{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,25]],"date-time":"2026-09-25T22:48:31Z","timestamp":1790376511830,"version":"4.1.0"},"reference-count":55,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2021,2,12]],"date-time":"2021-02-12T00:00:00Z","timestamp":1613088000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Toxins"],"abstract":"<jats:p>Opisthotonus refers to abnormal axial extension and arching of the trunk produced by excessive contractions of the paraspinal muscles. In childhood, the abnormal posture is most often related to dystonia in the setting of hypoxic injury or a number of other acquired and genetic etiologies. The condition is often painful, interferes with ambulation and quality of life, and is challenging to treat. Therapeutic options include oral benzodiazepines, oral and intrathecal baclofen, botulinum neurotoxin injections, and deep brain stimulation. Management of opisthotonus within the pediatric population has not been systematically reviewed. Here, we describe a series of seven children who presented to our institution with opisthotonus in whom symptom relief was achieved following administration of botulinum neurotoxin injections.<\/jats:p>","DOI":"10.3390\/toxins13020137","type":"journal-article","created":{"date-parts":[[2021,2,12]],"date-time":"2021-02-12T22:41:25Z","timestamp":1613169685000},"page":"137","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":15,"title":["Botulinum Neurotoxin Injections in Childhood Opisthotonus"],"prefix":"10.3390","volume":"13","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7340-1660","authenticated-orcid":false,"given":"Mariam","family":"Hull","sequence":"first","affiliation":[{"name":"Section of Pediatric Neurology and Developmental Neuroscience, Texas Children\u2019s Hospital and Baylor College of Medicine, Houston, TX 77030, USA"},{"name":"Parkinson\u2019s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2102-4282","authenticated-orcid":false,"given":"Mered","family":"Parnes","sequence":"additional","affiliation":[{"name":"Section of Pediatric Neurology and Developmental Neuroscience, Texas Children\u2019s Hospital and Baylor College of Medicine, Houston, TX 77030, USA"},{"name":"Parkinson\u2019s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joseph","family":"Jankovic","sequence":"additional","affiliation":[{"name":"Parkinson\u2019s Disease Center and Movement Disorders Clinic, Department of Neurology, Baylor College of Medicine, Houston, TX 77030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2021,2,12]]},"reference":[{"key":"ref_1","first-page":"492","article-title":"Case of Opisthotonos, Successfully Treated","volume":"10","author":"Nayler","year":"1803","journal-title":"Med. Phys. J."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"868","DOI":"10.1002\/mds.20831","article-title":"Arc de cercle and dysautonomia from anoxic injury","volume":"21","author":"Diesing","year":"2006","journal-title":"Mov. Disord."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"11","DOI":"10.1016\/B978-0-12-801772-2.00002-3","article-title":"Charcot, hysteria, and simulated disorders","volume":"139","author":"Goetz","year":"2016","journal-title":"Handb. Clin. Neurol."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1121","DOI":"10.1212\/CON.0000000000000755","article-title":"Psychogenic (Functional) Movement Disorders","volume":"25","author":"Thenganatt","year":"2019","journal-title":"Contin. Lifelong Learn. Neurol."},{"key":"ref_5","first-page":"641","article-title":"Opisthotonus and intrathecal treatment with baclofen (ITB) in children","volume":"167","author":"Ceulemans","year":"2007","journal-title":"Eur. J. Nucl. Med. Mol. Imaging"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.jns.2018.02.005","article-title":"Tardive syndromes","volume":"389","author":"Savitt","year":"2018","journal-title":"J. Neurol. Sci."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"253","DOI":"10.1007\/s00702-019-01986-1","article-title":"Hand, foot, and spine deformities in parkinsonian disorders","volume":"126","author":"Wijemanne","year":"2019","journal-title":"J. Neural Transm."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"240","DOI":"10.1212\/CPJ.0000000000000453","article-title":"Camptocormia: Etiology, Diagnosis, and Treatment Response","volume":"8","author":"Ali","year":"2018","journal-title":"Neurol. Clin. Pract."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"339","DOI":"10.1002\/mds.10096","article-title":"Intrafamilial phenotypic variability of the DYT1 dystonia: From asymptomaticTOR1A gene carrier status to dystonic storm","volume":"17","author":"Opal","year":"2002","journal-title":"Mov. Disord."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1111\/dmcn.12339","article-title":"Status dystonicus: A practice guide","volume":"56","author":"Allen","year":"2014","journal-title":"Dev. Med. Child Neurol."},{"key":"ref_11","first-page":"71","article-title":"Status dystonicus: Diagnosis and management of a rare and challenging entity","volume":"12","author":"Thakkar","year":"2019","journal-title":"J. Pediatr. Rehabil. Med."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"1694","DOI":"10.1542\/peds.2013-1030","article-title":"Neonatal Death Suspected To Be From Sepsis Was Found To Be Kernicterus With G6PD Deficiency","volume":"132","author":"Christensen","year":"2013","journal-title":"Pediatrics"},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"331","DOI":"10.1159\/000479324","article-title":"Intrathecal Baclofen Pump Implantation for Type 2 Gaucher Disease","volume":"52","author":"Hori","year":"2017","journal-title":"Pediatr. Neurosurg."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"645","DOI":"10.1177\/0883073811424465","article-title":"Neurologic Presentation, Diagnostics, and Therapeutic Insights in a Severe Case of Adenylosuccinate Lyase Deficiency","volume":"27","author":"Jurecka","year":"2011","journal-title":"J. Child Neurol."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"1325","DOI":"10.1002\/mds.25490","article-title":"Dystonic opisthotonus: A \u201cred flag\u201d for neurodegeneration with brain iron accumulation syndromes?","volume":"28","author":"Stamelou","year":"2013","journal-title":"Mov. Disord."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"88","DOI":"10.1542\/peds.63.1.88","article-title":"L-Glutaric acidemia: Investigation of a patient and his family","volume":"63","author":"Whelan","year":"1979","journal-title":"Pediatrics"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"284","DOI":"10.1111\/j.1442-200X.1994.tb03181.x","article-title":"A case of maple syrup urine disease misdiagnosed as Tetanus neonatorum on admission","volume":"36","author":"Kleijer","year":"1994","journal-title":"Pediatr. Int."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"239","DOI":"10.1007\/s11940-002-0041-1","article-title":"Meningitis in the neonate","volume":"4","author":"Kimberlin","year":"2002","journal-title":"Curr. Treat. Options Neurol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"304","DOI":"10.1186\/1752-1947-4-304","article-title":"Paroxysmal autonomic instability with dystonia in a patient with tuberculous meningitis: A case report","volume":"4","author":"Ramdhani","year":"2010","journal-title":"J. Med. Case Rep."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"1244","DOI":"10.3346\/jkms.2011.26.9.1244","article-title":"Dopa-responsive Dystonia with a Novel Initiation Codon Mutation in the GCH1 Gene Misdiagnosed as Cerebral Palsy","volume":"26","author":"Lee","year":"2011","journal-title":"J. Korean Med. Sci."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1007\/s10545-009-1076-1","article-title":"Aromatic l-amino acid decarboxylase deficiency: Clinical features, drug therapy and follow-up","volume":"32","author":"Manegold","year":"2009","journal-title":"J. Inherit. Metab. Dis."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"426","DOI":"10.1590\/0004-282x20180047","article-title":"Opisthotonus (arc de cercle) in anti-NMDAR encephalitis","volume":"76","year":"2018","journal-title":"Arq. Neuro Psiquiatr."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1168","DOI":"10.1002\/1531-8257(200011)15:6<1168::AID-MDS1016>3.0.CO;2-V","article-title":"Markedly severe dystonia in Japanese encephalitis","volume":"15","author":"Kalita","year":"2000","journal-title":"Mov. Disord."},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"2192","DOI":"10.1002\/mds.21170","article-title":"Neurological manifestations in Wilson\u2019s disease: Report of 119 cases","volume":"21","author":"Machado","year":"2006","journal-title":"Mov. Disord."},{"key":"ref_25","doi-asserted-by":"crossref","first-page":"6865","DOI":"10.4076\/1757-1626-2-6865","article-title":"Titration of high dose sedation is effective in severe tetanus: A case report","volume":"2","author":"Chun","year":"2009","journal-title":"Cases J."},{"key":"ref_26","doi-asserted-by":"crossref","first-page":"2266","DOI":"10.1016\/S0140-6736(14)61832-8","article-title":"An Old Woman with Pressure Ulcer, Rigidity, and Opisthotonus: Never For-get Tetanus!","volume":"384","author":"Verde","year":"2014","journal-title":"Lancet"},{"key":"ref_27","doi-asserted-by":"crossref","first-page":"6","DOI":"10.1016\/j.eucr.2015.12.004","article-title":"Voiding Dysfunction Induced by Tetanus: A Case Report","volume":"5","author":"Kira","year":"2016","journal-title":"Urol. Case Rep."},{"key":"ref_28","doi-asserted-by":"crossref","first-page":"415","DOI":"10.1016\/S0031-3955(16)33714-2","article-title":"Tetanus","volume":"26","author":"Stoll","year":"1979","journal-title":"Pediatr. Clin. N. Am."},{"key":"ref_29","doi-asserted-by":"crossref","first-page":"512","DOI":"10.4103\/0019-5359.58881","article-title":"Recurrent opisthotonus in catatonia: An atypical presentation","volume":"63","author":"Manjunatha","year":"2009","journal-title":"Indian J. Med. Sci."},{"key":"ref_30","doi-asserted-by":"crossref","first-page":"788","DOI":"10.1136\/jnnp.63.6.788","article-title":"Clinical features and natural history of axial predominant adult onset primary dystonia","volume":"63","author":"Bhatia","year":"1997","journal-title":"J. Neurol. Neurosurg. Psychiatry"},{"key":"ref_31","doi-asserted-by":"crossref","first-page":"15","DOI":"10.3329\/bmrcb.v35i1.2313","article-title":"Neurological findings and outcome in adult cerebral malaria","volume":"35","author":"Sattar","year":"2009","journal-title":"Bangladesh Med. Res. Counc. Bull."},{"key":"ref_32","doi-asserted-by":"crossref","first-page":"613","DOI":"10.1016\/j.jemermed.2008.04.038","article-title":"Opisthotonus in Neurosyphilis: A Case Report","volume":"41","author":"Chauhan","year":"2011","journal-title":"J. Emerg. Med."},{"key":"ref_33","doi-asserted-by":"crossref","first-page":"257","DOI":"10.1111\/j.1365-2044.1988.tb05582.x","article-title":"Opisthotonos and Propofol: A Possible Association","volume":"43","author":"Laycock","year":"1988","journal-title":"Anaesthesia"},{"key":"ref_34","doi-asserted-by":"crossref","first-page":"905","DOI":"10.1111\/j.1365-2044.1988.tb05640.x","article-title":"Propofol Opisthotonus and Epilpesy","volume":"43","author":"Jones","year":"2007","journal-title":"Anaesthesia"},{"key":"ref_35","doi-asserted-by":"crossref","first-page":"1288","DOI":"10.3109\/15563650.2014.974264","article-title":"Acute toxicity associated with the recreational use of the novel dissociative psychoactive substance methoxphenidine","volume":"52","author":"Hofer","year":"2014","journal-title":"Clin. Toxicol."},{"key":"ref_36","doi-asserted-by":"crossref","first-page":"338","DOI":"10.1016\/j.jocn.2019.07.052","article-title":"Posterior fossa pilocytic astrocytoma presenting with opisthotonus in an infant\u2014A case report","volume":"68","author":"Yates","year":"2019","journal-title":"J. Clin. Neurosci."},{"key":"ref_37","doi-asserted-by":"crossref","first-page":"176","DOI":"10.1002\/mdc3.12249","article-title":"Axial Dystonia Mimicking Stiff Person Syndrome","volume":"3","author":"Balint","year":"2015","journal-title":"Mov. Disord. Clin. Pract."},{"key":"ref_38","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1111\/j.1365-2044.1993.tb07035.x","article-title":"Opisthotonus and Hysteria","volume":"48","author":"Crabb","year":"1993","journal-title":"Anaesthesia"},{"key":"ref_39","doi-asserted-by":"crossref","first-page":"379","DOI":"10.1016\/j.ncl.2020.01.004","article-title":"Treatment of Tardive Dyskinesia","volume":"38","author":"Bashir","year":"2020","journal-title":"Neurol. Clin."},{"key":"ref_40","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1002\/mds.870010305","article-title":"Natural history and treatment of tardive dystonia","volume":"1","author":"Kang","year":"1986","journal-title":"Mov. Disord."},{"key":"ref_41","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1002\/mds.870130129","article-title":"Geste Device in Tardive Dystonia with Retrocollis and Opisthotonic Pos-turing","volume":"13","author":"Krack","year":"1998","journal-title":"Mov. Disord."},{"key":"ref_42","doi-asserted-by":"crossref","first-page":"2053","DOI":"10.1093\/brain\/121.11.2053","article-title":"The natural history of tardive dystonia. A long-term follow-up study of 107 cases","volume":"121","author":"Kiriakakis","year":"1998","journal-title":"Brain"},{"key":"ref_43","doi-asserted-by":"crossref","first-page":"525","DOI":"10.1007\/s40265-018-0874-x","article-title":"Treatment of Tardive Dyskinesia: A General Overview with Focus on the Vesicular Monoamine Transporter 2 Inhibitors","volume":"78","author":"Niemann","year":"2018","journal-title":"Drugs"},{"key":"ref_44","doi-asserted-by":"crossref","first-page":"354","DOI":"10.1212\/CPJ.0000000000000485","article-title":"Arc de cercle as a manifestation of focal epileptic seizures","volume":"8","author":"Knight","year":"2018","journal-title":"Neurol. Clin. Pract."},{"key":"ref_45","doi-asserted-by":"crossref","first-page":"1201","DOI":"10.1002\/mds.1215","article-title":"Intrathecal baclofen for dystonia","volume":"16","author":"Hou","year":"2001","journal-title":"Mov. Disord."},{"key":"ref_46","doi-asserted-by":"crossref","first-page":"627","DOI":"10.1002\/mdc3.12829","article-title":"Effects of Deep Brain Stimulation on Postural Trunk Deformities: A Systematic Review","volume":"6","author":"Lizarraga","year":"2019","journal-title":"Mov. Disord. Clin. Pract."},{"key":"ref_47","doi-asserted-by":"crossref","first-page":"1141","DOI":"10.1212\/WNL.41.7.1141","article-title":"Intrathecal Baclofen for Intractable Axial Dysto-nia","volume":"41","author":"Narayan","year":"1991","journal-title":"Neurology"},{"key":"ref_48","doi-asserted-by":"crossref","first-page":"1131","DOI":"10.1002\/mds.27072","article-title":"Botulinum toxin: State of the art","volume":"32","author":"Jankovic","year":"2017","journal-title":"Mov. Disord."},{"key":"ref_49","doi-asserted-by":"crossref","unstructured":"Anandan, C., and Jankovic, J. (2021). Botulinum Toxin in Movement Disorders: An Update. Toxins, 13.","DOI":"10.3390\/toxins13010042"},{"key":"ref_50","doi-asserted-by":"crossref","first-page":"84","DOI":"10.1016\/j.toxicon.2017.09.003","article-title":"An update on new and unique uses of botulinum toxin in movement disorders","volume":"147","author":"Jankovic","year":"2018","journal-title":"Toxicon"},{"key":"ref_51","doi-asserted-by":"crossref","first-page":"610","DOI":"10.1136\/bmj.1.855.610","article-title":"A Case of Opisthotonos Cured by Chloral-Hydrate","volume":"1","author":"Davis","year":"1877","journal-title":"BMJ"},{"key":"ref_52","doi-asserted-by":"crossref","first-page":"189","DOI":"10.2310\/7010.2006.00041","article-title":"Safety of high-dose botulinum toxin type A therapy for the treatment of pediatric spasticity","volume":"21","author":"Goldstein","year":"2006","journal-title":"J. Child Neurol."},{"key":"ref_53","doi-asserted-by":"crossref","first-page":"818","DOI":"10.1111\/j.1469-8749.2007.00818.x","article-title":"High dose botulinum toxin A for the treatment of lower extremity hypertonicity in children with cerebral palsy","volume":"49","author":"Willis","year":"2007","journal-title":"Dev. Med. Child Neurol."},{"key":"ref_54","doi-asserted-by":"crossref","first-page":"210","DOI":"10.1177\/0883073813495306","article-title":"Botulinum Toxin Type a in Children and Adolescents with Severe Cerebral Palsy: A Retrospective Chart Review","volume":"29","author":"Mesterman","year":"2014","journal-title":"J. Child Neurol."},{"key":"ref_55","doi-asserted-by":"crossref","first-page":"371","DOI":"10.1177\/0883073819830579","article-title":"Botulinum Toxins Type A (Bont-A) in the Management of Lower Limb Spasticity in Children: A Systematic Literature Review and Bayesian Network Meta-analysis","volume":"34","author":"Guyot","year":"2019","journal-title":"J. Child Neurol."}],"container-title":["Toxins"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2072-6651\/13\/2\/137\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T05:23:35Z","timestamp":1760160215000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2072-6651\/13\/2\/137"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,2,12]]},"references-count":55,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2021,2]]}},"alternative-id":["toxins13020137"],"URL":"https:\/\/doi.org\/10.3390\/toxins13020137","relation":{},"ISSN":["2072-6651"],"issn-type":[{"value":"2072-6651","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,2,12]]}}}