Insulinoma Manifesting Early Postpartum: Case Report and Review of the Literature

Eske Christiane Gertje, MD; Annemiek M.E. Walenkamp, MD, PhD; Andor W.J.M. Glaudemans, MD; Sander A.J.C. Ijtsma, MD; Klaas Hoogenberg, MD, PhD; Robin P.F. Dullaart, MD, PhD

AACE Clinical Case Reports (Jan 2015)

Insulinoma Manifesting Early Postpartum: Case Report and Review of the Literature

Eske Christiane Gertje, MD,
Annemiek M.E. Walenkamp, MD, PhD,
Andor W.J.M. Glaudemans, MD,
Sander A.J.C. Ijtsma, MD,
Klaas Hoogenberg, MD, PhD,
Robin P.F. Dullaart, MD, PhD

Affiliations

Eske Christiane Gertje, MD: From the Department of Internal Medicine, Martini Hospital Groningen, The Netherlands; Department of Endocrinology, University of Groningen and University Medical Center Groningen, Groningen, The Netherlands.
Annemiek M.E. Walenkamp, MD, PhD: Department of Medical Oncology, University of Groningen and University Medical Center Groningen, Groningen, The Netherlands
Andor W.J.M. Glaudemans, MD: Department of Nuclear Medicine and Molecular Imaging, University of Groningen and University Medical Center Groningen, Groningen, The Netherlands
Sander A.J.C. Ijtsma, MD: Department of Surgery, University of Groningen and University Medical Center Groningen, Groningen, The Netherlands
Klaas Hoogenberg, MD, PhD: From the Department of Internal Medicine, Martini Hospital Groningen, The Netherlands
Robin P.F. Dullaart, MD, PhD: Department of Endocrinology, University of Groningen and University Medical Center Groningen, Groningen, The Netherlands.; Address correspondence to Dr. Robin P.F. Dullaart; Department of Endocrinology, University Medical Center Groningen, PO Box 30001, 9700 RB Groningen, The Netherlands.

Journal volume & issue: Vol. 1, no. 4
pp. e230 – e239

Abstract

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ABSTRACT: Objective: Insulinomas are insulin-producing neuroendocrine tumors. Insulinomas presenting during pregnancy and the early postpartum period are very rare.Methods: A 33-year-old woman with hypoglycemia early postpartum is described. Abdominal computed tomography and endoscopic ultrasound showed 2 lesions of the pancreas. An 11C-5-hydroxy-l-tryptophan (11CHTP) positron emission tomography-computed tomography (PET-CT) scan demonstrated additional uptake in lymph nodes and liver, suggesting malignant insulinoma. Six months after pylorus-preserving pancreatoduodenectomy and excision of liver and lymph node metastases, tumor progression was noted on repeated 11C-HTP PET-CT scans without recurrent hypoglycemia. She was enrolled in a clinical trial and was randomized for dual pan-class I phosphoinositide 3-kinase inhibitor and mammalian target of rapamycin inhibitor treatment, on which there was no tumor progression during 21 months follow-up. A systematic search of PubMed and Medline with the search strategy ‘insulinoma AND pregnancy’ OR ‘insulinoma AND postpartum’ was performed to identify English-, Dutch-, and German-language publications. All publications about (malignant) insulinoma during pregnancy and in the early postpartum period (≤3 months postpartum) were reviewed in addition to the described case report.Results: Insulinoma manifesting during pregnancy or early after delivery has been described in 31 cases, including only 3 cases of malignant insulinoma. Management of malignant insulinoma requires an individualized approach; optimal medical treatment is evolving.Conclusion: The usefulness of 11C-HTP PET-CT in the diagnosis of malignant insulinoma was demonstrated in the present case. Hypoglycemia may particularly become manifest in the postpartum period when insulin action increases consequent to decreased levels of placenta-derived counterregulatory hormones after delivery.Abbreviations: 11C-HTTP 11C-5-hydroxy-l-tryptophan CT computed tomography EUS endoscopic ultrasound MEN-1 multiple endocrine neoplasia type 1 mTOR mammalian target of rapamycin MRI magnetic resonance imaging PET-CT positron emission tomography-computed tomography SRS somatostatin-receptor scintigraphy

Published in AACE Clinical Case Reports

ISSN: 2376-0605 (Online)
Publisher: Elsevier
Country of publisher: United States
LCC subjects: Medicine: Internal medicine: Specialties of internal medicine: Diseases of the endocrine glands. Clinical endocrinology
Website: https://www.journals.elsevier.com/aace-clinical-case-reports

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