Evidence map›Paper›PMID 41641310›Full record

ArticleAACE endocrinology and diabetes

Resolution of Refractory Nonislet Cell Tumor Hypoglycemia Following Resection of an Abdominal Solitary Fibrous Tumor.

Maxwell Horowitz, Selin Kurt, Sonali Lanjewar, John McAuliffe, Noah Bloomgarden, Simona Stefan

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Article in AACE endocrinology and diabetes. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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5 · Who and what money

Authors and funding

6 authors.

Maxwell HorowitzDepartment of Endocrinology, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.
Selin KurtDepartment of Pathology, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.
Sonali LanjewarDepartment of Pathology, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.
John McAuliffeDepartment of Surgery, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.
Noah BloomgardenDepartment of Endocrinology, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.
Simona StefanDepartment of Endocrinology, Albert Einstein College of Medicine and Montefiore Medical Center, Bronx, New York.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Introduction: Solitary fibrous tumors are exceptionally rare mesenchymal tumors that can present with paraneoplastic syndromes, the most common being nonislet cell tumor hypoglycemia. We describe a case of refractory hypoglycemia that resolved following surgical resection of a large abdominal solitary fibrous tumor. Case Discussion: A man in his 70s initially presented with abdominal pain and distention. On exam, a firm, nontender, and mobile mass was palpable in the upper abdomen, extending toward the midline. Imaging revealed a 26 cm abdominal mass, and biopsy confirmed solitary fibrous tumor. Two weeks after discharge, he experienced 3 consecutive nights of symptomatic hypoglycemia. Laboratory evaluation showed serum glucose of 27 mg/dL (70-140 mg/dL), suppressed c-peptide 0.2 ng/dL (1.00-4.00 ng/ml), insulin-like growth factor (IGF)-1 of 36 ng/dL (5-34 ng/dL) and IGF-2 of 241 ng/dL (267-616 ng/dL), with an IGF-2:IGF-1 ratio of 6.7:1. The patient underwent surgical resection, and immediately after hypoglycemia was resolved. Discussion: Nonislet cell tumor hypoglycemia is a rare paraneoplastic syndrome caused by ectopic secretion of IGF-2 and it is characterized by severe, recurrent, and treatment-resistant hypoglycemia. It is rare, and diagnosis requires exclusion of more common causes. Definitive management is surgical resection, which typically results in immediate resolution. Conclusion: This case highlights the importance of recognizing paraneoplastic syndrome in patients with unexplained hypoglycemia and underscores the curative potential of timely surgical resection.

Indexed as

IGF-2nonislet cell tumor hypoglycemiaparaneoplastic syndromerefractory hypoglycemiasolitary fibrous tumor

Identifiers

PMID41641310
PMCPMC12866086

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