Abstract
We report the case of a 32-year-old male with adult-onset diffuse nesidioblastosis causing hypoglycemia. Under the tentative diagnosis of insulinoma, localization procedures were carried out but no tumor was found. The presence of an insulinoma in the tail of the pancreas was suggested by selective intra-arterial calcium stimulation with hepatic venous sampling (ASVS). A distal pancreatectomy was performed under the assumed diagnosis of insulinoma in the tail based upon the ASVS. Diffuse nesidioblastosis was diagnosed by histopathological evaluation. During the post-operative course, the patient’s glucose and insulin levels were well controlled and uneventful without any medications or insulin for 7 months.
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Laidlaw GF. Nesidioblastoma, the islet tumor of the pancreas. Am J Pathol. 1938;2:125–34.
Hong R, Choi D-Y, Lim S-C. Hyperinsulinemic hyperglycemia due to diffuse nesidioblastosis in adults: a case report. World J Gastroenterol. 2008;14(1):140–2.
Fajans SS, Vinik AI. Insulin-producing islet cell tumors. Endocrinol Metab Clin North Am. 1989;18(1):45–74.
Van Der Wal BC, De Krijger RR, De Herder WW, Kwekkeboom DJ, van der Ham F, Bonjer HJ. Adult hyperinsulinemic hypoglycemia not caused by an insulinoma: a report of two cases. Virchows Arch. 2000;436(5):481–6.
Harness JK, Geelhoed GW, Thompson NW. Nesidioblastosis in adults. A surgical dilemma. Arch Surg. 1981;116(5):575–80.
Nakagawa A, Ueno K, Ito M, Okamoto S, Uehara K, Ito H, et al. Insulin responses to selective arterial calcium infusion under hyperinsulinemic euglycemia glucose clamps: case studies in adult nesidioblastosis and childhood insulinoma. Endocr J. 2007;54(1):27–33.
Toyomasu Y, Fukuchi M, Yoshida T, Tajima K, Osawa H, Motegi M, et al. Treatment of hyperinsulinemic hypoglycemia due to diffuse nesidioblastosis in adults: a case report. Am Surg. 2009;75:331–4.
Alvarez GC, Faria EN, Beck M, Girardon DT, Machado AC. Laparoscopic spleen-preserving distal pancreatectomy as treatment for nesidioblastosis after gastric bypass surgery. Obes Surg. 2007;17:550–2.
Masayuki F, Isao K, Kiyoshi T, Kazuhiko Y, Kinya H, Masataka T. Nesidioblastosis in an adult: a patient followed up for seven years postoperatively. Endocr Surg. 1999;16(2):145–51.
Arao T, Okada Y, Hirose Y, Tanaka Y. A rare case of adult-onset nesidioblastosis treated successfully with diazoxide. Endocr J. 2006;53(1):95–100.
Kenney B, Tormey CS, Qin L, Sosa JA, Jain D, Neto A. Adult nesidioblastosis. Clinicopathologic correlation between pre-operative selective arterial calcium stimulation studies and post-operative pathologic findings. J Pancreas. 2008;9(4):504–11.
Service G, Thompson GB, Service FJ, Andrews JC, Collazo-Clavell ML, Lloyd RV. Hyperinsulinemic hypoglycemia with nesidioblastosis after gastric-bypass surgery. N Engl J Med. 2005;353(3):249–54.
Anlauf M, Wienben D, Perren A, Sipos B, Komminoth P, Raffel A, et al. Persistent hyperinsulinemic hypoglycemia in 15 adults with diffuse nesidioblastosis: diagnostic criteria, incidence and characterization of β cell changes. Am J Pathol. 2005;29:524–33.
Raffel A, Krausch M, Anlauf M, Wieben D, Braunstein S, Klöppel G, et al. Diffuse nesidioblastosis as a cause of hyperinsulinemic hypoglycemia in adults: a diagnostic and therapeutic challenge. Surgery. 2007;141:179–84.
Boulanger C, Vezzosi D, Bennet A, Lorenzini F, Fauvel J, Caron P, et al. Normal pregnancy in a woman with nesidioblastosis treated with somatostatin analog octreotide. J Endocrinol Invest. 2004;27:465–70.
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Maeda, Y., Yokoyama, K., Takeda, K. et al. Adult-onset diffuse nesidioblastosis causing hypoglycemia. Clin J Gastroenterol 6, 50–54 (2013). https://doi.org/10.1007/s12328-012-0335-0
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DOI: https://doi.org/10.1007/s12328-012-0335-0