Background. To our knowledge, only three cases with ectopic submandibu
lar thyroid have been reported. No patient with simultaneous presence
of ectopic submandibular thyroid and normal thyroid gland has been rep
orted. Literature on this topic is reviewed. Methods. A 34-year-old wo
man with a longstanding asymptomatic right submandibular mass was eval
uated by ecography, computer tomography scan, and fine-needle aspirati
on. Literature review was performed using EBSCO-Medline Results. The m
ass was hard, nontender, and not movable with swallowing. A fine-needl
e aspiration revealed hyperplastic thyroid tissue. A second ecographic
-guided fine-needle aspiration confirmed cytologically ectopic thyroid
tissue. A computer tomography scan showed a calcified mass with cysti
c areas beneath and medial to the submandibular gland. Rests of thyroi
d tissue lateral to the trajectory of thyroglossal duct were seen. Thy
roid gland was atrophic. No metastatic lymph nodes were noted. Total s
erum thyroxine and triiodothyronine were normal. Basal and TRH-stimula
ted serum thyrotropin was suppressed. A Tc-99 scintigraphy disclosed a
nodular uptake of tracer in the submandibular area, and no uptake by
normal thyroid. Conclusions. We describe the first case, to our knowle
dge, with lateral multiple aberrant thyroid tissue and hypoplastic thy
roid gland. (C) 1996 John Wiley & Sons, Inc.