{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,26]],"date-time":"2026-09-26T05:30:36Z","timestamp":1790400636296,"version":"4.1.0"},"update-to":[{"DOI":"10.1371\/journal.pntd.0009430","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2021,6,30]],"date-time":"2021-06-30T00:00:00Z","timestamp":1625011200000}}],"reference-count":29,"publisher":"Public Library of Science (PLoS)","issue":"6","license":[{"start":{"date-parts":[[2021,6,18]],"date-time":"2021-06-18T00:00:00Z","timestamp":1623974400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.plosntds.org"],"crossmark-restriction":false},"short-container-title":["PLoS Negl Trop Dis"],"abstract":"<jats:sec id=\"sec001\">\n<jats:title>Background<\/jats:title>\n<jats:p>The term Nodding Syndrome (NS) refers to an atypical and severe form of childhood epilepsy characterized by a repetitive head nodding (HN). The disease has been for a long time limited to East Africa, and the cause is still unknown. The objective of this study was to confirm the existence of NS cases in Central African Republic (CAR).<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec002\">\n<jats:title>Methodology\/Principal findings<\/jats:title>\n<jats:p>This was a cross-sectional descriptive study in the general population. The identification of NS cases was conducted through a door-to-door survey in a village near Bangui along the Ubangui River. Based on Winkler\u2019s 2008 and the World Health Organization (WHO)\u2019s 2012 classifications, the confirmation of cases was done by a neurologist who also performed the electroencephalograms. No laboratory tests were done during this investigation. Treatment was offered to all patients. A total of 6,175 individuals was surveyed in 799 households. After reviewing the cases, we identified 5 NS cases in girls aged between 8 and 16. The age of onset of the seizures was between 5 and 12 years of age. Two cases were classified as \"HN plus\" according to Winkler\u2019s 2008 classification. Four NS cases were classified as probable and one as confirmed according to the WHO\u2019s 2012 classification. Three of them presented with developmental delay and cognitive decline, and one had an abnormally low height-for-age z-score. Electroencephalographic abnormalities were found in four patients.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"sec003\">\n<jats:title>Conclusions\/Significance<\/jats:title>\n<jats:p>Nodding Syndrome cases were described in CAR for the first time. Despite certain peculiarities, these cases are similar to those described elsewhere. Given that only a small part of the affected area was investigated, the study area along the Ubangui River needs to be expanded in order to investigate the association between <jats:italic>Onchocerca volvulus<\/jats:italic> and NS and also evaluate the real burden of NS in CAR.<\/jats:p>\n<\/jats:sec>","DOI":"10.1371\/journal.pntd.0009430","type":"journal-article","created":{"date-parts":[[2021,6,18]],"date-time":"2021-06-18T17:37:37Z","timestamp":1624037857000},"page":"e0009430","update-policy":"https:\/\/doi.org\/10.1371\/journal.pntd.corrections_policy","source":"Crossref","is-referenced-by-count":13,"title":["First description of Nodding Syndrome in the Central African Republic"],"prefix":"10.1371","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0020-3655","authenticated-orcid":true,"given":"Salvatore","family":"Metanmo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9505-683X","authenticated-orcid":true,"given":"Farid","family":"Boum\u00e9di\u00e8ne","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2171-2977","authenticated-orcid":true,"given":"Pierre-Marie","family":"Preux","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1919-1340","authenticated-orcid":true,"given":"Robert","family":"Colebunders","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3544-1239","authenticated-orcid":true,"given":"Joseph N.","family":"Siewe Fodjo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3010-851X","authenticated-orcid":true,"given":"Eric","family":"de Smet","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emmanuel","family":"Yangatimbi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3131-6983","authenticated-orcid":true,"given":"Andrea S.","family":"Winkler","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0054-7538","authenticated-orcid":true,"given":"Pascal","family":"Mbelesso","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel","family":"Ajzenberg","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2021,6,18]]},"reference":[{"issue":"12","key":"pntd.0009430.ref001","doi-asserted-by":"crossref","first-page":"714","DOI":"10.1016\/S1474-4422(03)00599-4","article-title":"Nodding disease: mystery of southern Sudan","volume":"2","author":"M Lacey","year":"2003","journal-title":"Lancet Neurol"},{"key":"pntd.0009430.ref002","unstructured":"World Health Organization. International Scientific Meeting on Nodding Syndrome 2012. https:\/\/www.who.int\/neglected_diseases\/diseases\/Nodding_syndrom_Kampala_Report_2012.pdf. Accessed January 14, 2021."},{"issue":"1","key":"pntd.0009430.ref003","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1111\/j.1600-0447.1965.tb04970.x","article-title":"Epilepsy in the Wapogoro Tribe in Tanganyika1","volume":"41","author":"LM Aall-Jilek","year":"1965","journal-title":"Acta Psychiatr Scand."},{"issue":"9","key":"pntd.0009430.ref004","doi-asserted-by":"crossref","first-page":"1374","DOI":"10.3201\/eid1909.130401","article-title":"Nodding syndrome","volume":"19","author":"SF Dowell","year":"2013","journal-title":"Emerg Infect Dis"},{"issue":"2","key":"pntd.0009430.ref005","doi-asserted-by":"crossref","first-page":"e0005201","DOI":"10.1371\/journal.pntd.0005201","article-title":"Nakalanga Syndrome: Clinical Characteristics, Potential Causes, and Its Relationship with Recently Described Nodding Syndrome","volume":"11","author":"K Foeger","year":"2017","journal-title":"PLoS Negl Trop Dis"},{"key":"pntd.0009430.ref006","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1016\/j.yebeh.2018.11.008","article-title":"Clinical presentations of onchocerciasis-associated epilepsy (OAE) in Cameroon","volume":"90","author":"JFN Siewe","year":"2019","journal-title":"Epilepsy Behav EB"},{"issue":"6","key":"pntd.0009430.ref007","doi-asserted-by":"crossref","first-page":"S506","DOI":"10.1016\/j.ibror.2019.07.1581","article-title":"Onchocerciasis-associated epilepsy in the Democratic Republic of Congo: Clinical description and relationship with microfilarial density.","volume":"1","author":"JNS Fodjo","year":"2019","journal-title":"IBRO Rep"},{"key":"pntd.0009430.ref008","first-page":"112","article-title":"Is nodding syndrome an Onchocerca volvulus-induced neuroinflammatory disorder? Uganda\u2019s story of research in understanding the disease","volume":"45","author":"R Idro","year":"2016","journal-title":"Int J Infect Dis IJID Off Publ Int Soc Infect Dis"},{"issue":"11","key":"pntd.0009430.ref009","doi-asserted-by":"crossref","first-page":"632","DOI":"10.1038\/s41582-018-0086-8","article-title":"Nodding syndrome discovered to be a tauopathy","volume":"14","author":"C Ridler","year":"2018","journal-title":"Nat Rev Neurol"},{"issue":"63(28)","key":"pntd.0009430.ref010","first-page":"603","article-title":"Prevalence of nodding syndrome\u2014Uganda, 2012\u20132013","volume":"18","author":"PJ Iyengar","year":"2014","journal-title":"MMWR Morb Mortal Wkly Rep"},{"issue":"5","key":"pntd.0009430.ref011","doi-asserted-by":"crossref","first-page":"e002540","DOI":"10.1136\/bmjopen-2012-002540","article-title":"Nodding syndrome in Ugandan children\u2014clinical features, brain imaging and complications: a case series","volume":"33","author":"R Idro","year":"2013","journal-title":"BMJ Open"},{"key":"pntd.0009430.ref012","doi-asserted-by":"crossref","first-page":"64","DOI":"10.1016\/j.seizure.2015.07.006","article-title":"Neurophysiological and clinical findings on Nodding Syndrome in 21 South Sudanese children and a review of the literature","volume":"31","author":"G de Polo","year":"2015","journal-title":"Seizure"},{"issue":"2","key":"pntd.0009430.ref013","doi-asserted-by":"crossref","first-page":"166","DOI":"10.1016\/S1474-4422(12)70321-6","article-title":"Clinical, neurological, and electrophysiological features of nodding syndrome in Kitgum, Uganda: an observational case series","volume":"12","author":"JJ Sejvar","year":"2013","journal-title":"Lancet Neurol"},{"issue":"12","key":"pntd.0009430.ref014","doi-asserted-by":"crossref","first-page":"2008","DOI":"10.1111\/j.1528-1167.2008.01671.x","article-title":"The head nodding syndrome\u2014clinical classification and possible causes","volume":"49","author":"AS Winkler","year":"2008","journal-title":"Epilepsia"},{"key":"pntd.0009430.ref015","doi-asserted-by":"crossref","first-page":"80","DOI":"10.1016\/j.ensci.2015.11.001","article-title":"Nodding syndrome: 2015 International Conference Report and Gulu Accord.","volume":"3","author":"PS Spencer","year":"2016","journal-title":"eNeurologicalSci"},{"issue":"11","key":"pntd.0009430.ref016","doi-asserted-by":"crossref","first-page":"e006476","DOI":"10.1136\/bmjopen-2014-006476","article-title":"Patients with nodding syndrome in Uganda improve with symptomatic treatment: a cross-sectional study","volume":"4","author":"R Idro","year":"2014","journal-title":"BMJ Open"},{"issue":"6","key":"pntd.0009430.ref017","doi-asserted-by":"crossref","first-page":"e66419","DOI":"10.1371\/journal.pone.0066419","article-title":"An Epidemiologic Investigation of Potential Risk Factors for Nodding Syndrome in Kitgum District, Uganda","volume":"18","author":"JL Foltz","year":"2013","journal-title":"PLoS ONE"},{"issue":"3","key":"pntd.0009430.ref018","first-page":"242","article-title":"Clinical and epidemiologic characteristics of nodding syndrome in Mundri County, southern Sudan","volume":"12","author":"JK Tumwine","year":"2012","journal-title":"Afr Health Sci"},{"issue":"3","key":"pntd.0009430.ref019","first-page":"307","article-title":"Sociocultural representations of epilepsy in schools in Bangui, Central African Republic","volume":"21","author":"P Mbelesso","year":"2009","journal-title":"Epilepsies"},{"issue":"7","key":"pntd.0009430.ref020","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1186\/s40249-018-0400-0","article-title":"Report of the first international workshop on onchocerciasis-associated epilepsy","volume":"22","author":"R Colebunders","year":"2018","journal-title":"Infect Dis Poverty"},{"issue":"5","key":"pntd.0009430.ref021","doi-asserted-by":"crossref","first-page":"337","DOI":"10.3390\/pathogens9050337","article-title":"Focus of Ongoing Onchocerciasis Transmission Close to Bangui, Central African Republic","volume":"9","author":"Metanmo S Smet E de","year":"2020","journal-title":"Pathog Basel Switz"},{"issue":"4","key":"pntd.0009430.ref022","doi-asserted-by":"crossref","first-page":"470","DOI":"10.1111\/j.0013-9580.2005.66104.x","article-title":"Epileptic seizures and epilepsy: definitions proposed by the International League Against Epilepsy (ILAE) and the International Bureau for Epilepsy (IBE).","volume":"46","author":"RS Fisher","year":"2005","journal-title":"Epilepsia"},{"issue":"246","key":"pntd.0009430.ref023","first-page":"1","article-title":"2000 CDC Growth Charts for the United States: methods and development","volume":"11","author":"RJ Kuczmarski","year":"2002","journal-title":"Vital Health Stat"},{"key":"pntd.0009430.ref024","unstructured":"World Health Organization. ICD-10 guide for mental retardation. 1996. https:\/\/apps.who.int\/iris\/handle\/10665\/63000. Accessed January 14, 2021"},{"key":"pntd.0009430.ref025","unstructured":"Emmanuel M, Bokor BR. Tanner Stages. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2020. http:\/\/www.ncbi.nlm.nih.gov\/books\/NBK470280\/ Accessed January 14, 2021"},{"issue":"5","key":"pntd.0009430.ref026","doi-asserted-by":"crossref","first-page":"1211","DOI":"10.4269\/ajtmh.18-0511","article-title":"Onchocerciasis-Associated Epilepsy with Head Nodding Seizures-Nodding Syndrome: A Case Series of 15 Patients from Western Uganda, 1994","volume":"99","author":"C Kaiser","year":"2018","journal-title":"Am J Trop Med Hyg"},{"issue":"6","key":"pntd.0009430.ref027","doi-asserted-by":"crossref","first-page":"508","DOI":"10.1684\/epd.2018.1012","article-title":"The natural history of nodding syndrome","volume":"20","author":"R Idro","year":"2018","journal-title":"Epileptic Disord Int Epilepsy J Videotape"},{"issue":"3","key":"pntd.0009430.ref028","doi-asserted-by":"crossref","first-page":"173","DOI":"10.1258\/td.2010.090373","article-title":"Clinical characteristics of people with head nodding in southern Tanzania","volume":"40","author":"AS Winkler","year":"2010","journal-title":"Trop Doct"},{"issue":"3","key":"pntd.0009430.ref029","first-page":"52","article-title":"Nodding syndrome\u2014South Sudan, 2011","volume":"61","author":"Centers for Disease Control and Prevention (CDC).","year":"2012","journal-title":"MMWR Morb Mortal Wkly Rep"}],"updated-by":[{"DOI":"10.1371\/journal.pntd.0009430","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2021,6,30]],"date-time":"2021-06-30T00:00:00Z","timestamp":1625011200000}}],"container-title":["PLOS Neglected Tropical Diseases"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pntd.0009430","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,6,30]],"date-time":"2021-06-30T17:55:29Z","timestamp":1625075729000},"score":1,"resource":{"primary":{"URL":"https:\/\/dx.plos.org\/10.1371\/journal.pntd.0009430"}},"subtitle":[],"editor":[{"given":"Mar\u00eda-Gloria","family":"Bas\u00e1\u00f1ez","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2021,6,18]]},"references-count":29,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2021,6,18]]}},"URL":"https:\/\/doi.org\/10.1371\/journal.pntd.0009430","relation":{"new_version":[{"id-type":"doi","id":"10.1371\/journal.pntd.0009430","asserted-by":"object"}]},"ISSN":["1935-2735"],"issn-type":[{"value":"1935-2735","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,6,18]]}}}