{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T15:38:17Z","timestamp":1792165097444,"version":"4.4.0"},"reference-count":12,"publisher":"Walter de Gruyter GmbH","issue":"7","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,7,28]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec id=\"j_jpem-2019-0389_abs_001_w2aab3b7b6b1b6b1aab1c16b2Aa\">\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>Prader-Willi Syndrome (PWS) is characterised by hyperphagia often leading to obesity; a known risk factor for insulin resistance and type 2 (T2) diabetes. We present a prepubertal girl with PWS who developed diabetes.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2019-0389_abs_002_w2aab3b7b6b1b6b1aab1c16b3Aa\">\n                  <jats:title>Case presentation<\/jats:title>\n                  <jats:p>Our case was diagnosed with PWS in infancy following investigation for profound central hypotonia and feeding difficulties. She commenced growth hormone (GH) aged 8 years for short stature and treatment improved linear growth. At age 12 years, she presented with polydipsia, polyuria and vulvovaginitis. She was overweight (BMI SDS\u00a0+1.43). Diabetes was diagnosed (Blood glucose\u00a0=\u00a024.2\u00a0mmol\/L, HbA1c\u00a0=\u00a0121\u00a0mmol\/mol or 13.2%). She was not acidotic and had negative blood ketones. Autoantibodies typical of type 1 diabetes were negative. She was initially treated with basal bolus insulin regime. GH was discontinued 3\u00a0months later due to concerns regarding GH-induced insulin resistance. Off GH, insulin requirements reduced to zero, allowing Metformin monotherapy. However off GH, she reported significant lethargy with static growth and increased weight. Combinations of Metformin with differing insulin regimes did not improve glucose levels. Liraglutide (GLP-1 agonist) and Metformin did not improve glucose levels nor her weight. Liraglutide and Empaglifozin (SGLT-2 inhibitor) therapy used in combination were well tolerated and demonstrated rapid normalisation of blood glucose and improvement in her HbA1c to within target (48\u00a0mmol\/mol) which was sustained after 6\u00a0months of treatment.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec id=\"j_jpem-2019-0389_abs_003_w2aab3b7b6b1b6b1aab1c16b4Aa\">\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Newer treatments for type 2 diabetes (e.\u00a0g. GLP-1 agonists or SGLT-2 inhibitors) offer potential treatment options for those with diabetes and PWS when conventional treatments are ineffective.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1515\/jpem-2019-0389","type":"journal-article","created":{"date-parts":[[2020,5,26]],"date-time":"2020-05-26T16:36:59Z","timestamp":1590511019000},"page":"951-955","source":"Crossref","is-referenced-by-count":20,"title":["Improvement in glycaemic parameters using SGLT-2 inhibitor and GLP-1 agonist in combination in an adolescent with diabetes mellitus and Prader-Willi syndrome: a case report"],"prefix":"10.1515","volume":"33","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4587-8744","authenticated-orcid":false,"given":"Toby","family":"Candler","sequence":"first","affiliation":[{"name":"Department of Paediatric Endocrinology , Bristol Royal Hospital for Children , University Hospitals Bristol NHS Foundation Trust , Bristol , UK"},{"name":"Medical Research Council The Gambia at London School of Hygiene and Tropical Medicine , London , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David","family":"McGregor","sequence":"additional","affiliation":[{"name":"Department of Paediatrics , Royal Devon and Exeter NHS Foundation Trust , Exeter , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kruthika","family":"Narayan","sequence":"additional","affiliation":[{"name":"Department of Paediatric Endocrinology , Bristol Royal Hospital for Children , University Hospitals Bristol NHS Foundation Trust , Bristol , UK"},{"name":"Department of Paediatric Endocrinology and Diabetes , Queensland Children's Hospital , Brisbane , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chris","family":"Moudiotis","sequence":"additional","affiliation":[{"name":"Department of Paediatrics , Royal Devon and Exeter NHS Foundation Trust , Exeter , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine P.","family":"Burren","sequence":"additional","affiliation":[{"name":"Department of Paediatric Endocrinology , Bristol Royal Hospital for Children , University Hospitals Bristol NHS Foundation Trust , Bristol , UK"},{"name":"Bristol Medical School , Department of Translational Health Sciences , University of Bristol , Bristol , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","published-online":{"date-parts":[[2020,5,24]]},"reference":[{"key":"2023040101122164225_j_jpem-2019-0389_ref_001_w2aab3b7b6b1b6b1ab2b2b1Aa","doi-asserted-by":"crossref","unstructured":"Angulo MA, Butler MG, Cataletto ME. 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