Early Improvement of Non-islet Cell Tumor Hypoglycemia by Chemotherapy Using Lenvatinib in a Case with Type 2 Diabetes and Hepatocellular Carcinoma Producing Big IGF-II

Izutsu, Takuma; Ito, Hiroyuki; Izumi, Fujio; Tamura, Hideki; Matsumoto, Suzuko; Antoku, Shinichi; Mori, Taisuke; Goto, Hiroaki

doi:10.2169/internalmedicine.5328-20

Cited by 5 publications

(3 citation statements)

References 25 publications

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“…Having tumor resection is the main therapy for NICTH. 13 Only 45 cases of DPS have been reported from 1979 to 2011, and the incidence is less than 5% as the manifestation of SFT. 5,9 The pathophysiology and mechanism of hypoglycemia in SFT due to paraneoplastic syndrome caused by a release of an incomplete form of IGF-II excreted ectopically by SFT has a high molecular weight, activates insulin receptors, and terminates hepatic gluconeogenesis.…”

Section: Discussionmentioning

confidence: 99%

Doege-Potter Syndrome: A Rare Paraneoplastic Syndrome of Solid Fibrous Tumors Reported in Indonesia

Widjaja

Syarief

Nathania

et al. 2023

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Introduction: Solitary fibrous tumors (SFT) is a rare group of tumor. Even rarer, it is associated with Doege-Potter syndrome (DPS), which manifests as hypoglycemia due to paraneoplastic syndrome. From searching through national reports and articles, this was the first case of DPS in Indonesia. Case: A 60-year-old woman was admitted to the emergency department with dyspnea and unconscious. The patient’s glucose level showed 21mg/dL. A solid mass on the patient’s right lung was shown on the chest X-ray. A previous biopsy concluded that the patient had a solid fibrous tumor with a history of profound hypoglycemia. The patient underwent tumor resection and got a biopsy of the tumor. After resection, the patient showed no sign of hypoglycemia, and the blood glucose level was stable. We concluded that the patient had DPS, a paraneoplastic syndrome associated with the solid fibrous tumor. Conclusion: Performing an adequate examination and giving prompt treatments can increase the patient’s quality of life, even though it is a rare disease and hard to diagnose. The patient’s condition, evaluated from a clinical condition, chest X-ray, and blood glucose, was better than before getting radical resection.

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Section: Discussionmentioning

confidence: 99%

Doege-Potter Syndrome: A Rare Paraneoplastic Syndrome of Solid Fibrous Tumors Reported in Indonesia

Widjaja

Syarief

Nathania

et al. 2023

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“…Moreover, tumor cells did not seem to have enough enzymes to act on pro-IGF-2; thus, the excess pro-IGF-2 competes with IGF-1 and IGF-2 in binding to IGFBP to form a 40–50–kDa binary complex. In that case, the process results in an excess of free IGF-2 ( 37 ) and IGF-1 in the plasma ( 38 , 39 ). The increased IGF-1 causes a negative feedback, leading to a decrease in the upstream GH secretion, followed by a decline in IGF-1 and IGFBP-3 levels.…”

Section: Discussionmentioning

confidence: 99%

Targeting-intratumoral-lactic-acidosis transcatheter-arterial-chemoembolization for non-islet cell tumor hypoglycemia secondary to a liver metastatic solitary fibrous tumor: A case report and literature review

Jin

Zhong²,

Lin³

et al. 2022

Front. Endocrinol.

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Doege–Potter syndrome is a rare paraneoplastic syndrome characterized by non-islet cell tumor hypoglycemia secondary to a solitary fibrous tumor. Doege–Potter syndrome always presents with recurrent fasting hypoglycemia, which can occasionally be life-threatening. The best choice of treatment for Doege–Potter syndrome and solitary fibrous tumor is complete resection. However, when it is unfeasible, local-regional treatment can be used as a palliative therapy. Herein, we report a case of a 46-year-old man with Doege–Potter syndrome that occurred secondary to the liver and pancreatic metastatic solitary fibrous tumors. After he received six rounds of targeting-intratumoral-lactic-acidosis transcatheter-arterial-chemoembolization (TILA-TACE) treatment in our hospital, his hypoglycemia was clinically cured, and the liver metastatic tumor was well controlled. We suggest that TILA-TACE can be considered when curative resection is unfeasible for metastatic liver solitary fibrous tumors to help a patient obtain further surgery opportunities.

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“…Given the lack of clinical recognition of NICTH, its prevalence is likely to be underestimated. IGF-II-induced hypoglycemia has been reported more often in individuals with solid tumors of mesenchymal or epithelial origin, such as hepatocellular carcinomas (HCC), adrenocortical carcinoma, fibrosarcomas, and mesotheliomas [1,3,4,5,6,7,8,9,10]. The primary clinical manifestations of NICTH are recurrent hypoglycemia and a series of hypoglycemic syndromes that may occur before or after tumor detection [11].…”

Section: Introductionmentioning

confidence: 99%

Recurrent Non-islet Cell Tumor Hypoglycemia Secondary to Hepatocellular Carcinoma: Case Report and Literature Review

He,

Gong,

Pan

et al. 2023

Z Gastroenterol

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Rationale Non-islet cell tumor hypoglycemia (NICTH) is a paraneoplastic syndrome caused by tumors other than insulinoma that is primarily due to excessive production of insulin-like growth factor-II (IGF-II). The prevalence of NICTH is likely underestimated because of a lack of clinical recognition. Patient Concerns A 41-year-old male with massive malignant liver tumors presented with recurrent severe hypoglycemia, weight loss, and liver cirrhosis. Diagnosis NICTH related to IGF-II produced by hepatocellular carcinoma was diagnosed based on clinical symptoms, biochemical tests, and elevated IGF-II/IGF-I ratio. Intervention Initial treatment with intravenous glucose and parenteral nutrition showed limited efficacy. Glucocorticoids and recombinant human growth hormone led to progressive improvement in blood glucose levels. Outcome Due to extensive tumor burden and liver failure, surgical resection was not feasible, and the patient ultimately succumbed to refractory hypoglycemia and passed away in two weeks. Lessons Early recognition and diagnosis of NICTH are crucial in patients with recurrent hypoglycemia and large tumors. Surgical resection is the preferred treatment option, but supportive care and pharmacological interventions, such as glucocorticoids and growth hormone, can help manage refractory hypoglycemia. Further research is needed to explore novel treatment options, including anti-IGF-I and -IGF-II neutralizing antibodies.

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Early Improvement of Non-islet Cell Tumor Hypoglycemia by Chemotherapy Using Lenvatinib in a Case with Type 2 Diabetes and Hepatocellular Carcinoma Producing Big IGF-II

Cited by 5 publications

References 25 publications

Doege-Potter Syndrome: A Rare Paraneoplastic Syndrome of Solid Fibrous Tumors Reported in Indonesia

Doege-Potter Syndrome: A Rare Paraneoplastic Syndrome of Solid Fibrous Tumors Reported in Indonesia

Targeting-intratumoral-lactic-acidosis transcatheter-arterial-chemoembolization for non-islet cell tumor hypoglycemia secondary to a liver metastatic solitary fibrous tumor: A case report and literature review

Recurrent Non-islet Cell Tumor Hypoglycemia Secondary to Hepatocellular Carcinoma: Case Report and Literature Review

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