{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,4,17]],"date-time":"2025-04-17T04:08:03Z","timestamp":1744862883097,"version":"3.40.4"},"reference-count":12,"publisher":"PAGEPress Publications","license":[{"start":{"date-parts":[[2025,4,16]],"date-time":"2025-04-16T00:00:00Z","timestamp":1744761600000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Eur J Transl Myol"],"abstract":"<jats:p>Objective: To report on a patient with hypermobile Ehlers-Danlos syndrome (EDS) with SFN in whom frequent dislocations during sleep led to sleep deprivation.\r\nCase report: The patient is a 19-year-old female with hypermobile EDS manifested by frequent dislocations of large joints and small fiber neuropathy (SFN) presenting as recurrent syncope. The dislocations occurred spontaneously or triggered by voluntary or involuntary movements with such frequency, even during the night, that sleep was disturbed. The maximum sleep duration was between 4 and 5 hours. Bedding, analgesics, muscle relaxants and physiotherapy only marginally improved the pain and discomfort caused by the dislocations.\r\nConclusions: This case demonstrates that hypermobile EDS can manifest phenotypically with insomnia due to pain and discomfort from frequent dislocations during the night. Patients with hypermobile EDS should undergo polysomnography to determine the cause of the sleep disturbance and initiate the most appropriate treatment for insomnia in a timely manner.\r\n\u00a0\r\n\u00a0<\/jats:p>","DOI":"10.4081\/ejtm.2025.12946","type":"journal-article","created":{"date-parts":[[2025,4,16]],"date-time":"2025-04-16T09:18:37Z","timestamp":1744795117000},"source":"Crossref","is-referenced-by-count":0,"title":["Insomnia due to a dislocation storm in hypermobile Ehlers-Danlos syndrome with small fibre neuropathy and recurrent syncope"],"prefix":"10.4081","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2839-7305","authenticated-orcid":false,"given":"Josef","family":"Finsterer","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"2549","published-online":{"date-parts":[[2025,4,16]]},"reference":[{"key":"174534","unstructured":"Malfait F, Symoens S, Syx D. Classic Ehlers-Danlos Syndrome. 2007 May 29 [Updated 2024 Feb 1]. In: Adam MP, Feldman J, Mirzaa GM, et al., editors. GeneReviews\u00ae [Internet]. Seattle (WA): University of Washington, Seattle; 1993-2024. Available from: https:\/\/www.ncbi.nlm.nih.gov\/books\/NBK1244\/"},{"key":"174535","doi-asserted-by":"crossref","unstructured":"Housset V, Nourissat G. Arthroscopic capsular plication for multidirectional shoulder instability in hypermobile Ehlers-Danlos syndrome patients. Arthrosc Tech 2021;10:e2767-73.","DOI":"10.1016\/j.eats.2021.08.024"},{"key":"174536","doi-asserted-by":"crossref","unstructured":"Fernandez A, Aubry-Rozier B, Vautey M, et al. Small fiber neuropathy in hypermobile Ehlers Danlos syndrome\/hypermobility spectrum disorder. J Intern Med 2022;292:957-60.","DOI":"10.1111\/joim.13539"},{"key":"174537","doi-asserted-by":"crossref","unstructured":"Pascarella A, Provitera V, Lullo F, et al. Evidence of small fiber neuropathy in a patient with Ehlers-Danlos syndrome, hypermobility-type. Clin Neurophysiol 2016;127:1914-6.","DOI":"10.1016\/j.clinph.2015.12.004"},{"key":"174538","unstructured":"Hakim A. Hypermobile Ehlers-Danlos Syndrome. 2004. In: Adam MP, Feldman J, Mirzaa GM, Pagon RA, Wallace SE, Bean LJH, Gripp KW, Amemiya A, editors. GeneReviews\u00ae [Internet]. Seattle (WA): University of Washington, Seattle; 1993\u20132024."},{"key":"174539","doi-asserted-by":"crossref","unstructured":"Miller SM, Miller MB. Treatment of obstructive sleep apnea with hypoglossal nerve stimulation in a patient with Ehlers-Danlos syndrome. J Clin Sleep Med 2023;19:631-2.","DOI":"10.5664\/jcsm.10368"},{"key":"174540","doi-asserted-by":"crossref","unstructured":"Khan MH, Ahmed U. Narcolepsy and orthostatic intolerance in an adult with Ehlers-Danlos syndrome. Prim Care Companion CNS Disord 2022;24:21cr03138.","DOI":"10.4088\/PCC.21cr03138"},{"key":"174541","doi-asserted-by":"crossref","unstructured":"Sedky K, Gaisl T, Bennett DS. Prevalence of obstructive sleep apnea in joint hypermobility syndrome: a systematic review and meta-analysis. J Clin Sleep Med 2019;15:293-99.","DOI":"10.5664\/jcsm.7636"},{"key":"174542","doi-asserted-by":"crossref","unstructured":"St\u00f6berl AS, Gaisl T, Giunta C, et al. Obstructive sleep apnoea in children and adolescents with Ehlers-Danlos syndrome. Respiration 2019;97:284-91.","DOI":"10.1159\/000494328"},{"key":"174543","doi-asserted-by":"crossref","unstructured":"Domany KA, Hantragool S, Smith DF, et al. Sleep disorders and their management in children with Ehlers-Danlos syndrome referred to sleep clinics. J Clin Sleep Med 2018;14:623-9.","DOI":"10.5664\/jcsm.7058"},{"key":"174544","doi-asserted-by":"crossref","unstructured":"Gaisl T, Giunta C, Bratton DJ, et al. Obstructive sleep apnoea and quality of life in Ehlers-Danlos syndrome: a parallel cohort study. Thorax 2017;72:729-35.","DOI":"10.1136\/thoraxjnl-2016-209560"},{"key":"174545","unstructured":"Sumi Y, Kadotani H. [Sleep Disorders and Autonomic Nervous System Dysfunction]. Brain Nerve 2022;74:279-82. Japanese."}],"container-title":["European Journal of Translational Myology"],"original-title":[],"link":[{"URL":"https:\/\/www.pagepressjournals.org\/bam\/article\/download\/12946\/13017","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.pagepressjournals.org\/bam\/article\/download\/12946\/13017","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,16]],"date-time":"2025-04-16T09:18:41Z","timestamp":1744795121000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.pagepressjournals.org\/bam\/article\/view\/12946"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,4,16]]},"references-count":12,"URL":"https:\/\/doi.org\/10.4081\/ejtm.2025.12946","relation":{},"ISSN":["2037-7460","2037-7452"],"issn-type":[{"value":"2037-7460","type":"electronic"},{"value":"2037-7452","type":"print"}],"subject":[],"published":{"date-parts":[[2025,4,16]]}}}