Retrospective case review of pyriform sinus fistulae of third branchial arch origin commonly presenting as acute suppurative thyroiditis in children.

OBJECTIVE: Abnormalities of the third branchial arch are less common than those of the second arch and usually present with left thyroid lobe inflammation. This paper describes 15 cases of pyriform sinus fistulae of third branchial arch origin usually presenting as recurrent thyroid abscess on the left side. METHOD: A retrospective review of 15 cases of third arch fistulae managed 2000 and 2008, diagnosed based on histopathology and radiological evidence of a fistulous tract, and treated with fistulectomy with left hemithyroidectomy. RESULTS: All patients (six boys and nine girls, aged three to 15 years) presented with recurrent low neck inflammation. Pre-operative ultrasound, computed tomography fistulography and barium swallow demonstrated a third arch fistulous tract, left-sided in all cases. The fistula was detected intra-operatively and pathologically in all cases. Surgery (successful in all cases) emphasised complete recurrent laryngeal nerve and ipsilateral pyriform sinus exposure, to facilitate tract excision, with left hemithyroidectomy. There was no recurrence over three to five years' follow up. CONCLUSION: Paediatric recurrent low neck inflammatory episodes, due to thyroidal abscess, especially left-sided, should raise suspicion of pyriform sinus fistulae.

Simultaneous occurrence of a thyroglossal duct cyst and a lingual thyroid in the absence of an orthotopic thyroid gland.

OBJECTIVE: We report an extremely rare case of the simultaneous occurrence of a thyroglossal duct cyst and a lingual thyroid in the absence of an orthotopic thyroid gland, in a seven-year-old girl from South India. METHOD: Case report and a review of the English language literature on the subject. RESULTS: The patient presented with a mass on the tongue that had been present for three years, and an anterior neck swelling that had been present for two years. Examination revealed a midline, pinkish, firm mass present on the posterior one-third of the tongue. The neck showed a midline cystic swelling in the infrahyoid position. Radiological imaging confirmed the clinical findings, revealing the absence of her thyroid gland in the normal location. Sistrunk's procedure was performed leaving behind a lingual thyroid. At 13-month follow up, the patient was euthyroid with no recurrence. CONCLUSION: To our knowledge the association of a lingual thyroid and a thyroglossal cyst has only been reported once in the literature. The presence of a lingual thyroid in the absence of a normally located thyroid gland or functioning thyroid tissue along the thyroglossal tract, confirmed by radionuclide and computed tomography imaging, may indicate the failure of the normal descent of the thyroid gland during embryonic development. This probable absence of the descent of the thyroid raises questions regarding the origin of thyroglossal duct cysts.

Rhinosporidiosis of the upper airways and trachea.

OBJECTIVE: We report an extremely rare case of rhinosporidiosis with involvement of both larynx and trachea, together with coexisting nasal, nasopharyngeal and oropharyngeal lesions, in a 32-year-old man. METHOD: Case report and review of the world literature concerning laryngotracheal and disseminated rhinosporidiosis. RESULTS: A 32-year-old, South Indian man presented with a nasal mass of four years' duration, with progressive hoarseness for one year. Strawberry-like rhinosporidial masses were seen in both nasal cavities. Fibre-optic laryngoscopic examination revealed reddish masses with whitish surface specks, involving the glottis, subglottis and trachea. Computed tomography revealed soft tissue opacities involving both nasal cavities and the nasopharynx and extending to the oropharynx, with involvement of the larynx and trachea. Direct laryngoscopy and rigid bronchoscopy guided excision of the laryngeal and tracheal lesions was performed. CONCLUSION: Rhinosporidiosis is a chronic, granulomatous disease which usually affects the mucous membranes of the nose and nasopharynx. Lower dissemination into the trachea is extremely rare. Laryngotracheal involvement poses many diagnostic and therapeutic challenges, due to the potential risk of bleeding and aspiration. In the presented case, local spread of rhinosporidiosis, due to direct spillage of spores from the nasopharynx into the larynx during episodes of bleeding or previous surgery, may have resulted in laryngotracheal involvement, although systemic spread cannot be excluded.

Complete congenital third branchial fistula with left-sided, recurrent, suppurative thyroiditis.

OBJECTIVE: We report an extremely rare case of a complete congenital third branchial arch fistula in a nine-year-old boy. METHOD: A case report and a review of the English literature concerning third branchial arch fistula of congenital origin are presented. RESULTS: A nine-year-old boy presented with a history of a small opening in the middle third of the anterior neck since birth, with recurrent surrounding swelling. There was no history of surgical drainage or spontaneous rupture. Computed tomography with contrast injection into the external cervical opening revealed a patent tract from the neck skin to the base of the pyriform sinus. Complete excision of the tract up to the pyriform sinus with left hemithyroidectomy was performed. Follow up at 22 months showed no recurrence. CONCLUSION: To our knowledge, this case represents a very rare occurrence of the congenital variety of complete third branchial arch fistula at an unusual site. This case indicates that third branchial arch fistula can be complete, and may present in the anterior neck, an unusual site. In such cases, computed tomography fistulography and injection of dye into the pyriform sinus enables intra-operative delineation of the tract.

Disseminated rhinosporidiosis with unusual involvement of the nail apparatus.

Rhinosporidiosis is an uncommon chronic granulomatous disease caused by Rhinosporidium seeberi. It primarily affects the nasal mucosa and the nasopharynx. The disease may disseminate to the skin, larynx, trachea, genitalia, bones and even the internal organs. Although simultaneous onset of nasal and disseminated lesions has been reported, dissemination usually occurs several years after the onset of nasal lesions. We report a rare case of disseminated rhinosporidiosis involving both nares, the nasal mucosa, nasopharynx, oral cavity and oropharynx, as well as widespread cutaneous lesions over the trunk and the limbs with nail involvement. This last feature has not, to our knowledge, been previously reported.