Severe Hypoglycemia Caused by Recurrent Sarcomatoid Carcinoma in the Pelvic Cavity: A Case Report

Abstract

Nonislet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome characterized by persistent, severe hypoglycemia in different tumor types of mesochymal or epithelial origin; however, NICTH is infrequently induced by sarcomatoid carcinoma (SC). Despite some sarcomatoid and epithelioid characteristics in few cases of malignancies from epithelium, NICTH induced by recurrent SC in pelvic cavity in this report is extremely rare.We report a case in which NICTH caused by recurrence and pulmonary metastases from SC in the pelvic cavity, and the computed tomography scan revealed multiple pelvic masses and multiple large masses in the pulmonary fields. During the treatment of intestinal obstruction, the patient presented paroxysmal loss of consciousness and sweating. Her glucose even reached 1.22 mmol/L while the serum glycosylated hemoglobin was normal and previous history of diabetes or use of oral hypoglycemic agents and insulin denied.The laboratory examination showed that the low level of insulin, C-peptide, and growth hormone levels in the course of hypoglycemic episodes suggesting to the diagnosis of hypoglycemia induced by nonislet cell tumor, and the decreased levels of insulin-like growth factor (IGF)-I and IGFBP3 and the high expression of big IGF-II in the serum further confirmed the diagnosis of NICTH. Because of the widely pelvic recurrence and pulmonary metastases were unresected, the patient was discharged from the hospital after 2 weeks treatment with dexamethasone and glucose and unfortunately died 1 week later.NICTH caused by SC in the pelvic cavity is extremely rare case in clinical. The aim of this report was to present the importance to examine big IGF-II expression in patient's serum in order to reach the diagnosis of NICTH in cases of intractable cancer-associated hypoglycemia.

FIGURE 1

Diagnosis of PCSC. A: Abdominal MRI scan revealed a large (30 × 25 × 15 cm) heterogeneous pelvic mass and seemed to be widely adherent to rectum, posterior vaginal wall, and partial pelvic wall. B: Immunohistochemical results identified the diagnosis of sarcomatoid carcinoma/undifferentiated carcinoma (positive for PCK, weakly positive for EMA and CK7 and negative for CD125, WT1, D2-40, HMB-45, Caldesmon, Desmin, S100, DOG1, and CD117). MRI = magnetic resonance imaging; PCSC = SC in the pelvic cavity.

FIGURE 2

Diagnosis of NICTH. A: Abdominal CT scan revealed the pelvic recurrence and pulmonary metastases. B: Lane 3 showed that IGF-II was mainly detected as high molecular weight band in the sample from the patient, and lanes 2 and 3 showed the concentration of total IGF-II (ie, mature and big) of the patient's serum was higher than that in the control subject. CT = computed tomography; IGF-II = insulin-like growth factor II; NICTH = nonislet cell tumor hypoglycemia.