(Adults - Diabetes mellitus), #220515767908
Case Report of Non-insulin Dependent Hypoglycaemia in Recurrent Solitary Fibrous Tumour of the Lung.
Syarifah Syahirah Syed Abas, Endocrine Unit, Hospital Serdang; Tharani Naidu, Medical Department, Hospital Serdang; Radhamani Rajakumar, Endocrine Unit, Hospital Serdang; Syahrizan Samsuddin, Endocrine Unit, Hospital Serdang
Email Address :
Phone Number :
Non-islet cell tumor hypoglycemia (NICTH) is a rare paraneoplastic syndrome that secretes insulin growth factor 2 (IGF2), which results in stimulation of the insulin receptor and subsequently induces hypoglycemia. Most commonly, IGF-2–linked hypoglycemia has been observed in patients with solid tumors that are either of mesenchymal or epithelial origin such as hepatocellular carcinomas, adrenocortical tumors, fibrosarcomas and mesotheliomas.
A 69 years old man with underlying hypertension and history of left excision solitary fibrous tumor of left lung 7 years ago presented with recurrent left lung exudative pleural effusion. Chest radiography and CT imaging revealed left lung mass which he underwent US guided biopsy. The histopathological analysis demonstrated recurrence of solitary fibrous tumor of the left lung and he was planned for a debulking surgery. Unfortunately, while waiting for the surgery, he presented with impaired consciousness and serum glucose on admission was 1.4mmol/L. He had no history of hypoglycemia in the past. He developed recurrent hypoglycemia despite on continuous dextrose infusion and dietary intervention.
At the time of hypoglycemia, the levels of insulin and C-peptide were suppressed consistent with non-insulin mediated hypoglycemia. Additionally, IGF1 levels were below the normal range and his renal, liver and adrenal function were normal. He was commenced on oral prednisolone and subsequently underwent median sternotomy and tumour debulking surgery. Histopathogical examination confirmed recurrent left lung solitary fibrous tumor. Hypoglycemia resolved after tumor resection and he was not requiring prednisolone or dextrose infusion. After 1 year of follow up, the patient remained well without any further reported hypoglycemic episodes.
Solitary fibrous tumor is a rare tumor that induces non-islet cell tumor hypoglycemia (NICTH) due to overproduction of IGF2 and interestingly few cases reported episodes of hypoglycemia on recurrence of the tumour rather than at the initial presentation.