{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T15:22:39Z","timestamp":1787757759640,"version":"build-2784847793"},"reference-count":10,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"6","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2021,11]]},"abstract":"<jats:sec>\n                    <jats:title>Abstract<\/jats:title>\n                    <jats:p>Mucopolysacharidosis type I is a multisystem disease and often presents with neurobehavioral problems, corneal clouding, cardiac valve involvement, hepatomegaly, coarse facies, and skeletal abnormalities. It has three subtypes \u2013 with Hurler subtype (MPS-1H) being the most severe phenotype with early neurological involvement, rapid progression and mortality, while the other two subtypes \u2013 Hurler-Scheie (MPS-1H\/S) and Scheie (MPS-1S) are of intermediate and milder severity, respectively. Even though neuropsychiatric symptoms have often been reported in the pediatric age group, MPS type I presenting as a major psychiatric illness in adulthood has rarely been reported in literature. Here, we report a female presenting as bipolar affective disorder in the fourth decade of life, where neuroimaging and systemic involvement gave a clue to the diagnosis.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.4103\/0028-3886.333455","type":"journal-article","created":{"date-parts":[[2021,12,28]],"date-time":"2021-12-28T01:47:50Z","timestamp":1640656070000},"page":"1753-1755","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":1,"title":["Mucopolysacharidosis Type I Presenting as Bipolar Affective Disorder"],"prefix":"10.4103","volume":"69","author":[{"given":"Udit U","family":"Saraf","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jithu","family":"Jose","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Syam","family":"Krishnan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sapna Erat","family":"Sreedharan","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"R1-48-20240907","doi-asserted-by":"crossref","first-page":"759","DOI":"10.1038\/gim.2014.25","article-title":"The natural history of MPS I: Global perspectives from the MPS I registry","volume":"16","author":"Beck","year":"2014","journal-title":"Genet Med"},{"key":"R2-48-20240907","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1016\/j.ymgme.2017.08.009","article-title":"Developmental and behavioral aspects of mucopolysaccharidoses with brain manifestations \u2014 Neurological signs and symptoms","volume":"122","author":"Shapiro","year":"2017","journal-title":"Mol Genet Meta"},{"key":"R3-48-20240907","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.ymgme.2017.09.010","article-title":"Practical management of behavioral problems in mucopolysaccharidoses disorders","volume":"122S","author":"Escolar","year":"2017","journal-title":"Mol Genet Metab"},{"key":"R4-48-20240907","doi-asserted-by":"crossref","first-page":"123","DOI":"10.1016\/j.ymgme.2013.11.014","article-title":"Neurocognitive and neuropsychiatric phenotypes associated with the mutation L238Q of the alpha-L-iduronidase gene in Hurler-Scheie syndrome","volume":"111","author":"Ahmed","year":"2014","journal-title":"Mol Genet Metab"},{"key":"R5-48-20240907","doi-asserted-by":"crossref","first-page":"666","DOI":"10.1007\/s00234-004-1215-1","article-title":"Brain MRI findings in patients with mucopolysaccharidosis types I and II and mild clinical presentation","volume":"46","author":"Matheus","year":"2004","journal-title":"Neuroradiology"},{"key":"R6-48-20240907","doi-asserted-by":"crossref","first-page":"55","DOI":"10.1186\/1750-1172-6-55","article-title":"Enzyme replacement therapy and\/or hematopoietic stem cell transplantation at diagnosis in patients with mucopolysaccharidosis type I: Results of a European consensus procedure","volume":"6","author":"de Ru","year":"2011","journal-title":"Orphanet J Rare Dis"},{"key":"R7-48-20240907","doi-asserted-by":"crossref","first-page":"453","DOI":"10.4103\/2230-8210.95712","article-title":"Gadve, Dipti Sarma, Uma K. Saikia Short stature with umbilical hernia \u2013 Not always due to cretinism: A report of two cases","volume":"16","author":"Sharvil","year":"2012","journal-title":"Indian J Endocrinol Metab"},{"key":"R8-48-20240907","doi-asserted-by":"crossref","first-page":"86","DOI":"10.4103\/0976-237X.94554","article-title":"Clinical manifestation of Hurler syndrome in a 7 year old child","volume":"3","author":"Sharma","year":"2012","journal-title":"Contemp Clin Dent"},{"key":"R9-48-20240907","doi-asserted-by":"crossref","first-page":"bcr2016215416","DOI":"10.1136\/bcr-2016-215416","article-title":"Mucopolysaccharidosis type I disguised as rickets","volume":"2016","author":"Chakraborty","year":"2016","journal-title":"BMJ Case Rep"},{"key":"R10-48-20240907","doi-asserted-by":"crossref","first-page":"bcr2013202730","DOI":"10.1136\/bcr-2013-202730","article-title":"Deep anterior lamellar keratoplasty in case of Hurler-Scheie syndrome","volume":"2014","author":"Sati","year":"2014","journal-title":"BMJ Case Rep"}],"container-title":["Neurology India"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/0028-3886.333455","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T14:52:21Z","timestamp":1787755941000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/0028-3886.333455"}},"subtitle":["A Case Report"],"short-title":[],"issued":{"date-parts":[[2021,11]]},"references-count":10,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2021]]}},"URL":"https:\/\/doi.org\/10.4103\/0028-3886.333455","relation":{},"ISSN":["0028-3886"],"issn-type":[{"value":"0028-3886","type":"print"}],"subject":[],"published":{"date-parts":[[2021,11]]}}}