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Letters to the Editor |
,
Dragan Mici and
Vera Popovif
Institute of Endocrinology, Diabetes, and Diseases of Metabolism University Clinical Center 11000 Belgrade, Yugoslavia
To the editor:
We read with great interest the paper by
Hirshberg et al. (1), who in their 30-yr
experience have not convincingly seen islet hyperplasia that could be
etiologically related to the patient’s hypoglycemia. In contrast,
recently Service et al. (2) have described five
patients with severe postprandial hypoglycemia in whom partial
pancreatic resection guided by calcium stimulation was carried out.
These patients did not have insulinoma. We have recognized two patients
with this syndrome and would like to confirm the existence of this
unique nonneoplastic hyperinsulinemic hypoglycemic syndrome. Patient 1
was a 41-yr-old female who presented with a 4-yr history of
postprandial hypoglycemic episodes. Recently she developed spontaneous
hypoglycemic episodes, particularly during the nighttime. Three years
before admission she had a 72-h fast test, which was negative. Due to
frequent hypoglycemic episodes she entered our Institute for a repeat
72-h fast test, which was positive. Within the first 24 h of
fasting the test was disrupted due to neuroglycopenic symptoms, a
glucose level of 1.8 mmol/L, and an insulin to glucose ratio of 0.52
(insulin, 16.9 mU/L). Patient 2 was a 53-yr-old female with a history
of hypoglycemic episodes occurring both postprandial and during the
nighttime. She had these symptoms 1.5 yr before admission. A 72-h fast
test was negative; the second test was positive. After entering our
Institute a third 72-h fast was interrupted within the first 24 h
with a glucose level of 2.3 mmol/L, an insulin to glucose ratio of 0.47
(insulin, 19.6 mIU/L), and a C-peptide level of 0.29 nmol/L. Computed
tomography imaging, magnetic resonance imaging, and celiac angiography
were negative for a pancreatic insulinoma. Intraoperative ultrasound of
the pancreas was negative. Both patients underwent partial
pancreatectomy (70–75%). The pathological finding was islet cell
hyperplasia and nesidioblastosis. No insulinoma was found.
Immunohistochemistry was as follows: chromogranin A-positive,
insulin-positive cells. Patient 1 was free of hypoglycemic episodes for
7 months postoperatively, after which hypoglycemic episodes reoccurred.
A repeat 72-h fast was interrupted after 10 h of fasting with a
glucose level of 2.0 mmol/L and an insulin level of 9.7 IU/L. Diazoxide
(800 mg/day) in divided doses was ineffective. An 
-glucosidase
inhibitor prior to meals was introduced, but this treatment after a
while was ineffective as well. Thus, this patient underwent
total pancreatectomy, and no insulinoma was detected. She is now
diabetic. Patient 2 did not suffer any hypoglycemic episodes during the
13-month follow up and with a repeat 48-h fast. In conclusion, we
confirm the possibility of the existence of noninsulinoma
pancretogenous hypoglycemia, which was proposed by Service et
al. (2).
Footnotes
e Received December 4, 2000. Address
correspondence to: Mirjana 
umarac-Dumanovi, Institute of
Endocrinology, Diabetes, and Diseases of Metabolism, University
Clinical Center, Dr Subotica 13, 11000 Belgrade, Yugoslavia.
f We thank the two surgeons Prof. Dr. Jankovic and Prof. Dr. Milicevic (Surgical Clinic, Belgrade University Clinical Center, Belgrade, Yugoslavia) for performing the operations and Dr. Cerovic (Pathology Department of VMA, Belgrade, Yugoslavia) for performing immunohistochemical analysis.
References
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