Abstract

A 37 year-old woman was referred to our hospital complaining of dyspnea and bilateral swelling of the submandibular region which were developed 6 months ago. Although the submandibular glands seemed to be diffusely enlarged, with suspicion to amyloidosis, an incisional biopsy of one gland stained with Congo red was performed but normal glandular tissue was found. As a result, abdominal fat pad biopsy was done to confirm the diagnosis. Further review of MRI data indicated that the submandibular swelling was due to down-shifting of the submandibular glands brought about by enlargement of the tongue and floor-of-mouth muscles. Abdominal fat pad biopsy showed deposition of amyloid fibrils. Findings were consistent with amyloidosis. In this case the most remarkable findings were swelling in submandibular area secondary to down-shifting of the submandibular glands and macroglossia which had partially compromised the airway. Abdominal fat pad biopsy is easier and less invasive than other methods and shows amyloid fibril deposition in 70 to 80% of cases.

Key words: Amyloidosis, macroglossia, submandibular gland.