
The lesion was monitored closely over a period of 1 year with the
expectation of improvement by involution, however, the lesion showed no
signs of reducing in size and in fact enlarged over this duration
(Figure 3 ). After a multidisciplinary discussion, immediate
embolization of the feeder vessels and subsequent surgical resection of
the lesion was proposed in order to improve the quality of life for the
patient and prevent fatal airway obstruction. Embolization of the
lingual arteries (LA’s) was performed through a transfemoral approach.
Using the bilateral lingual arteriograms, the feeder vessels from the
right and left LA’s were selectively embolized as close to the lesion as
possible using absorbable gelatin sponge particles (Gelfoam, 100 to 200
μm) under fluoroscopic guidance. Post-embolization angiograms of the
LA’s were performed to confirm if all feeder vessels had been occluded
(Figure 4)
One month post embolization, a colour change from deep red/purple to a
lighter shade was noted and the lesion seemed to have reduced slightly
in size slightly as shown by its surface having mucosal folds as opposed
to appearing stretched and shiny prior to embolization (figure
5 ). Despite this, feeding and airway management were still a major
problem for the patient. A month later, naso-tracheal intubation under
endoscopic guidance was performed and a modified key hole surgical
technique was used to reduce the bulk of the lesion (figure 6 ). A
full thickness elliptical wedge incision was made on the dorsum of the
tongue using electrocautery, accompanied by two diverging incisions on
the anterior portion of the tongue. Hemorrhage was controlled by
compression of the posterior aspect of the tongue using moist gauze
after which excess tissue from the central and anterior portion of the
tongue was excised. Suturing was done in various planes using vicryl 3.0
after attaining the best fitting of the tongue into the oral cavity. In
the immediate post-operative period, there was slight swelling of the
tongue which resolved in a few days.
Postoperative hospital stay was uneventful with no medical or surgical
complications. The patient was fed via a nasogastric tube for a period
of 5 days after which it was removed and oral feeding begun. The patient
was discharged on the 8th day after surgery. Histology
of the tissue specimen revealed the diagnosis to be a capillary
hemangioma (figure 7 ). At the final follow up visit, the
tongue had healed entirely and could fit within the mouth comfortably.
The patient showed marked improvement in speech and swallowing
functionality with no other complaints (figure 8 and 9 ).