{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,2]],"date-time":"2026-09-02T02:28:36Z","timestamp":1788316116679,"version":"build-2803163510"},"reference-count":13,"publisher":"American Academy of Pediatrics (AAP)","issue":"1","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,7,1]]},"abstract":"<jats:p>Insulin-resistant diabetes in Rabson\u2013Mendenhall syndrome (RMS) is relatively unresponsive to first-line antidiabetic treatments, including metformin and insulin. We report 2 patients with RMS treated with 2 different sodium-glucose cotransporter inhibitors 2: empagliflozin in an 11-year-old boy and dapagliflozin in a 12-year-old girl. In the first patient, we began empagliflozin at 2.5 mg\/day and increased the dose to 10 mg\/day over 3 months. During treatment with empagliflozin, the amount of time during which the patient maintained serum glucose in the 70 to 180 mg\/dL target range increased by 2 hours per day. Hemoglobin A1C dropped from &amp;gt;14% to 11.9%, urinary calcium increased almost twofold, and \u03b2-hydroxybutyrate remained &amp;lt;2.5 mmol\/L. Because glycemic control did not further improve with dose escalation, we reverted to the 2.5 mg\/day dose. We initiated dapagliflozin in a second patient at 5 mg\/day and witnessed a reduction of hemoglobin A1C from 8.5% to 6.2% after 6 months and a mild increase in urinary excretion of phosphorus but not calcium. Insulin levels fell by &amp;gt;50%. In 2 patients with RMS, empagliflozin and dapagliflozin were well tolerated and improved glycemic control without significantly increasing ketonemia. Renal calcium excretion should be carefully monitored.<\/jats:p>","DOI":"10.1542\/peds.2021-055671","type":"journal-article","created":{"date-parts":[[2022,6,2]],"date-time":"2022-06-02T08:13:36Z","timestamp":1654157616000},"source":"Crossref","is-referenced-by-count":14,"title":["SGLT2i Improves Glycemic Control in Patients With Congenital Severe Insulin Resistance"],"prefix":"10.1542","volume":"150","author":[{"given":"Alfonso","family":"Galderisi","sequence":"first","affiliation":[{"name":"aDepartment of Woman and Child\u2019s Health, University of Padova, Padova, Veneto, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"William","family":"Tamborlane","sequence":"additional","affiliation":[{"name":"bDepartment of Pediatrics, Yale University, New Haven, Connecticut"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simeon I.","family":"Taylor","sequence":"additional","affiliation":[{"name":"cDepartment of Medicine, University of Maryland School of 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Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"417","published-online":{"date-parts":[[2022,6,2]]},"reference":[{"issue":"4","key":"2023110816072881700_B1","doi-asserted-by":"crossref","first-page":"498","DOI":"10.1210\/er.2010-0020","article-title":"Genetic syndromes of severe insulin resistance","volume":"32","author":"Semple","year":"2011","journal-title":"Endocr Rev"},{"issue":"7","key":"2023110816072881700_B2","doi-asserted-by":"crossref","first-page":"669","DOI":"10.1111\/j.1463-1326.2011.01406.x","article-title":"Canagliflozin, a novel inhibitor of sodium glucose co-transporter 2, dose dependently reduces calculated renal threshold for glucose excretion and increases urinary glucose excretion in healthy subjects","volume":"13","author":"Sha","year":"2011","journal-title":"Diabetes Obes Metab"},{"key":"2023110816072881700_B3","doi-asserted-by":"crossref","first-page":"k119","DOI":"10.1136\/bmj.k119","article-title":"Cardiovascular outcomes associated with 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meta-analysis","volume":"96","author":"Li","year":"2017","journal-title":"Medicine (Baltimore)"},{"issue":"22","key":"2023110816072881700_B12","doi-asserted-by":"crossref","first-page":"2117","DOI":"10.1056\/NEJMoa1504720","article-title":"Empagliflozin, cardiovascular outcomes, and mortality in type 2 diabetes","volume":"373","author":"Zinman","year":"2015","journal-title":"N Engl J Med"},{"key":"2023110816072881700_B13","doi-asserted-by":"crossref","first-page":"8867875","DOI":"10.1155\/2020\/8867875","article-title":"The role of SGLT2 inhibitor on the treatment of diabetic retinopathy","volume":"2020","author":"Sha","year":"2020","journal-title":"J Diabetes 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