{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T07:19:38Z","timestamp":1777879178048,"version":"3.51.4"},"reference-count":39,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T00:00:00Z","timestamp":1764547200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2025,12,1]],"date-time":"2025-12-01T00:00:00Z","timestamp":1764547200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"name":"Khoo Teck Puat Health Fund","award":["STAR18107"],"award-info":[{"award-number":["STAR18107"]}]},{"name":"Khoo Teck Puat Health Fund","award":["STAR19204"],"award-info":[{"award-number":["STAR19204"]}]},{"name":"Khoo Teck Puat Health Fund","award":["STAR21203"],"award-info":[{"award-number":["STAR21203"]}]},{"DOI":"10.13039\/501100001349","name":"National Medical Research Council","doi-asserted-by":"publisher","award":["Clinical Scientist Award"],"award-info":[{"award-number":["Clinical Scientist Award"]}],"id":[{"id":"10.13039\/501100001349","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001349","name":"National Medical Research Council","doi-asserted-by":"publisher","award":["MOH-001688-00"],"award-info":[{"award-number":["MOH-001688-00"]}],"id":[{"id":"10.13039\/501100001349","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001349","name":"National Medical Research Council","doi-asserted-by":"publisher","award":["MOH-001704-00"],"award-info":[{"award-number":["MOH-001704-00"]}],"id":[{"id":"10.13039\/501100001349","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001349","name":"National Medical Research Council","doi-asserted-by":"publisher","award":["NMRC\/CIRG\/1398\/2014"],"award-info":[{"award-number":["NMRC\/CIRG\/1398\/2014"]}],"id":[{"id":"10.13039\/501100001349","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Therapeutic Advances in Endocrinology"],"published-print":{"date-parts":[[2025,12]]},"abstract":"<jats:p>\n                    SHORT syndrome is a rare genetic multisystemic disorder caused by a loss-of-function mutation in the phosphoinositide-3-kinase regulatory subunit 1 (\n                    <jats:italic toggle=\"yes\">PIK3R1<\/jats:italic>\n                    ) gene. The disease\u2019s acronym represents its key features: short stature, hyperextensibility, ocular depression, Rieger anomaly, and teeth delay. Insulin resistance, hyperglycemia, and diabetes mellitus are common endocrinological manifestations of this condition. Currently, there are no established guidelines for the treatment of diabetes in SHORT syndrome patients. In this report, we describe a young adult male patient of Chinese descent with atypical diabetes mellitus associated with SHORT syndrome. This case was challenging due to the patient\u2019s young-onset diabetes and poor diabetes control, complicated by insulin resistance from lipodystrophy, and a strong aversion to insulin injections. By utilizing a combination of oral anti-glycemic agents with complementary mechanisms of action (metformin, sodium-glucose co-transporter 2 (SGLT-2) inhibitors, sulfonylureas, thiazolidinediones, and GLP-1 agonists), insulin therapy was delayed. The patient\u2019s blood glucose levels improved significantly, with HbA1c decreased from 14% to 8.8% within 6\u2009months of starting the multi-agent regimen, and further improved to 7.4% with a fasting plasma glucose of 4.8\u2009mmol\/L. With an oral medication regimen that the patient found acceptable, both his quality of life and adherence to treatment improved. These findings provide useful insights into tailoring an individualized diabetes treatment plan.\n                  <\/jats:p>","DOI":"10.1177\/20420188251405363","type":"journal-article","created":{"date-parts":[[2025,12,23]],"date-time":"2025-12-23T12:16:07Z","timestamp":1766492167000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Diabetes mellitus in SHORT syndrome managed with multi-agent oral therapies: a case report and literature review"],"prefix":"10.1177","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-5756-2891","authenticated-orcid":false,"given":"Yufan","family":"Yang","sequence":"first","affiliation":[{"name":"School of Medicine, Tsinghua University, Beijing, China"},{"name":"Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8479-6194","authenticated-orcid":false,"given":"Si Hua Clara","family":"Tan","sequence":"additional","affiliation":[{"name":"Clinical Research Unit, Khoo Teck Puat Hospital, Singapore, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Su Chi","family":"Lim","sequence":"additional","affiliation":[{"name":"Clinical Research Unit, Khoo Teck Puat Hospital, Singapore, Singapore"},{"name":"Diabetes Centre, Admiralty Medical Centre, Singapore, Singapore"},{"name":"Saw Swee Hock School of Public Health, Singapore, Singapore"},{"name":"Lee Kong Chian School of Medicine, Nanyang Technological University, Singapore, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5593-907X","authenticated-orcid":false,"given":"Wann Jia","family":"Loh","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Changi General Hospital, Simei Street, Singapore 529889, Singapore Duke-NUS Medical School, Singapore, Singapore"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2025,12,23]]},"reference":[{"key":"e_1_3_4_2_2","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12688"},{"key":"e_1_3_4_3_2","doi-asserted-by":"publisher","DOI":"10.1038\/s41568-019-0216-7"},{"key":"e_1_3_4_4_2","doi-asserted-by":"publisher","DOI":"10.1136\/bcr-2013-201554"},{"key":"e_1_3_4_5_2","article-title":"2. Diagnosis and classification of diabetes: standards of care in diabetes\u20142025","volume":"48","author":"American Diabetes Association Professional Practice Committee","year":"2024","unstructured":"American Diabetes Association Professional Practice Committee. 2. Diagnosis and classification of diabetes: standards of care in diabetes\u20142025. Diabetes Care 2024; 48: S27\u2013S49.","journal-title":"Diabetes Care"},{"key":"e_1_3_4_6_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.05.023"},{"key":"e_1_3_4_7_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13052-017-0362-z"},{"key":"e_1_3_4_8_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.1320470619"},{"key":"e_1_3_4_9_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1994.tb13142.x"},{"key":"e_1_3_4_10_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.05.019"},{"key":"e_1_3_4_11_2","doi-asserted-by":"publisher","DOI":"10.1186\/1471-2350-15-51"},{"key":"e_1_3_4_12_2","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.45.1468"},{"key":"e_1_3_4_13_2","doi-asserted-by":"publisher","DOI":"10.1111\/jdi.12825"},{"key":"e_1_3_4_14_2","doi-asserted-by":"publisher","DOI":"10.1186\/s13098-019-0477-z"},{"key":"e_1_3_4_15_2","doi-asserted-by":"publisher","DOI":"10.1002\/mgg3.1385"},{"key":"e_1_3_4_16_2","doi-asserted-by":"publisher","DOI":"10.1111\/jdi.13549"},{"key":"e_1_3_4_17_2","doi-asserted-by":"publisher","DOI":"10.1507\/endocrj.EJ20-0291"},{"key":"e_1_3_4_18_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.62660"},{"key":"e_1_3_4_19_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.63078"},{"key":"e_1_3_4_20_2","first-page":"220","article-title":"SHORT syndrome: a good case can break an old acronym","volume":"70","author":"Ruiz-Garc\u00eda I","year":"2023","unstructured":"Ruiz-Garc\u00eda I, S\u00e1nchez-Torralvo FJ, G\u00f3mez-Zumaquero JM, et al. SHORT syndrome: a good case can break an old acronym. Endocrinol Diabetes Nutr (Engl Ed) 2023; 70: 220\u2013222.","journal-title":"Endocrinol Diabetes Nutr (Engl Ed"},{"key":"e_1_3_4_21_2","doi-asserted-by":"publisher","DOI":"10.1515\/jpem-2024-0244"},{"key":"e_1_3_4_22_2","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2024.1467364"},{"key":"e_1_3_4_23_2","doi-asserted-by":"publisher","DOI":"10.1111\/jdi.70088"},{"key":"e_1_3_4_24_2","doi-asserted-by":"publisher","DOI":"10.1111\/jdi.70062"},{"key":"e_1_3_4_25_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2013.06.005"},{"key":"e_1_3_4_26_2","first-page":"100768","article-title":"The first SHORT syndrome in a Taiwanese boy: a case report and review of the literature","volume":"27","author":"Lee C-L","year":"2021","unstructured":"Lee C-L, Chuang C-K, Chiu H-C, et al. The first SHORT syndrome in a Taiwanese boy: a case report and review of the literature. 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