Letter to the Editor
Hormone
Research
Horm Res 1997;48:135-136
Michael Stumvoll a
Andreas Pritsche3
Andreas Pickertb
Dietrich Overkampa
Features of Malignancy in a
Benign Pheochromocytoma
Medizinische Universitätsklinik. Tiibingcn.
und
Städtisches Klinikum, Heilbronn,
Deutschland
Pheochromocytomas are rare conditions
with a prevalence of 1-2/100,000 in the gen
eral population and 1/1,000 hypertensive
subjects [ 1 ]. 10% of pheochromocytomas arc
malignant and various attempts have been
made to find useful prognostic indicators of
malignancy. In general, increased plasma or
urine dopamine concentrations or increased
homovanillic acid excretion and lack of 131-
methyliodo-benzylguanidinc uptake have
been associated with malignancy [2], How
ever, to date no specific metabolic, radio-
logic or histopathologic features of either be
nign or malignant pheochromocytomas al
lowing the safe diagnosis of one or the other
have been identified. The diagnosis of malig
nant pheochromocytoma can be made only
in the presence of local tissue invasion or
distant métastasés. We present a benign
pheochromocytoma exhibiting several fea
tures suggestive of malignant disease.
A 20-year-o!d patient was referred to hos
pital with a 2-year history of paroxysmal pal
pitations most recently accompanied by se
vere headache and flushing. Latest blood
pressure readings were persistently above
200 mm Hg systolic and 120 mm Hg diastol
ic. In addition, an increased dislike of sweets
together with exaggerated salt-hunger was re
ported. Except for hypertension the physical
examination was unrevealing. Abdominal
ultrasound showed a left adrenal mass which
was confirmed by CT scan but undetectable
by methyliodo-benzylguanidine scanning.
Laboratory investigations performed in
the appropriate manner revealed 10-fold ele
vated plasma and urinary' norepinephrine
concentrations and 3- to 4-fold elevated plas
ma and urine dopamine levels as shown in
table 1. Neuron-specific enolase was at the
upper limit of normal (12.9 pg/1). Resting
scrum aldosterone of 459 pg/ml (normal
range <160) and plasma renin activity of
7.98 ng/ml/h (normal range < 1.6) were in
creased, while prolactin, growth hormone,
thyroid function tests, calcitonin, adrenocor-
ticotropin and cortisol were all normal.
The patient underwent left adrenalecto
my and thorough abdominal exploration.
With the adrenal capsule intact no signs of
local tissue invasion by the tumor were de
tected. Microscopy confirmed the diagnosis
of pheochromocytoma but showed neither
angioinvasive nor capsule penetration. Post-
operatively, blood pressure and catechol
amine levels returned to normal. During the
follow-up period of 55 months, the patient
has led a normal life without clinical symp
toms of pheochromocytoma or elevated
urine or plasma catecholamines (table 1).
The intraoperative findings and the his
tology report in conjunction with a 5-year
follow-up without recurrence of symptoms
or abnormal catecholamines allowed us to
establish the diagnosis of a benign pheochro
mocytoma in this patient. Preopcratively,
however, several findings were indicative of
malignant pheochromocytoma.
Young age at presentation has generally
been associated with an increased likelihood
of malignancy [3], Futhermore, although
norepinephrine was the predominantly ele-
KARGER
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This article is also accessible online at:
http://BioMedNet.com/karger
Dr. Michael Stumvoll
Medizinische Universitätsklinik
Otfried-Müller-Strasse 10
D-72076 Tübingen (Germany)
Tel. (7071) 2982711. Fax (7071 ) 298278<i