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Introduction
Lymphangioma circumscriptum is a lymphatic malformation that is localised to an area of skin, subcutaneous tissue or sometimes muscle. This condition may be present at any age but is usually noted at birth or appears during childhood. The commonest sites are the axillary folds, shoulders, flanks, proximal parts of the limbs and the perineum.1 Very few cases of lymphangioma circumscriptum of the tongue have been reported.2-4 The usual treatment for such cases includes local excision or sclerotherapy.1 We herein describe a case of lymphangioma circumscriptum of the tongue which was successfully managed using intralesional injections of bleomycin alone.
Case report
A 19-year-old man presented to the out-patient department with an 11-year history of a lesion on the dorsum of the tongue. The lesion had grown in size minimally over the years; however, over the past few months, it had started bleeding spontaneously and following trivial trauma. There was no pain, respiratory difficulty or history of macroglossia.
On examination, there was a cluster of small vesicles on the dorsum of the tongue, each about 1-2 mm in size, just proximal to the circumvallate papillae (Figure 1). The majority of vesicles were straw-coloured, with a few being red. No lesions were seen elsewhere in the oral cavity. There was no bleeding on palpation. The tongue was indurated in the area of the lesion.
Fig. 1
(a) Pre-treatment photograph of the lesion in the posterior part of the oral tongue, containing multiple confluent vesicles, some of which were haemorrhagic in nature. (b) Closer view.
A biopsy was performed, which established the diagnosis of lymphangioma circumscriptum.
The patient's routine blood investigations and chest radiography were normal. Magnetic resonance imaging of the tongue was performed. This revealed the presence of an enhancing mass, approximately 3 cm in diameter, involving the dorsum of the tongue, which also had a deeper component involving the tongue musculature.
Sclerotherapy was performed with intralesional bleomycin. A total of three bleomycin injections of 0.75 U/kg each were given, two weeks apart. After each injection, the patient was observed for 24 hours for any adverse reactions such as fever or dyspnoea. No significant side effects were noted. After the first injection, there was a significant...