{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,12]],"date-time":"2026-03-12T04:50:43Z","timestamp":1773291043653,"version":"3.50.1"},"reference-count":50,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2009,5,1]],"date-time":"2009-05-01T00:00:00Z","timestamp":1241136000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Spanish Ministry of Health"},{"name":"Consumption in cooperation"},{"name":"Fundaci\u00f3n Canaria de Investigaci\u00f3n y Salud"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2009,6,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The aim of this study was to determine the effectiveness and safety of available treatment alternatives for degenerative ataxias (DA). We systematically reviewed studies that assess pharmacological, rehabilitative, or psychological treatments in patients with DA. Studies were included if they fulfilled prespecified criteria. All included clinical trials were scored for methodological quality. Main outcome measures were clinical status of neurological disorder, adverse events, and patient\u2010based factors. Twenty\u2010five studies were included. Most studies were of small sample sizes, wide age variations, and low scientific validity. Only one study gave information on physical rehabilitation and none on psychological therapy. The remaining 24 studies reported on the effects of different pharmacological treatments. Outcomes such as functional capacity and psychological functioning of patients were evaluated by few studies. Some evidence supports that 5\u2010hydroxytryptophan is more effective than placebo improving neurological symptoms in patients with Friedreich ataxia (FA), olivopontocerebellar atrophy, or cerebellar atrophy. Idebenone is more effective than placebo for halting and reversing the hypertrophic cardiomyopathy associated with FA, but it seems unable to improve neurological semiology. Limited evidence for other therapies was found. No relevant side effects for drugs that shown some degree of effectiveness were reported. Availability of quality studies to evaluate the safety and efficacy of treatments for most DA is scarce. No valid information on the actual value of physical rehabilitation and psychological support as treatments for DA is available. Further investigations with improved trial designs are necessary. \u00a9 2009 Movement Disorder Society<\/jats:p>","DOI":"10.1002\/mds.22564","type":"journal-article","created":{"date-parts":[[2009,5,1]],"date-time":"2009-05-01T15:58:51Z","timestamp":1241193531000},"page":"1111-1124","source":"Crossref","is-referenced-by-count":59,"title":["Effectiveness and safety of treatments for degenerative ataxias: A systematic review"],"prefix":"10.1002","volume":"24","author":[{"given":"M.Mar","family":"Trujillo\u2010Mart\u00edn","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pedro","family":"Serrano\u2010Aguilar","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fernando","family":"Monton\u2010\u00c1lvarez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Romen","family":"Carrillo\u2010Fumero","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2009,6,18]]},"reference":[{"key":"e_1_2_7_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(83)92879-9"},{"key":"e_1_2_7_3_2","doi-asserted-by":"publisher","DOI":"10.1097\/00019052-200008000-00014"},{"key":"e_1_2_7_4_2","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/114.2.855"},{"key":"e_1_2_7_5_2","doi-asserted-by":"publisher","DOI":"10.1002\/mds.21798"},{"key":"e_1_2_7_6_2","unstructured":"SerranoP PeresteloL AlonsoJ.MahtaniV.Generic and specific questionnaires of health\u2010related quality of life in adults and their applicability in the area of the rare diseases. Project financed by the Carlos III Health Institute (Madrid Spain): FIS\u2010ETS 03\/10106.2005."},{"key":"e_1_2_7_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/0197-2456(95)00134-4"},{"key":"e_1_2_7_8_2","first-page":"119","article-title":"R\u00e9gression del'ataxie c\u00e9rebelleuse humaine sous administration a long terme de 5\u2010hydroxytryptophane","volume":"292","author":"Trouillas P","year":"1981","journal-title":"C R Acad Sci Paris"},{"key":"e_1_2_7_9_2","first-page":"415","article-title":"R\u00e9gression du syndrome c\u00e9r\u00e9belleux sous administration a long terme de 5\u2010HTP ou de l'association 5\u2010HTP\u2010bens\u00e9razide","volume":"138","author":"Trouillas P","year":"1982","journal-title":"Rev Neurol"},{"key":"e_1_2_7_10_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.1988.00520350055016"},{"key":"e_1_2_7_11_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.1995.00540290042016"},{"key":"e_1_2_7_12_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.1995.00540290037015"},{"key":"e_1_2_7_13_2","doi-asserted-by":"publisher","DOI":"10.1055\/s-2002-34494"},{"key":"e_1_2_7_14_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000055872.50364.FC"},{"key":"e_1_2_7_15_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000068549.52812.0F"},{"key":"e_1_2_7_16_2","doi-asserted-by":"publisher","DOI":"10.1136\/heart.87.4.346"},{"key":"e_1_2_7_17_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(99)01341-0"},{"key":"e_1_2_7_18_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.27.1.70"},{"key":"e_1_2_7_19_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.31.3.288"},{"key":"e_1_2_7_20_2","doi-asserted-by":"publisher","DOI":"10.1097\/00002826-200003000-00010"},{"key":"e_1_2_7_21_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00702-004-0216-x"},{"key":"e_1_2_7_22_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.58.9.1451"},{"key":"e_1_2_7_23_2","doi-asserted-by":"publisher","DOI":"10.1017\/S0317167100036672"},{"key":"e_1_2_7_24_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.38.4.649"},{"key":"e_1_2_7_25_2","doi-asserted-by":"publisher","DOI":"10.1034\/j.1600-0404.2001.00299.x"},{"key":"e_1_2_7_26_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.38.9.1478"},{"key":"e_1_2_7_27_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.39.5.749-b"},{"key":"e_1_2_7_28_2","doi-asserted-by":"publisher","DOI":"10.1017\/S0317167100031863"},{"key":"e_1_2_7_29_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000125691.74109.53"},{"key":"e_1_2_7_30_2","doi-asserted-by":"crossref","first-page":"301","DOI":"10.33588\/rn.3504.2001006","article-title":"Clorhidrato de buspirona en el tratamiento de la ataxia cerebelosa","volume":"35","author":"Andrade\u2010Filho AS","year":"2002","journal-title":"Rev Neurol"},{"key":"e_1_2_7_31_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.62.4.621"},{"key":"e_1_2_7_32_2","doi-asserted-by":"crossref","first-page":"907","DOI":"10.33588\/rn.3910.2004331","article-title":"Efectos de un programa de ejercicios f\u00edsicos sobre variables neurol\u00f3gicas cuantitativas en pacientes con ataxia espinocerebelosa tipo 2 en estadio leve","volume":"39","author":"P\u00e9rez\u2010Avila I","year":"2004","journal-title":"Rev Neurol"},{"key":"e_1_2_7_33_2","doi-asserted-by":"publisher","DOI":"10.1097\/00005650-199206000-00002"},{"key":"e_1_2_7_34_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.2044-8341.1959.tb00467.x"},{"key":"e_1_2_7_35_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.28.12.1259"},{"key":"e_1_2_7_36_2","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(07)70220-X"},{"key":"e_1_2_7_37_2","doi-asserted-by":"publisher","DOI":"10.1001\/archneur.64.4.558"},{"key":"e_1_2_7_38_2","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.63.5.633"},{"key":"e_1_2_7_39_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.43.5.931"},{"key":"e_1_2_7_40_2","first-page":"666","article-title":"Risk factors for developing eosinophilia myalgia syndrome among L\u2010tryptophan users in New York","volume":"20","author":"Back EE","year":"1993","journal-title":"J Rheumatol"},{"key":"e_1_2_7_41_2","doi-asserted-by":"publisher","DOI":"10.1111\/1523-1747.ep12356368"},{"key":"e_1_2_7_42_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199008093230601"},{"key":"e_1_2_7_43_2","doi-asserted-by":"publisher","DOI":"10.1001\/jama.1992.03480010085029"},{"key":"e_1_2_7_44_2","first-page":"60","article-title":"Bias or biology: evaluating the epidemiologic studies of L\u2010tryptophan and the eosinophilia\u2010myalgia syndrome","volume":"46","author":"Horwitz RI","year":"1996","journal-title":"J Rheumatol Suppl"},{"key":"e_1_2_7_45_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000156802.15466.79"},{"key":"e_1_2_7_46_2","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2006.096008"},{"key":"e_1_2_7_47_2","doi-asserted-by":"publisher","DOI":"10.1080\/08870449608400270"},{"key":"e_1_2_7_48_2","doi-asserted-by":"publisher","DOI":"10.1097\/00012272-198510000-00005"},{"key":"e_1_2_7_49_2","doi-asserted-by":"publisher","DOI":"10.1002\/bs.3830190102"},{"key":"e_1_2_7_50_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000219042.60538.92"},{"key":"e_1_2_7_51_2","doi-asserted-by":"publisher","DOI":"10.3310\/hta8150"}],"container-title":["Movement Disorders"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fmds.22564","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fmds.22564","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/movementdisorders.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/mds.22564","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,20]],"date-time":"2025-10-20T14:23:19Z","timestamp":1760970199000},"score":1,"resource":{"primary":{"URL":"https:\/\/movementdisorders.onlinelibrary.wiley.com\/doi\/10.1002\/mds.22564"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2009,6,15]]},"references-count":50,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2009,6,15]]}},"alternative-id":["10.1002\/mds.22564"],"URL":"https:\/\/doi.org\/10.1002\/mds.22564","archive":["Portico"],"relation":{},"ISSN":["0885-3185","1531-8257"],"issn-type":[{"value":"0885-3185","type":"print"},{"value":"1531-8257","type":"electronic"}],"subject":[],"published":{"date-parts":[[2009,6,15]]}}}