When Hypoglycemia Speaks Louder Than the Chest: A Decade-Late Recurrence of IGF-2-Mediated Non-Islet Cell Tumor Hypoglycemia
https://doi.org/10.15605/jafes.041.S1
- Author:
Li Li Kwan
1
;
Deviga Lachumanan
2
Author Information
1. Department of Internal Medicine, Hospital Port Dickson
2. Endocrinology Unit, Department of Internal Medicine, Hospital Port Dickson
- Publication Type:Journal Article
- MeSH:
Insulin-Like Growth Factor II;
Hypoglycemia;
Neoplasms
- From:
Journal of the ASEAN Federation of Endocrine Societies
2026;41(S1):67-68
- CountryPhilippines
- Language:English
-
Abstract:
Introduction:Non-islet cell tumor hypoglycemia (NICTH) is a rare
paraneoplastic syndrome caused by tumor secretion of
insulin-like growth factor-2 (IGF-2), leading to recurrent
hypoinsulinemic hypoglycemia. It is most commonly
associated with large mesenchymal tumors, such as solitary fibrous tumor, particularly those arising from the pleura
or lungs. This phenomenon, also known as Doege–Potter
syndrome, may precede tumor detection or signal tumor
recurrence. We report a striking case of late malignant
recurrence presenting solely with hypoglycemia after
a decade of remission.
Case:A 68-year-old female was initially presented in 2016
with respiratory symptoms and recurrent symptomatic
fasting and post-prandial hypoglycemia, and was found
to have a large left upper lobe mass. Tumor resection in
2017 resulted in complete resolution of hypoglycemia.
She remained asymptomatic for several years. In
2023, she developed recurrent hypoglycemia without
respiratory or constitutional symptoms. Biochemical
evaluation demonstrated hypoinsulinemic hypoglycemia
with suppressed insulin and C-peptide, low IGF-1, and
markedly elevated IGF-2, resulting in an IGF-2:IGF-1 ratio
of 25, consistent with IGF-2-mediated NICTH. Computed
tomography imaging revealed a large left thoracic mass
with invasion into the intercostal muscles and diaphragm
with extension toward the stomach, associated with
contralateral lung nodules and possible liver metastases,
suggesting recurrent malignant disease. Hypoglycemia
improved with glucocorticoid therapy, and the patient
was referred for oncological assessment. Despite initiation
of systemic chemotherapy, her disease progressed and she
succumbed during treatment.
Conclusion:This case highlights several important lessons: Firstly,
recurrent hypoinsulinemic hypoglycemia warrants
evaluation for NICTH even in the absence of tumor-related
symptoms. Secondly, solitary fibrous tumors may recur
or undergo malignant transformation after prolonged
disease-free intervals; and glucocorticoids provide effective
metabolic control but do not alter oncologic prognosis.
Long-term surveillance should be considered in patients
with prior solitary fibrous tumors due to the risk of delayed
recurrence and paraneoplastic complications.
- Full text:2026080316533294176EP_A088.pdf
