{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T07:40:04Z","timestamp":1756366804414,"version":"3.44.0"},"reference-count":19,"publisher":"BMJ","issue":"7","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2024,7,16]]},"published-print":{"date-parts":[[2024,7]]},"abstract":"<jats:title>Summary<\/jats:title>\n               <jats:p>Shapiro\u2019s syndrome is a rare neurological disease. The triad of Shapiro\u2019s syndrome includes episodes of hyperhidrosis, hypothermia and complete\/partial agenesis of the corpus callosum. We report a case of a young male who had episodic chills, increased sweating and fatigue. During these episodes, he was found to have bradycardia, hypotension and hypothermia. Clinical and neurological examinations were unremarkable. The MRI of the brain revealed agenesis of the corpus callosum. There was a good response to carbamazepine therapy.<\/jats:p>","DOI":"10.1136\/bcr-2024-260998","type":"journal-article","created":{"date-parts":[[2024,7,29]],"date-time":"2024-07-29T12:53:51Z","timestamp":1722257631000},"page":"e260998","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Shapiro\u2019s syndrome: episodic hypothermic hyperhidrosis"],"prefix":"10.1136","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-0991-9563","authenticated-orcid":false,"given":"Deepa","family":"Avadhani","sequence":"first","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research (Deemed to be University), Chennai, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lakshmi Narasimhan","family":"Ranganathan","sequence":"additional","affiliation":[{"name":"Department of Neurology, Sri Ramachandra Institute of Higher Education and Research (Deemed to be University), Chennai, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chandramouleeswaran","family":"Venkataraman","sequence":"additional","affiliation":[{"name":"Department of Neurology, Madras Medical College, Chennai, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Balasubramanian","family":"Samivel","sequence":"additional","affiliation":[{"name":"Department of Neurology, Madras Medical College, Chennai, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2024,7,29]]},"reference":[{"key":"2025082800220410000_17.7.e260998.1","doi-asserted-by":"publisher","DOI":"10.1212\/01.CPJ.0000437350.47610.3a"},{"key":"2025082800220410000_17.7.e260998.2","doi-asserted-by":"crossref","first-page":"423","DOI":"10.1093\/brain\/92.2.423","article-title":"Spontaneous recurrent hypothermia accompanying agenesis of the corpus callosum","volume":"92","author":"Shapiro","year":"1969","journal-title":"Brain (Bacau)"},{"key":"2025082800220410000_17.7.e260998.3","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1016\/j.jns.2013.03.014","article-title":"Insights into thermoregulation: a clinico-radiological description of shapiro syndrome","volume":"329","author":"Pazderska","year":"2013","journal-title":"J Neurol Sci"},{"key":"2025082800220410000_17.7.e260998.4","doi-asserted-by":"publisher","DOI":"10.1093\/qjmed\/hcm106"},{"key":"2025082800220410000_17.7.e260998.5","doi-asserted-by":"publisher","DOI":"10.1097\/00005792-199509000-00004"},{"key":"2025082800220410000_17.7.e260998.6","doi-asserted-by":"publisher","DOI":"10.3389\/fgene.2022.958570"},{"key":"2025082800220410000_17.7.e260998.7","doi-asserted-by":"crossref","first-page":"756","DOI":"10.1007\/s00415-011-6237-0","article-title":"Recurrent hypothermia with hyperhidrosis in two siblings: familial Shapiro syndrome variant","volume":"259","author":"Belcastro","year":"2012","journal-title":"J Neurol"},{"key":"2025082800220410000_17.7.e260998.8","doi-asserted-by":"crossref","unstructured":"Salomao R , Pedroso JL , Barsottini O . Three Shapiro syndrome cases: diagnostic challenge of clinical spectrum of the spontaneous paroxysmal hypothermia syndrome (P2.123). Neurol (ECronicon) 2018;90. doi:10.1212\/WNL.90.15_supplement.P2.123","DOI":"10.1212\/WNL.90.15_supplement.P2.123"},{"key":"2025082800220410000_17.7.e260998.9","doi-asserted-by":"crossref","first-page":"453","DOI":"10.1016\/j.ejpn.2014.02.001","article-title":"Shapiro\u2019s syndrome: defining the clinical spectrum of the spontaneous paroxysmal hypothermia syndrome","volume":"18","author":"Tambasco","year":"2014","journal-title":"Eur J Paediatr Neurol"},{"key":"2025082800220410000_17.7.e260998.10","doi-asserted-by":"crossref","first-page":"911332","DOI":"10.3389\/fneur.2022.911332","article-title":"A new perspective of hypothalamic disease: Shapiro\u2019s syndrome","volume":"13","author":"Ren","year":"2022","journal-title":"Front Neurol"},{"key":"2025082800220410000_17.7.e260998.11","doi-asserted-by":"crossref","first-page":"359","DOI":"10.1093\/brain\/96.2.359","article-title":"Agenesis of the corpus callosum associated with relapsing hypothermia a clinico-pathological report","volume":"96","author":"No\u00ebl","year":"1973","journal-title":"Brain (Bacau)"},{"key":"2025082800220410000_17.7.e260998.12","doi-asserted-by":"publisher","DOI":"10.1055\/s-2008-1052343"},{"key":"2025082800220410000_17.7.e260998.13","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1016\/0887-8994(94)90069-8","article-title":"Episodic spontaneous hypothermia with hyperhidrosis: implications for pathogenesis","volume":"10","author":"Sheth","year":"1994","journal-title":"Pediatr Neurol"},{"key":"2025082800220410000_17.7.e260998.14","doi-asserted-by":"crossref","first-page":"378","DOI":"10.1111\/j.1469-8749.2010.03854.x","article-title":"Spontaneous periodic hypothermia and hyperhidrosis: a possibly novel cerebral neurotransmitter disorder","volume":"53","author":"Rodrigues Masruha","year":"2011","journal-title":"Dev Med Child Neurol"},{"key":"2025082800220410000_17.7.e260998.15","doi-asserted-by":"crossref","first-page":"190","DOI":"10.1111\/j.1469-8749.2011.04145.x","article-title":"Spontaneous periodic hypothermia and hyperhidrosis: its relation with spontaneous endogenous hypermelatoninemia","volume":"54","author":"Duman","year":"2012","journal-title":"Dev Med Child Neurol"},{"key":"2025082800220410000_17.7.e260998.16","doi-asserted-by":"publisher","DOI":"10.1136\/pgmj.2007.066050"},{"key":"2025082800220410000_17.7.e260998.17","doi-asserted-by":"crossref","first-page":"153","DOI":"10.7326\/0003-4819-102-2-153","article-title":"Hemodynamic characteristics of patients with hypothermia due to occult infection and other causes","volume":"102","author":"Morris","year":"1985","journal-title":"Ann Intern Med"},{"key":"2025082800220410000_17.7.e260998.18","doi-asserted-by":"publisher","DOI":"10.2214\/AJR.05.0146"},{"key":"2025082800220410000_17.7.e260998.19","first-page":"235","article-title":"Clonidine therapy for Shapiro\u2019s syndrome","volume":"82","author":"Walker","year":"1992","journal-title":"Q J Med"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2024-260998","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T07:22:16Z","timestamp":1756365736000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2024-260998"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7]]},"references-count":19,"journal-issue":{"issue":"7","published-online":{"date-parts":[[2024,7,29]]},"published-print":{"date-parts":[[2024,7]]}},"alternative-id":["10.1136\/bcr-2024-260998"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2024-260998","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2024,7]]}}}