Neoadjuvant Imatinib Therapy for Gastrointestinal Stromal Tumors Associated With Non-islet Cell Tumor Hypoglycemia (NICTH): A Case Report.

Non-islet cell tumor hypoglycemia (NICTH) is a rare paraneoplastic syndrome characterized by insulin-like growth factor-2 (IGF-2) release, often associated with diverse tumor types. Gastrointestinal stromal tumors (GISTs), sarcomatous lesions of the gastrointestinal tract, are rarely associated with NICTH. We present a unique case of a 58-year-old patient diagnosed with a GIST exhibiting recurrent hypoglycemia suggestive of NICTH. Despite normal IGF-2 levels, the IGF-2/IGF-1 ratio supported the NICTH diagnosis, which was confirmed histologically. Imaging revealed a large intraperitoneal mass. Hypoglycemia was managed with high-dose dextrose and hydrocortisone. Treatment with the tyrosine kinase inhibitor, imatinib, was initiated. Surprisingly, imatinib not only reduced the tumor size but also improved hypoglycemia. The study highlights the complexities in managing NICTH and its underlying causes. Current diagnostic limitations, treatment modalities, and unexpected therapeutic responses challenge standard approaches. This emphasizes the need for personalized oncological strategies.

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Human subjects: Consent was obtained or waived by all participants in this study. Conflicts of interest: In compliance with the ICMJE uniform disclosure form, all authors declare the following: Payment/services info: All authors have declared that no financial support was received from any organization for the submitted work. Financial relationships: All authors have declared that they have no financial relationships at present or within the previous three years with any organizations that might have an interest in the submitted work. Other relationships: All authors have declared that there are no other relationships or activities that could appear to have influenced the submitted work.