Mechanisms and Management of Non-islet Cell Tumour Hypoglycaemia in Gastrointestinal Stromal Tumour: Case Report and a Review of Published Studies

Davda, Reena; Seddon, Beatrice

doi:10.1016/j.clon.2006.12.008

Cited by 25 publications

(17 citation statements)

References 28 publications

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“…When these active complexes or unbound IGF-II bind to insulin and IGF receptors in the liver and peripheral tissues there is decreased release of glucose into circulation and increased peripheral glucose metabolism resulting in hypoglycemia. Although plasma IGF-II levels were within normal range, the ratio of IGF-II to IGF-I was elevated at 9.6:1, consistent with NICTH [20]. Given the humorally mediated symptoms related to these metastases, an aggressive approach for cytoreduction is warranted.…”

Section: Discussionmentioning

confidence: 93%

Multidisciplinary Management of Metastatic Hemangiopericytoma Presenting with Hypoglycemia

Pv¹

2017

Ann Hematol Oncol

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Section: Discussionmentioning

confidence: 93%

Multidisciplinary Management of Metastatic Hemangiopericytoma Presenting with Hypoglycemia

Pv¹

2017

Ann Hematol Oncol

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“…Under normal conditions, the liver produces IGF-II, in a growth hormone-independent manner, and IGF-II forms a biologically inactive ternary complex with IGF binding proteins (IGFBP), the most common of which are IGFBP3 and acid labile subunit (ALS) [12, 13]. Tumor-produced IGF-II has an equal affinity for IGFBPs compared to IGFI and II.…”

Section: Discussionmentioning

confidence: 99%

A case of extragastrointestinal stromal tumor complicated by severe hypoglycemia: a unique presentation of a rare tumor

2016

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BackgroundNon-Islet Cell Tumor Hypoglycemia (NICTH) is a rare paraneoplastic cause of hypoglycemia arising from excess tumor production of insulin-like growth factor. The objective of this report is to describe an unusual case of Extragastrointestinal Stromal Tumor (EGIST) associated NICTH.Case presentationA 64 year-old African female was brought to the emergency room with a 1-month history of recurrent syncope, weight loss, and abdominal bloating. Serum blood glucose was discovered to 39 mg/dL, when insulin, proinsulin, and C-peptide were suppressed. Computed tomography scan revealed a diffuse extraintestinal metastatic disease process, and a biopsy confirmed the diagnosis of an Extragastrointestinal Stromal Tumor (EGIST). IGF-I and II levels were 27 ng/ml and 262 ng/ml respectively, and the ratio of IGF-II to IGF-I was calculated as 9.7:1, suggestive of IGF-II-mediated NICTH. Acutely, the patient’s hypoglycemia resolved with dextrose and glucagon infusion. Long-term euglycemia was achieved with prednisone and imatinib therapy.ConclusionsNICTH should be considered when hypoglycemia occurs in the setting of low serum insulin levels. Whereas definitive treatment of EGIST involves surgical resection, immunotherapy with tyrosine kinase inhibitors and corticosteroids have been shown to alleviate hypoglycemia in cases where surgery is delayed or not feasible.

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“…En los pacientes con hipoglicemia paraneoplásica se ha encontrado niveles plasmáticos de insulina baja, niveles elevados de 'big IGF-II' ( factor incompleto de crecimiento similar a la insulina tipo II), producto de un error en el procesamiento del IGF II, y aumento de su biodisponibilidad por una distribución alterada entre sus proteínas transportadoras (24)(25)(26) . El diagnóstico de hipoglicemia paraneoplásica se vio facilitado por los antecedentes negativos de diabetes mellitus, el no uso de fármacos o drogas (hipoglicemiantes orales, insulina, ácido acetil salicílico, alcohol, entre otros), ausencia de infecciones u otras enfermedades metabólicas, nutricionales, y por la presencia de derrame pericárdico masivo.…”

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Síndrome de Doege Potter y derrame pericárdico, como manifestaciones iniciales de neoplasia pulmonar

2013

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Síndrome de Doege Potter y derrame pericárdico, como manifestaciones iniciales de neoplasia pulmonarResumen Comunicamos el caso de un paciente de 52 años que ingresa a nuestro hospital por presentar derrame pericárdico masivo y manifestaciones de hipoglicemia de manera repetitiva, sin antecedentes de tabaquismo, diabetes mellitus, ni consumo de fármacos. Fue sometido a pericardiocentesis, obteniéndose 1 430 mL de líquido de aspecto serosanguinolento, el mismo que se remitió para estudio, demostrándose un exudado polimorfonuclear, adenosina deaminasa (ADA) positivo y presencia de células compatibles con adenocarcinoma metastático. Se observó en la radiografía de tórax y tomografía axial computarizada pulmonar la presencia de una masa en la región parahiliar izquierda compatible con una neoplasia maligna, con derrame pleural y pericárdico. Se trató de una presentación poco frecuente de neoplasia maligna pulmonar, con metástasis pericárdica, derrame pleural izquierdo y un síndrome paraneoplásico endocrino, caracterizado por hipoglicemia a repetición, como manifestación inicial. Palabras clave: Enfermedades pulmonares; neoplasias; metástasis de la neoplasia; derrame pleural; síndromes paraneoplásicos endocrinos. AbstractA 52 year-old man was admitted to our hospital because of large pericardial effusion and symptoms of recurrent hypoglycaemia. He had no history of smoking or diabetes mellitus and was taking no medication. Diagnostic pericardiocentesis was performed and 1 430 mL of bloody-stained fluid was obtained. The exudate showed neutrophils positive to adenosin deaminase (ADA) test. Chest X-ray and computed tomography showed a left lung hilar mass resembling a lung neoplasm, with pleural and pericardial effusion. It was a lung cancer of unusual presentation, with pleural and pericardial metastasis and a paraneoplastic syndrome characterized by recurrent hypoglycaemia as initial manifestation.

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Mechanisms and Management of Non-islet Cell Tumour Hypoglycaemia in Gastrointestinal Stromal Tumour: Case Report and a Review of Published Studies

Cited by 25 publications

References 28 publications

Multidisciplinary Management of Metastatic Hemangiopericytoma Presenting with Hypoglycemia

Multidisciplinary Management of Metastatic Hemangiopericytoma Presenting with Hypoglycemia

A case of extragastrointestinal stromal tumor complicated by severe hypoglycemia: a unique presentation of a rare tumor

Síndrome de Doege Potter y derrame pericárdico, como manifestaciones iniciales de neoplasia pulmonar

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