Abstract
Non-islet cell tumor hypoglycemia is a rare paraneoplastic condition caused by an extra-pancreatic tumor. We report a rare case of hypoglycemia caused by a relapsing pelvic solitary fibrous tumor associated with Big-IGF-2 production.A 72-year-old woman was admitted to our hospital because of loss of consciousness and hypoglycemia. She had a history of ovarian solitary fibrous tumor, which has relapsed. From investigation, serum levels of insulin and C-peptide were suppressed; IGF-1 was slightly reduced and IGF-2 was within the normal range, but the IGF-2: IGF-1 ratio was elevated, indicating the presence of Big-IGF-2 secreting non-islet cell tumor. Contrast-enhanced computed tomography (CT) showed a large pelvic mass. She was then submitted to surgical resection of the mass, which histologically proved to be a solitary fibrous tumor. Three months later, she remains asymptomatic.Non-islet cell tumor hypoglycemia should be considered in the differential diagnosis of patients presenting with tumors and recurrent hypoglycemia.
Highlights
Non-islet cell tumor hypoglycemia (NICTH) is a rare cause of refractory hypoglycemia that is associated with large tumors usually of epithelial or mesenchymal origin such as solitary fibrous tumor (SFT) [1,2]
It is caused by tumoral secretion of incompletely processed insulin-like growth factor (IGF)-2, termed Big-IGF-2 or pro-IGF-2 [3,4]
We report a case of hypoglycemia caused by a relapsing pelvic SFT associated with Big-IGF-2 production
Summary
Non-islet cell tumor hypoglycemia (NICTH) is a rare cause of refractory hypoglycemia that is associated with large tumors usually of epithelial or mesenchymal origin such as solitary fibrous tumor (SFT) [1,2]. The patient was submitted to surgical resection of the tumor, which histologically proved to be mesenchymal neoplasia consisting of spindle cells, without evident pleomorphism; no significant mitotic activity or necrosis was identified; immunohistochemical staining showed diffuse positivity for BCL2 and focal positivity for CD34, with negativity for CD99, S100, desmin, caldesmon, and alpha-actin; the proliferative index for Ki-67 was less than 10%. These features were consistent with the relapse of ovarian SFT (Figure 2).
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