{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,20]],"date-time":"2026-07-20T14:17:47Z","timestamp":1784557067223,"version":"3.55.0"},"reference-count":36,"publisher":"Bioscientifica","issue":"11","license":[{"start":{"date-parts":[[2024,9,5]],"date-time":"2024-09-05T00:00:00Z","timestamp":1725494400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["ec.bioscientifica.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,9,5]]},"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Rapid-onset obesity with hypoventilation, hypothalamic dysfunction, and autonomic dysregulation (ROHHAD) is rare, and manifestations of autonomic dysregulation are diverse and may be overlooked. We aimed to evaluate the incidence of these manifestations.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Patients with ROHHAD syndrome reported before and after 2019 were divided into groups 1 and 2. Patients who were diagnosed at three regional hospitals in China were included in group 3. We collected the age of each specific term of the ROHHAD (neurogenic tumor, NET) acronym and the detailed manifestations of each term, and compared them among the three groups.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      A total of 16 patients were diagnosed within the 2-year period. Two had neurogenic tumors and cognitive and behavioral abnormalities before developing rapid obesity. At least 93.8% of the patients had \u2265 4 symptoms of autonomic dysregulation. When comparing autonomic dysregulation among groups 1\u20133, the rates of cardiovascular manifestations were NA vs 12.8% vs 81.2%; gastrointestinal disturbances were 11.4% vs 8.5% vs 62.5%; strabismus was 25.7% vs 12.8% vs 62.5%; sleep disturbance was NA vs 6.4% vs 50.0%; and abnormal pain threshold was NA vs 10.6% vs 25.0% (all\n                      <jats:italic>P<\/jats:italic>\n                      &lt; 0.05). The rates of cognitive and behavioral abnormalities were NA vs 29.8% and 87.5% (\n                      <jats:italic>P<\/jats:italic>\n                      &lt; 0.01).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Rapid-onset obesity is not always the first sign of ROHHAD syndrome. Higher rates of autonomic dysregulation and cognitive and behavioral abnormalities with multiple manifestations of autonomic dysregulation coexisted in our cohort, indicating that evaluations of autonomic function and the limbic system should be strengthened when assessing this condition.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1530\/ec-24-0189","type":"journal-article","created":{"date-parts":[[2024,9,5]],"date-time":"2024-09-05T11:46:44Z","timestamp":1725536804000},"update-policy":"https:\/\/doi.org\/10.1530\/crossmarkpolicy-10","source":"Crossref","is-referenced-by-count":4,"title":["Emphasizing autonomic dysregulation evaluation contributes to the diagnosis of ROHHAD syndrome"],"prefix":"10.1530","volume":"13","author":[{"given":"Yi","family":"Wang","sequence":"first","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yingying","family":"Xu","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rongrong","family":"Xie","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Children\u2019s Hospital of Soochow University, Suzhou, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bingyan","family":"Cao","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuan","family":"Ding","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiayun","family":"Guo","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaoqiao","family":"Li","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaolin","family":"Ni","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zheng","family":"Yuan","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linqi","family":"Chen","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Children\u2019s Hospital of Soochow University, Suzhou, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liyang","family":"Liang","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Sun Yat-sen Memorial Hospital, Sun Yat-sen University, Guangzhou, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1262-7383","authenticated-orcid":true,"given":"Chunxiu","family":"Gong","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Genetics and Metabolism, Beijing Children\u2019s Hospital, Capital Medical University, Beijing, China"},{"name":"National Center for Children\u2019s Health, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"416","reference":[{"key":"bib1","series-title":"American Journal of Diseases of Children","first-page":"155","article-title":"Primary alveolar hypoventilation syndrome (Ondine\u2019s curse)","volume":"110","author":"Fishman","year":"1965","unstructured":"Fishman LSSamson JH & Sperling DR. 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(https:\/\/doi.org\/10.3389\/fped.2022.910099)"},{"key":"bib7","series-title":"Journal of Clinical Endocrinology and Metabolism","first-page":"dgaa247","article-title":"ROHHAD(NET) syndrome: systematic review of the clinical timeline and recommendations for diagnosis and prognosis","volume":"105","author":"Harvengt","year":"2020","unstructured":"Harvengt JGernay CMastouri MFarhat NLebrethon MCSeghaye MC & Bours V. ROHHAD(NET) syndrome: systematic review of the clinical timeline and recommendations for diagnosis and prognosis. Journal of Clinical Endocrinology and Metabolism2020105dgaa247. (https:\/\/doi.org\/10.1210\/clinem\/dgaa247)"},{"key":"bib8","series-title":"Journal of the Endocrine Society","first-page":"bvad103","article-title":"Blood lymphocyte subsets and proinflammatory cytokine profile in ROHHAD(NET) and non-ROHHAD(NET) obese individuals","volume":"7","author":"Fava","year":"2023","unstructured":"Fava DMorandi FPrigione IAngelelli ABocca PPistorio AVolpi SPatti GPepino CCasalini E, et al.Blood lymphocyte subsets and proinflammatory cytokine profile in ROHHAD(NET) and non-ROHHAD(NET) obese individuals. Journal of the Endocrine Society20237bvad103. (https:\/\/doi.org\/10.1210\/jendso\/bvad103)"},{"key":"bib9","series-title":"Journal of Pediatric Endocrinology and Metabolism","first-page":"1102","article-title":"Rituximab therapy in ROHHAD(NET) syndrome","volume":"35","author":"Hawton","year":"2022","unstructured":"Hawton KACDoffinger RRamanan AVLangton Hewer SCEvans HJGiri D & Shield JPH. Rituximab therapy in ROHHAD(NET) syndrome. 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Case report: molecular characterisation of adipose-tissue derived cells from a patient with ROHHAD syndrome. Frontiers in Pediatrics2023111128216. (https:\/\/doi.org\/10.3389\/fped.2023.1128216)"},{"key":"bib12","series-title":"Journal of Pediatric Endocrinology and Metabolism","first-page":"418","article-title":"Rapid-onset obesity, hypothalamic dysfunction, hypoventilation, and autonomic dysregulation syndrome - neuro-endocrine tumours (ROHHAD-NET): case series and learning points","volume":"36","author":"Hawton","year":"2023","unstructured":"Hawton KHilliard TLangton-Hewer SCBurren CCrowne ECHamilton-Shield JP & Giri D. Rapid-onset obesity, hypothalamic dysfunction, hypoventilation, and autonomic dysregulation syndrome - neuro-endocrine tumours (ROHHAD-NET): case series and learning points. Journal of Pediatric Endocrinology and Metabolism202336418\u2013423. (https:\/\/doi.org\/10.1515\/jpem-2022-0376)"},{"key":"bib13","series-title":"Frontiers in Pediatrics","first-page":"919921","article-title":"Case Report: considerations of nocturnal ventilator support in ROHHAD syndrome in chronic care of childhood central hypoventilation with hypothalamus dysfunction","volume":"10","author":"Zhao","year":"2022","unstructured":"Zhao RDong XGao Z & Han F. Case Report: considerations of nocturnal ventilator support in ROHHAD syndrome in chronic care of childhood central hypoventilation with hypothalamus dysfunction. Frontiers in Pediatrics202210919921. (https:\/\/doi.org\/10.3389\/fped.2022.919921)"},{"key":"bib14","series-title":"Annals of Neurology","first-page":"279","article-title":"ZSCAN1 autoantibodies are associated with pediatric paraneoplastic ROHHAD","volume":"92","author":"Mandel-Brehm","year":"2022","unstructured":"Mandel-Brehm CBenson LATran BKung AFMann SAVazquez SERetallack HSample HAZorn KCKhan LM, et al.ZSCAN1 autoantibodies are associated with pediatric paraneoplastic ROHHAD. Annals of Neurology202292279\u2013291. (https:\/\/doi.org\/10.1002\/ana.26380)"},{"key":"bib15","series-title":"Clinical Endocrinology","first-page":"72","article-title":"Identification of clinical factors related to antibody-mediated immune response to the subfornical organ","volume":"97","author":"Nakamura-Utsunomiya","year":"2022","unstructured":"Nakamura-Utsunomiya AGoda SHayakawa SSonoko SHoorn EJBlanchard ASaito-Hakoda AKakimoto HHachiya RKamimura M, et al.Identification of clinical factors related to antibody-mediated immune response to the subfornical organ. Clinical Endocrinology20229772\u201380. (https:\/\/doi.org\/10.1111\/cen.14737)"},{"key":"bib16","series-title":"Journal of Pediatric Genetics","first-page":"309","article-title":"Acute COVID-19 infection in a pediatric patient with ROHHAD","volume":"11","author":"Udrea","year":"2022","unstructured":"Udrea DSLopez MAvesar MQureshi SMoretti AAbd-Allah SA & Chandnani HK. Acute COVID-19 infection in a pediatric patient with ROHHAD. Journal of Pediatric Genetics202211309\u2013312. (https:\/\/doi.org\/10.1055\/s-0040-1718874)"},{"key":"bib17","series-title":"Anaesthesia Reports","first-page":"e12172","article-title":"Peri-operative management of a child with ROHHAD-NET syndrome undergoing neural crest tumour excision","volume":"10","author":"Puri","year":"2022","unstructured":"Puri SYaddanapudi S & Menon P. Peri-operative management of a child with ROHHAD-NET syndrome undergoing neural crest tumour excision. Anaesthesia Reports202210e12172. (https:\/\/doi.org\/10.1002\/anr3.12172)"},{"key":"bib18","series-title":"Frontiers in Pediatrics","first-page":"854367","article-title":"Case report: COVID-19-associated ROHHAD-like syndrome","volume":"10","author":"Artamonova","year":"2022","unstructured":"Artamonova INPetrova NALyubimova NAKolbina NYBryzzhin AVBorodin AVLevko TAMamaeva EAPervunina TMVasichkina ESet al.Case report: COVID-19-associated ROHHAD-like syndrome. Frontiers in Pediatrics202210854367. (https:\/\/doi.org\/10.3389\/fped.2022.854367)"},{"key":"bib19","series-title":"Pharmaceutics","first-page":"2148","article-title":"The effect of Zn-substitution on the morphological, magnetic, cytotoxic, and in vitro hyperthermia properties of polyhedral ferrite magnetic nanoparticles","volume":"13","author":"Fizesan","year":"2021","unstructured":"Fizesan IIacovita CPop AKiss BDudric RStiufiuc RLucaciu CM & Loghin F. The effect of Zn-substitution on the morphological, magnetic, cytotoxic, and in vitro hyperthermia properties of polyhedral ferrite magnetic nanoparticles. Pharmaceutics2021132148. (https:\/\/doi.org\/10.3390\/pharmaceutics13122148)"},{"key":"bib20","series-title":"International Journal of Endocrinology and Metabolism","first-page":"e111571","article-title":"A case report of ROHHAD syndrome in an 8-year-old Iranian boy","volume":"19","author":"Amjadipour","year":"2021","unstructured":"Amjadipour AShahkar L & Hanafi F. A case report of ROHHAD syndrome in an 8-year-old Iranian boy. International Journal of Endocrinology and Metabolism202119e111571. (https:\/\/doi.org\/10.5812\/ijem.111571)"},{"key":"bib21","series-title":"Journal of Clinical Sleep Medicine","first-page":"1723","article-title":"Chewing gum: alternative therapy to oxygen intolerance","volume":"18","author":"Sunkonkit","year":"2022","unstructured":"Sunkonkit KSelvadurai SYeh EAHamilton J & Narang I. Chewing gum: alternative therapy to oxygen intolerance. Journal of Clinical Sleep Medicine2022181723\u20131726. 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(https:\/\/doi.org\/10.3389\/fped.2020.573227)"},{"key":"bib25","series-title":"Journal of Clinical Lipidology","first-page":"789","article-title":"Rhabdomyolysis due to rosuvastatin in a patient with ROHHAD syndrome","volume":"15","author":"Vijayakanthi","year":"2021","unstructured":"Vijayakanthi NFelner EIRomero R & Daley TC. Rhabdomyolysis due to rosuvastatin in a patient with ROHHAD syndrome. Journal of Clinical Lipidology202115789\u2013792. (https:\/\/doi.org\/10.1016\/j.jacl.2021.09.002)"},{"key":"bib26","series-title":"Annals of Indian Academy of Neurology","first-page":"307","article-title":"Thoracic ganglioneuroma presenting with paraneoplastic cerebellitis and ROHHAD-NET syndrome","volume":"25","author":"Tiwari","year":"2022","unstructured":"Tiwari SArimbrakkunnan MYadav TVyas VRathod KK & Vishwajeet V. Thoracic ganglioneuroma presenting with paraneoplastic cerebellitis and ROHHAD-NET syndrome. Annals of Indian Academy of Neurology202225307\u2013309. 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