{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:08:02Z","timestamp":1787018882949,"version":"build-2736575974"},"reference-count":32,"publisher":"SAGE Publications","issue":"1","license":[{"start":{"date-parts":[[2021,10,20]],"date-time":"2021-10-20T00:00:00Z","timestamp":1634688000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["T35HL007744"],"award-info":[{"award-number":["T35HL007744"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Child Neurol"],"published-print":{"date-parts":[[2022,1]]},"abstract":"<jats:sec>\n                    <jats:title>Objective:<\/jats:title>\n                    <jats:p>The purpose of our study was to understand the healthcare burden and incidence of Krabbe disease (Krabbe).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Retrospective analysis of Krabbe patients identified October 1, 2015 through December 31, 2020, ages birth through age 3, evaluated in two national databases. We estimated point prevalence and incidence from year 2016 data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>We identified 98 unique Krabbe patients with 736 visits including 260 were inpatient admissions. Total healthcare charges were $51.5 million dollars. We determined a point prevalence of 34 68 Krabbe patients in 2016 ages 0 3 years. This estimates a birth incidence of ~1 in 310,000 live births. Significance: Krabbe disease patients had over $51 million in health care charges and hundreds of hospitalizations. Estimated prevalence and birth incidence is similar to rates observed from newborn screening. Our findings show the tremendous health impacts of Krabbe disease, and provide guidance for efforts in screening and treatment planning.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/08830738211027717","type":"journal-article","created":{"date-parts":[[2021,10,20]],"date-time":"2021-10-20T23:08:10Z","timestamp":1634771290000},"page":"12-19","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":14,"title":["Hospitalization Burden and Incidence of Krabbe Disease"],"prefix":"10.1177","volume":"37","author":[{"given":"Gabrielle","family":"Ghabash","sequence":"first","affiliation":[{"name":"University of Utah School of Medicine, Salt Lake City, UT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacob","family":"Wilkes","sequence":"additional","affiliation":[{"name":"Intermountain Healthcare, Salt Lake City, UT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bradley J.","family":"Barney","sequence":"additional","affiliation":[{"name":"Division of Critical Care, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8775-147X","authenticated-orcid":false,"given":"Joshua L.","family":"Bonkowsky","sequence":"additional","affiliation":[{"name":"Division of Pediatric Neurology, Primary Children\u2019s Center for Personalized Medicine, Primary Children\u2019s Hospital, Department of Pediatrics, University of Utah School of Medicine, Salt Lake City, UT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2021,10,20]]},"reference":[{"key":"bibr1-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1006\/mgme.2000.2990"},{"key":"bibr2-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.14258"},{"key":"bibr3-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2016.35"},{"key":"bibr4-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1002\/jnr.23793"},{"key":"bibr5-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-018-0766-x"},{"key":"bibr6-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.12.012"},{"key":"bibr7-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1002\/jnr.23905"},{"key":"bibr8-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuron.2020.03.031"},{"key":"bibr9-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1172\/JCI133953"},{"key":"bibr10-08830738211027717","author":"Li Y","year":"2020","journal-title":"Mol Ther"},{"key":"bibr11-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.2346-19.2020"},{"key":"bibr12-08830738211027717","first-page":"6","author":"Blackwell K","year":"2020","journal-title":"Int J Neonatal Screen"},{"key":"bibr13-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1038\/s41436-020-0764-y"},{"key":"bibr14-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1912108116"},{"key":"bibr15-08830738211027717","first-page":"111","author":"Soderholm HE","year":"2020","journal-title":"Pediatr Neurol"},{"key":"bibr16-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1038\/gim.2015.211"},{"key":"bibr17-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1007\/BF02185759"},{"key":"bibr18-08830738211027717","doi-asserted-by":"publisher","DOI":"10.4161\/rdis.25212"},{"key":"bibr19-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1053\/spsu.2002.29367"},{"key":"bibr20-08830738211027717","unstructured":"Healthcare Cost and Utilization Project. Overview of the Kids\u2019 Inpatient Database (KID). www.hcup-us.ahrq.gov\/kidoverview.jsp."},{"key":"bibr21-08830738211027717","unstructured":"National Vital Statistics System. Birth Data. Centers for Disease Control and Prevention. Accessed at https:\/\/www.cdc.gov\/nchs\/nvss\/births.htm. Accessed October 23, 2021."},{"key":"bibr22-08830738211027717","unstructured":"Orsini JJ, Escolar ML, Wasserstein MP, et al. Krabbe Disease. 2000 Jun 19 [Updated 2018 Oct 11]. In: Adam MP, Ardinger HH, Pagon RA, et al. editors. GeneReviews\u00ae [Internet]. Seattle (WA): University of Washington, Seattle; 1993-2021. Available from: https:\/\/www.ncbi.nlm.nih.gov\/books\/NBK1238\/"},{"key":"bibr23-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1212\/CPJ.0000000000000783"},{"key":"bibr24-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-020-01489-3"},{"key":"bibr25-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1186\/s13023-019-1018-4"},{"key":"bibr26-08830738211027717","author":"Yoon IC","year":"2020","journal-title":"Blood"},{"key":"bibr27-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbmt.2018.06.020"},{"key":"bibr28-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000004418"},{"key":"bibr29-08830738211027717","first-page":"49","author":"Brimley CJ","year":"2013","journal-title":"Pediatr Neurol"},{"key":"bibr30-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1002\/ppul.25251"},{"key":"bibr31-08830738211027717","first-page":"1","author":"Bonkowsky JL","year":"2018","journal-title":"JAMA"},{"key":"bibr32-08830738211027717","doi-asserted-by":"publisher","DOI":"10.1186\/s41687-019-0093-6"}],"container-title":["Journal of Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/08830738211027717","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/08830738211027717","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/08830738211027717","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T20:40:10Z","timestamp":1777495210000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/08830738211027717"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,10,20]]},"references-count":32,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2022,1]]}},"alternative-id":["10.1177\/08830738211027717"],"URL":"https:\/\/doi.org\/10.1177\/08830738211027717","relation":{},"ISSN":["0883-0738","1708-8283"],"issn-type":[{"value":"0883-0738","type":"print"},{"value":"1708-8283","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,10,20]]}}}