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About a rare case of hypoglycemia: Non-islet cell tumor hypoglycemia (NICTH)
A propósito de un caso raro de hipoglucemia: Hipoglucemia por tumor de células no islotes (NITCH)
Roberto Sierra-Poyatos
Corresponding author
roberto.sierra@quironsalud.es

Corresponding author.
, Jersy Cárdenas-Salas, Maite Ortega-Juaristi, Clotilde Vázquez-Martínez
Servicio de Endocrinología y Nutrición. Hospital Universitario Fundación Jiménez Díaz, Madrid, Spain
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The physical examination was not relevant except for decreased vesicular murmur in the right lower hemithorax&#46; Weight and body mass index were 52<span class="elsevierStyleHsp" style=""></span>kg and 24<span class="elsevierStyleHsp" style=""></span>kg&#47;m<span class="elsevierStyleSup">2</span> respectively&#46; During a hypoglycemic episode the following results were found&#58; blood glucose 37<span class="elsevierStyleHsp" style=""></span>mg&#47;dl&#44; C-peptide 0&#46;09<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;0&#46;81&#8211;3&#46;85&#41;&#44; insulin &#60;0&#46;50<span class="elsevierStyleHsp" style=""></span>&#956;UI&#47;ml &#40;3&#8211;25&#41;&#44; anti-insulin antibody &#60;5<span class="elsevierStyleHsp" style=""></span>U&#47;ml &#40;&#60;5&#41; and chromogranin A 59&#46;8<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;19&#46;4&#8211;98&#46;1&#41;&#46; A chest X-ray showed opacity in the lower right lobe &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#46; Chest CT scan showed a non-invasive well-defined mass sized 10<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>10<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>20<span class="elsevierStyleHsp" style=""></span>cm&#44; displacing bronchial structures and a right pleural effusion with a maximum thickness of 15<span class="elsevierStyleHsp" style=""></span>mm&#46; This was suggestive of a primary pleural tumor&#44; being a solitary fibrous tumor &#40;SFT&#41; or mesothelioma the most plausible diagnostic options&#46; NICTH was suspected and lab tests revealed&#58; IGF2 510<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;350&#8211;1000&#41;&#44; IGF1 &#60;25<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;54&#46;60&#8211;204&#46;4&#41;&#44; IGFBP3 1&#46;43<span class="elsevierStyleHsp" style=""></span>ng&#47;ml &#40;2&#46;2&#8211;4&#46;5&#41; and IGF2&#47;IGF1 ratio 20&#46;4 &#40;&#60;3&#41;&#46; During the preoperative period&#44; hypoglycemia was effectively prevented with a fractionated enriched diet&#44; intravenous glucose infusion and prednisone 30<span class="elsevierStyleHsp" style=""></span>mg&#47;daily&#46; A 1850<span class="elsevierStyleHsp" style=""></span>g solid mass of 22&#46;6<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>14&#46;5<span class="elsevierStyleHsp" style=""></span>cm<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>9&#46;7<span class="elsevierStyleHsp" style=""></span>cm solid mass was resected via a right posterolateral thoracotomy &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; The microscopic examination revealed a well-defined proliferation with highly-cellular focal areas that alternate with paucicellular areas&#44; edema&#44; focal necrosis&#44; moderate atypia and a mitotic index of 4 mitosis&#47;10 high-power fields &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>C&#41;&#46; Immunohistochemistry was positive for CD34&#44; BCL2&#44; CD99 and IGF1R establishing the diagnosis of a non-invasive SFT &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>D and E&#41;&#46; No radiological signs of disease recurrence were observed in the CT scan performed 2 months after surgery and glucose-lowering treatment &#40;linagliptin&#47;metformin 2&#46;5&#47;850<span class="elsevierStyleHsp" style=""></span>mg BID&#41; was resumed due to poor glycemic control&#46; She did not receive postoperative radiotherapy and the tumor did not show recurrence for the first year after surgery&#46; Then&#44; a minimal pleural thickening with progressive growth was observed and radical fractionated radiotherapy was administered &#40;total dose 5750<span class="elsevierStyleHsp" style=""></span>cGy&#41;&#46; There were no changes in glycemic control&#44; T2DM treatment or hypoglycemic events during recurrence&#46; In her last radiological control&#44; 6 years after surgery&#44; there were no signs of recurrence&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Non-islet cell tumor hypoglycemia is a rare cause of hypoglycemia&#44; due to a tumor overproduction of pro-IGF-2 &#40;Big-IGF2&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Tumors of mesenchymal or hepatic origin are the most common casue&#44; but hypoglycemia has also been described in other benign and malignant tumors&#58; fibrosarcomas&#44; mesotheliomas&#44; hemangiopericytoma&#44; Lymphoma&#47;Leukemia&#44; gastrointestinal stromal tumor &#40;GIST&#41;&#44; yolk cell tumor&#44; plasmocytoma&#44; Leydig cell tumor&#44; phyllodes tumor of the breast and adrenocortical&#44; pancreatic and medullary thyroid carcinoma&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;3</span></a> In a review&#44; a total of 290 cases were identified in the literature between 1988 and 2013&#44; hypoglycemia was de first manifestation in 48&#37; of cases&#44; age ranged from 2 to 87 years &#40;mean 56&#46;4&#41;&#44; tumor diameter tended to be very large &#40;between 10&#8211;20<span class="elsevierStyleHsp" style=""></span>cm&#41; and there was no gender preference&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Other findings include hypokalemia and acromegaloid changes&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Normally IGF2 is limited to the vascular space in the form of high molecular weight complexes&#44; 80&#37; as ternary complexes &#40;With IGFBP3 and acid-labile subunit&#41;&#44; 20&#37; as binary complexes &#40;With IGFBP3&#41; and very little as free IGF2&#46; Abnormal forms of IGF2 &#40;Big-IGF2&#41; cannot form these complexes resulting in higher amounts of low molecular weight complexes and free forms of IGF2&#44; that can cross the capillary membranes to interact with insulin receptors causing hypoglycemia by inhibiting gluconeogenesis&#44; glycogenolysis&#44; and ketogenesis&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Additionally&#44; IGF2 suppresses insulin&#44; GH &#40;with resultant low IGF1&#41; and glucagon secretion&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#8211;5</span></a> Solitary fibrous tumor&#44; first described in 1870 by Wagner&#44; is a rare soft tissue neoplasm&#44; with an incidence of 0&#46;2 per 100&#44;000&#47;year&#44; that causes less than 5&#37; of primary pleural tumors&#46; It can also be found in other locations such as retroperitoneal&#44; hepatic or pelvic locations&#46; Typically&#44; it is usually diagnosed in the 5th&#8211;6th decades&#44; there is no sex preference and they are benign in most cases&#44; although patients may recur and metastasize on occasions&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> They usually present with respiratory symptoms &#40;cough&#44; dyspnea&#44; chest pain&#41; and though one third of tumors are diagnosed incidentally&#46; Hypoglycemia is present in only 4&#37; of patients &#40;Doege-Potter syndrome&#41;&#44; although most of them have an overproduction of Big-IGF2&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#44;7</span></a> NICTH is suspected when other most frequent causes of hypoinsulinemic hypoglycemia &#40;low insulin&#47;C-peptide&#47;proinsulin&#47;betahydroxybutyrate levels&#41;&#44; in the absence of a hypoglycemic agent&#44; are ruled out&#46; Typically&#44; high IGF2&#47;IGF1 ratio &#40;&#62;3&#41;&#44; low IGF1 and normal to high IGF2 levels are the main biochemical features&#46; In some cases&#44; IGF2 could be low&#44; but the presence of higher amounts of high molecular weight IGF2 &#40;Big-IGF2&#41; confirms the diagnosis&#46; Of note&#44; false positive IGF2&#47;IGF1 ratio could be present in patients with malnutrition and sepsis and false negative in those with renal failure &#40;renal failure is associated with low levels of IGFBP3 that can influence IGF1 and IGF2 levels&#41;&#46; Transparietal puncture biopsy is not enough as a diagnostic procedure and a complete resection is needed with a further pathological evaluation and demonstration of positive immunostaining for CD34 and signal transducer and activator of transcription 6 &#40;STAT6&#41;&#46;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7&#44;8</span></a> Recently&#44; NAB2-STAT6 fusion genes&#44; that convert a transcriptional repressor &#40;NAB2&#41; into a transcriptional activator &#40;NAB2-STAT6&#41; have been proposed for the pathogenesis of SFT&#44; leading to overexpression of early growth response 1 &#40;EGR1&#41; target genes &#40;IGF2&#44; H19&#44; and RRAD&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> A decreased activity of the enzyme PCSK4 &#40;Proprotein Convertase Subtilisin&#47;Kexin Type 4&#41; that impairs the processing of pro-IGF2 may also contribute to the overproduction of big IGF2&#46;<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> The prognosis is good&#44; even in malignant forms&#44; when a complete resection is achieved&#46; If there is a case of recurrence&#44; local radiotherapy can be considered&#44; but chemotherapy has very limited benefits&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Hypoglycemia in NITCH improves with corticosteroid treatment&#44; glucagon infusions and recombinant growth hormone &#40;rGH&#41; have been used as well with favorable results&#44; but diazoxide and somatostatin analogs are not useful&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Antibodies against both mature and pro IGF2&#44; anti-IGF2 small interfering RNA and methods that enhance PCSK4 activity<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> are therapies under investigation for the treatment of this rare form of hypoglycemia&#46; In conclusion&#44; based on this case report&#44; NICTH should be suspected in patients with hypoglycemia of unclear etiology&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0015" class="elsevierStylePara elsevierViewall">The authors state that they have not received funding for carrying out this study&#46;</p></span></span>"
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es en pt

¿Es usted profesional sanitario apto para prescribir o dispensar medicamentos?

Are you a health professional able to prescribe or dispense drugs?

Você é um profissional de saúde habilitado a prescrever ou dispensar medicamentos