{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2024,8,7]],"date-time":"2024-08-07T21:14:32Z","timestamp":1723065272233},"reference-count":10,"publisher":"Walter de Gruyter GmbH","issue":"2","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2018,1,26]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2017-0398_s_999_w2aab3b7c56b1b6b1aab1c16b1Aa\">\n                  <jats:title>Background:<\/jats:title>\n                  <jats:p>Insulin-mediated pseudoacromegaly is a rarely described pediatric phenotype. We present two patients displaying excessive growth associated with marked acanthosis nigricans, hyperinsulinemia and metabolic dysregulation.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2017-0398_s_998_w2aab3b7c56b1b6b1aab1c16b2Aa\">\n                  <jats:title>Case presentation:<\/jats:title>\n                  <jats:p>Both patients, of First Nations descent, presented with excessive growth \u2013 patient one at 3.92 years (height z-score +3.75) and patient two at 9.0 years (height z-score 5.15). Insulin-like growth factor-1 (IGF-1) levels were normal with appropriate growth hormone suppression, yet marked hyperinsulinemia. Prepubescent growth velocities exceeded 9 cm\/year, resulting in final adult height predictions exceeding 3 standard deviations (SDs) of predicted. Clinical courses were complicated by type 2 diabetes, marked acanthosis nigricans and long-standing psychosocial distress.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2017-0398_s_997_w2aab3b7c56b1b6b1aab1c16b3Aa\">\n                  <jats:title>Conclusions:<\/jats:title>\n                  <jats:p>Pediatric patients with insulin-mediated pseudoacromegaly are at risk of significant physical, metabolic and psychosocial comorbidities. Unlike adults, the implications in childhood prompt consideration for therapies to decelerate linear growth and avert progression to metabolic dysregulation. Increased recognition of this condition may improve pathophysiological understanding, diagnostic criteria and therapeutic options.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2017-0398","type":"journal-article","created":{"date-parts":[[2017,12,18]],"date-time":"2017-12-18T22:16:32Z","timestamp":1513635392000},"page":"235-238","source":"Crossref","is-referenced-by-count":3,"title":["Insulin-mediated pseudoacromegaly: a report of two pediatric patients"],"prefix":"10.1515","volume":"31","author":[{"given":"Mark","family":"Inman","sequence":"first","affiliation":[{"name":"Pediatric Endocrinologist, Clinical Assistant Professor, Department of Pediatrics , University of Saskatchewan , 103 Hospital Drive , Saskatoon , SK S7N0W8, Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Munier A.","family":"Nour","sequence":"additional","affiliation":[{"name":"Department of Pediatrics , University of Saskatchewan, Saskatoon , Saskatchewan , Canada"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"374","published-online":{"date-parts":[[2017,12,18]]},"reference":[{"key":"2021021317335115459_j_jpem-2017-0398_ref_001_w2aab3b7c56b1b6b1ab2b2b1Aa","unstructured":"Flier JS, Moller DE, Moses AC, O\u2019Rahilly S, Chaiken RL, et al. Insulin mediated pseudoacromegaly: clinical and biochemical characterization of a syndrome of selective insulin resistance. J Clin Endocriol Metab 1993;76:1533\u201341."},{"key":"2021021317335115459_j_jpem-2017-0398_ref_002_w2aab3b7c56b1b6b1ab2b2b2Aa","unstructured":"Kausch Ch, Bergemann Ch, Hamann A, Matthaei S. Insulin-mediated pseudoacromegaly in a patient with severe insulin resistance: association of defective insulin-stimulated glucose transport with impaired phosphatidylinositol 3-kinase activity in fibroblasts. Exp Clin Endocrinol Diabetes 1999;107:148\u201354."},{"key":"2021021317335115459_j_jpem-2017-0398_ref_003_w2aab3b7c56b1b6b1ab2b2b3Aa","unstructured":"Kumar S, Durrington PN, O\u2019Rahilly S, Laing I, Humphreys PJ, et al. Severe insulin resistance, diabetes mellitus, hypertriglyceridemia, and pseudoacromegaly. J Clin Endocriol Metab 1996;81:3465\u20139."},{"key":"2021021317335115459_j_jpem-2017-0398_ref_004_w2aab3b7c56b1b6b1ab2b2b4Aa","doi-asserted-by":"crossref","unstructured":"Sam AH, Tan T, Meeran K. Insulin-mediated \u201cpseudoacromegaly\u201d. Hormones 2011;10;156\u201361.","DOI":"10.14310\/horm.2002.1306"},{"key":"2021021317335115459_j_jpem-2017-0398_ref_005_w2aab3b7c56b1b6b1ab2b2b5Aa","unstructured":"Yaqub A, Yaqub N. Insulin-mediated pseudoacromegaly: a case report and review of the literature. W V Med J 2008;104:12\u20135."},{"key":"2021021317335115459_j_jpem-2017-0398_ref_006_w2aab3b7c56b1b6b1ab2b2b6Aa","doi-asserted-by":"crossref","unstructured":"Srinivasan S, Waters MJ, Rowland JE, Baxter RC, Verge CF. Hyperinsulinism and overgrowth without obesity. 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J Biol Chem 2006;281:25869\u201374.","DOI":"10.1074\/jbc.M605189200"},{"key":"2021021317335115459_j_jpem-2017-0398_ref_009_w2aab3b7c56b1b6b1ab2b2b9Aa","doi-asserted-by":"crossref","unstructured":"Fradkin JE, Eastman RC, Lesniak MA, Roth J. Specificity spillover at the hormone receptor-exploring its role in human disease. N Engl J Med 1989;320:640\u20135.","DOI":"10.1056\/NEJM198903093201005"},{"key":"2021021317335115459_j_jpem-2017-0398_ref_010_w2aab3b7c56b1b6b1ab2b2c10Aa","doi-asserted-by":"crossref","unstructured":"Dib K, Whitehead JP, Humphreys PJ, Soos MA, Baynes KC, et al. Impaired activation of Phosphoinositide 3-Kinase by insulin in fibroblasts from patients with severe insulin resistance and pseudoacromegaly. 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