{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T07:19:00Z","timestamp":1760080740680},"reference-count":20,"publisher":"Springer Science and Business Media LLC","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,7,25]]},"DOI":"10.7759\/cureus.27264","type":"journal-article","created":{"date-parts":[[2022,7,26]],"date-time":"2022-07-26T06:21:44Z","timestamp":1658816504000},"source":"Crossref","is-referenced-by-count":7,"title":["Repurposing of Empagliflozin as a Possible Treatment for Neutropenia and Inflammatory Bowel Disease in Glycogen Storage Disease Type Ib: A Case Report"],"prefix":"10.7759","author":[{"given":"Konstantinos","family":"Makrilakis","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aikaterini","family":"Barmpagianni","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria","family":"Veiga-da-Cunha","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","reference":[{"key":"ref1","doi-asserted-by":"publisher","DOI":"10.1007\/s10545-014-9772-x","article-title":"Type I glycogen storage diseases: disorders of the glucose-6-phosphatase\/glucose-6-phosphate transporter complexes","volume":"38","author":"Chou JY","year":"2015","unstructured":"Chou JY, Jun HS, Mansfield BC. Type I glycogen storage diseases: disorders of the glucose-6-phosphatase\/glucose-6-phosphate transporter complexes. J Inherit Metab Dis. 2015, 38:511-519. 10.1007\/s10545-014-9772-x","journal-title":"J Inherit Metab Dis"},{"key":"ref2","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2014.128","article-title":"Diagnosis and management of glycogen storage disease type I: a practice guideline of the American College of Medical Genetics and Genomics","volume":"16","author":"Kishnani PS","year":"2014","unstructured":"Kishnani PS, Austin SL, Abdenur JE, et al.. Diagnosis and management of glycogen storage disease type I: a practice guideline of the American College of Medical Genetics and Genomics. Genet Med. 2014, 16:e1. 10.1038\/gim.2014.128","journal-title":"Genet Med"},{"key":"ref3","doi-asserted-by":"publisher","DOI":"10.1097\/MOH.0000000000000474","article-title":"Neutropenia in glycogen storage disease Ib: outcomes for patients treated with granulocyte colony-stimulating factor","volume":"26","author":"Dale DC","year":"2019","unstructured":"Dale DC, Bolyard AA, Marrero T, et al.. Neutropenia in glycogen storage disease Ib: outcomes for patients treated with granulocyte colony-stimulating factor. Curr Opin Hematol. 2019, 26:16-21. 10.1097\/MOH.0000000000000474","journal-title":"Curr Opin Hematol"},{"key":"ref4","doi-asserted-by":"publisher","DOI":"10.1007\/S00431-002-1017-6","article-title":"Consensus guidelines for management of glycogen storage disease type 1b - European Study on Glycogen Storage Disease Type 1","volume":"161","author":"Visser G","year":"2002","unstructured":"Visser G, Rake J, Labrune P, et al.. Consensus guidelines for management of glycogen storage disease type 1b - European Study on Glycogen Storage Disease Type 1. Eur J Pediatr. 2002, 161:120-123. 10.1007\/S00431-002-1017-6","journal-title":"Eur J Pediatr"},{"key":"ref5","doi-asserted-by":"publisher","DOI":"10.1080\/08880018.2018.1440675","article-title":"Prolonged granulocyte colony stimulating factor use in glycogen storage disease type 1b associated with acute myeloid leukemia and with shortened telomere length","volume":"35","author":"Li AM","year":"2018","unstructured":"Li AM, Thyagu S, Maze D, et al.. Prolonged granulocyte colony stimulating factor use in glycogen storage disease type 1b associated with acute myeloid leukemia and with shortened telomere length. Pediatr Hematol Oncol. 2018, 35:45-51. 10.1080\/08880018.2018.1440675","journal-title":"Pediatr Hematol Oncol"},{"key":"ref6","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1816143116","article-title":"Failure to eliminate a phosphorylated glucose analog leads to neutropenia in patients with G6PT and G6PC3 deficiency","volume":"116","author":"Veiga-da-Cunha M","year":"2019","unstructured":"Veiga-da-Cunha M, Chevalier N, Stephenne X, et al.. Failure to eliminate a phosphorylated glucose analog leads to neutropenia in patients with G6PT and G6PC3 deficiency. Proc Natl Acad Sci USA. 2019, 116:1241-1250. 10.1073\/pnas.1816143116","journal-title":"Proc Natl Acad Sci USA"},{"key":"ref7","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2019004465","article-title":"Treating neutropenia and neutrophil dysfunction in glycogen storage disease type Ib with an SGLT2 inhibitor","volume":"136","author":"Wortmann SB","year":"2020","unstructured":"Wortmann SB, Van Hove JL, Derks TG, et al.. Treating neutropenia and neutrophil dysfunction in glycogen storage disease type Ib with an SGLT2 inhibitor. Blood. 2020, 136:1033-1043. 10.1182\/blood.2019004465","journal-title":"Blood"},{"key":"ref8","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-020-01503-8","article-title":"Improved inflammatory bowel disease, wound healing and normal oxidative burst under treatment with empagliflozin in glycogen storage disease type Ib","volume":"15","author":"Gr\u00fcnert SC","year":"2020","unstructured":"Gr\u00fcnert SC, Elling R, Maag B, et al.. Improved inflammatory bowel disease, wound healing and normal oxidative burst under treatment with empagliflozin in glycogen storage disease type Ib. Orphanet J Rare Dis. 2020, 15:218. 10.1186\/s13023-020-01503-8","journal-title":"Orphanet J Rare Dis"},{"key":"ref9","doi-asserted-by":"publisher","DOI":"10.1186\/s13052-021-01100-w","article-title":"Crohn disease-like enterocolitis remission after empagliflozin treatment in a child with glycogen storage disease type Ib: a case report","volume":"47","author":"Rossi A","year":"2021","unstructured":"Rossi A, Miele E, Fecarotta S, et al.. Crohn disease-like enterocolitis remission after empagliflozin treatment in a child with glycogen storage disease type Ib: a case report. Ital J Pediatr. 2021, 47:149. 10.1186\/s13052-021-01100-w","journal-title":"Ital J Pediatr"},{"key":"ref10","doi-asserted-by":"publisher","DOI":"10.1111\/ped.14629","article-title":"Empagliflozin ameliorated neutropenia in a girl with glycogen storage disease Ib","volume":"63","author":"Mikami M","year":"2021","unstructured":"Mikami M, Arai A, Mizumoto H. Empagliflozin ameliorated neutropenia in a girl with glycogen storage disease Ib. Pediatr Int. 2021, 63:1394-1396. 10.1111\/ped.14629","journal-title":"Pediatr Int"},{"key":"ref11","doi-asserted-by":"publisher","DOI":"10.1002\/jmd2.12278","article-title":"Sodium-glucose cotransporter type 2 channel inhibitor: Breakthrough in the treatment of neutropenia in patients with glycogen storage disease type 1b?","volume":"63","author":"Kaczor M","year":"2022","unstructured":"Kaczor M, Greczan M, Kierus K, et al.. Sodium-glucose cotransporter type 2 channel inhibitor: Breakthrough in the treatment of neutropenia in patients with glycogen storage disease type 1b?. JIMD Rep. 2022, 63:199-206. 10.1002\/jmd2.12278","journal-title":"JIMD Rep"},{"key":"ref12","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-022-02345-2","article-title":"Understanding the role of SGLT2 inhibitors in glycogen storage disease type Ib: the experience of one UK centre","volume":"17","author":"Halligan RK","year":"2022","unstructured":"Halligan RK, Dalton RN, Turner C, Lewis KA, Mundy HR. Understanding the role of SGLT2 inhibitors in glycogen storage disease type Ib: the experience of one UK centre. Orphanet J Rare Dis. 2022, 17:195. 10.1186\/s13023-022-02345-2","journal-title":"Orphanet J Rare Dis"},{"key":"ref13","doi-asserted-by":"publisher","DOI":"10.1155\/1999\/506915","article-title":"The Crohn's Disease Activity Index, its derivatives and the Inflammatory Bowel Disease Questionnaire: a review of instruments to assess Crohn's disease","volume":"13","author":"Yoshida EM","year":"1999","unstructured":"Yoshida EM. The Crohn's Disease Activity Index, its derivatives and the Inflammatory Bowel Disease Questionnaire: a review of instruments to assess Crohn's disease. Can J Gastroenterol. 1999, 13:65-73. 10.1155\/1999\/506915","journal-title":"Can J Gastroenterol"},{"key":"ref14","doi-asserted-by":"publisher","DOI":"10.1080\/00365521.2017.1315740","article-title":"The utility of fecal calprotectin in predicting the need for escalation of therapy in inflammatory bowel disease","volume":"52","author":"Kwapisz L","year":"2017","unstructured":"Kwapisz L, Gregor J, Chande N, Yan B, Ponich T, Mosli M. The utility of fecal calprotectin in predicting the need for escalation of therapy in inflammatory bowel disease. Scand J Gastroenterol. 2017, 52:846-850. 10.1080\/00365521.2017.1315740","journal-title":"Scand J Gastroenterol"},{"key":"ref15","doi-asserted-by":"publisher","DOI":"10.35772\/ghm.2019.01031","article-title":"Cardiovascular disease and 1,5-anhydro-d-glucitol","volume":"1","author":"Ikeda N","year":"2019","unstructured":"Ikeda N, Hiroi Y. Cardiovascular disease and 1,5-anhydro-d-glucitol. Glob Health Med. 2019, 1:83-87. 10.35772\/ghm.2019.01031","journal-title":"Glob Health Med"},{"key":"ref16","doi-asserted-by":"publisher","DOI":"10.1152\/ajpendo.1992.263.2.E268","article-title":"Origin and disposal of 1,5-anhydroglucitol, a major polyol in the human body","volume":"263","author":"Yamanouchi T","year":"1992","unstructured":"Yamanouchi T, Tachibana Y, Akanuma H, et al.. Origin and disposal of 1,5-anhydroglucitol, a major polyol in the human body. Am J Physiol. 1992, 263:E268-E273. 10.1152\/ajpendo.1992.263.2.E268","journal-title":"Am J Physiol"},{"key":"ref17","doi-asserted-by":"publisher","DOI":"10.1016\/j.lfs.2004.10.016","article-title":"SLC5A9\/SGLT4, a new Na+-dependent glucose transporter, is an essential transporter for mannose, 1,5-anhydro-D-glucitol, and fructose","volume":"76","author":"Tazawa S","year":"2005","unstructured":"Tazawa S, Yamato T, Fujikura H, et al.. SLC5A9\/SGLT4, a new Na+-dependent glucose transporter, is an essential transporter for mannose, 1,5-anhydro-D-glucitol, and fructose. Life Sci. 2005, 76:1039-1050. 10.1016\/j.lfs.2004.10.016","journal-title":"Life Sci"},{"key":"ref18","doi-asserted-by":"publisher","DOI":"10.1002\/jimd.12187","article-title":"Inborn errors of metabolite repair","volume":"43","author":"Veiga-da-Cunha M","year":"2020","unstructured":"Veiga-da-Cunha M, Van Schaftingen E, Bommer GT. Inborn errors of metabolite repair. J Inherit Metab Dis. 2020, 43:14-24. 10.1002\/jimd.12187","journal-title":"J Inherit Metab Dis"},{"key":"ref19","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2020006306","article-title":"When sugar isn't sweet: neutropenia in GSD-Ib","volume":"136","author":"Weinstein DA","year":"2020","unstructured":"Weinstein DA. When sugar isn't sweet: neutropenia in GSD-Ib. Blood. 2020, 136:1015-1016. 10.1182\/blood.2020006306","journal-title":"Blood"},{"key":"ref20","doi-asserted-by":"publisher","DOI":"10.1016\/j.gim.2022.04.001","article-title":"Efficacy and safety of empagliflozin in glycogen storage disease type Ib: data from an international questionnaire","author":"Gr\u00fcnert SC","year":"2022","unstructured":"Gr\u00fcnert SC, Derks TG, Adrian K, et al.. Efficacy and safety of empagliflozin in glycogen storage disease type Ib: data from an international questionnaire. Genet Med. 2022, 1-8. 10.1016\/j.gim.2022.04.001","journal-title":"Genet Med"}],"container-title":["Cureus"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.cureus.com\/articles\/97701-repurposing-of-empagliflozin-as-a-possible-treatment-for-neutropenia-and-inflammatory-bowel-disease-in-glycogen-storage-disease-type-ib-a-case-report","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,2,9]],"date-time":"2024-02-09T19:35:52Z","timestamp":1707507352000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.cureus.com\/articles\/97701-repurposing-of-empagliflozin-as-a-possible-treatment-for-neutropenia-and-inflammatory-bowel-disease-in-glycogen-storage-disease-type-ib-a-case-report"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,7,25]]},"references-count":20,"URL":"https:\/\/doi.org\/10.7759\/cureus.27264","relation":{},"ISSN":["2168-8184"],"issn-type":[{"value":"2168-8184","type":"print"}],"subject":[],"published":{"date-parts":[[2022,7,25]]}}}