Insulinoma Manifesting Early Postpartum: Case Report and Review of the Literature
Gertje, Eske Christiane LU
; Walenkamp, Annemiek M.E.
; Glaudemans, Andor W.J.M.
; Ijtsma, Sander A.J.C.
; Hoogenberg, Klaas
and Dullaart, Robin P.F.
(2015)
In AACE Clinical Case Reports
1(4).
p.230-239
- Abstract
- 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... (More)
- 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 (Less)
Please use this url to cite or link to this publication:
https://lup.lub.lu.se/record/5575f148-4854-4261-884c-caa959f118b8
- author
- Gertje, Eske Christiane
LU
; Walenkamp, Annemiek M.E.
; Glaudemans, Andor W.J.M.
; Ijtsma, Sander A.J.C.
; Hoogenberg, Klaas
and Dullaart, Robin P.F.
- publishing date
- 2015
- type
- Contribution to journal
- publication status
- published
- subject
- in
- AACE Clinical Case Reports
- volume
- 1
- issue
- 4
- pages
- 230 - 239
- external identifiers
-
- scopus:85124250782
- ISSN
- 2376-0605
- DOI
- 10.4158/EP14549.CR
- language
- English
- LU publication?
- no
- id
- 5575f148-4854-4261-884c-caa959f118b8
- alternative location
- https://www.sciencedirect.com/science/article/pii/S237606052030345X
- date added to LUP
- 2025-03-06 14:35:30
- date last changed
- 2025-04-04 13:54:02
@article{5575f148-4854-4261-884c-caa959f118b8,
abstract = {{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}},
author = {{Gertje, Eske Christiane and Walenkamp, Annemiek M.E. and Glaudemans, Andor W.J.M. and Ijtsma, Sander A.J.C. and Hoogenberg, Klaas and Dullaart, Robin P.F.}},
issn = {{2376-0605}},
language = {{eng}},
number = {{4}},
pages = {{230--239}},
series = {{AACE Clinical Case Reports}},
title = {{Insulinoma Manifesting Early Postpartum: Case Report and Review of the Literature}},
url = {{http://dx.doi.org/10.4158/EP14549.CR}},
doi = {{10.4158/EP14549.CR}},
volume = {{1}},
year = {{2015}},
}