Sporadic adrenomedullary hyperplasia (AMH) is characterized by a clini
cal history of hypertension, increased plasma and/or urinary catechola
mine levels and histomorphometric evidence of increased adrenal medull
ary mass in the absence of MEN 2 syndrome. The case of a 42-year-old f
emale patient is reported who presented with typical clinical and labo
ratory findings of episodic hypertension and elevated plasma and urina
ry catecholamines. Sonography and computed tomography revealed no abno
rmality, but I-131-metaiodobenzylguanidine (I-131-MIBG) scintigraphy s
howed increased uptake in the right adrenal. Transabdominal unilateral
adrenalectomy was performed, The right adrenal gland was macroscopica
lly inconspicuous. Upon histomorphometry, however, an increased adrena
l medullary cell mass was shown, thus confirming AMH. Two years follow
ing surgery the patient is asymptomatic and normotensive.