{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T16:26:50Z","timestamp":1790872010731,"version":"4.1.0"},"reference-count":28,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2009,8,11]],"date-time":"2009-08-11T00:00:00Z","timestamp":1249948800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Pediatric Blood &amp; Cancer"],"published-print":{"date-parts":[[2009,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Kinsbourne syndrome or opsoclonus myoclonus syndrome (OMS) is characterized by rapid, involuntary, irregular conjugate eye movements (opsoclonus), myoclonic jerking of the limbs and trunk, ataxia, and behavioral disturbances. In general, the outcome of neurologic and behavioral symptoms is poor. Studies on quality of life (Qol) and recovery after very long\u2010term follow\u2010up of children with OMS are lacking.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We studied long\u2010term cancer survival, neurologic recovery, and Qol of a consecutive series of eight patients with OMS that were treated in our center. Two cross\u2010sectional follow\u2010up evaluations were conducted at a 3\u2010year interval (T1 and T2).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>In four out of eight children with OMS a neuroblastoma (NBL) was diagnosed and surgically removed completely. All children received immuno\u2010modulatory therapy, consisting of prednisone (three children), prednisone and ACTH (four children), or prednisone, ACTH, and gammaglobulin (one child). At T1, median follow\u2010up time was 9.5 years (range 3\u201314 years) after OMS onset and at T2, 11.6 years (range 6\u201317 years). Neurologic functioning improved in all children, reflected by a significant improvement of the mean <jats:italic>Z<\/jats:italic>\u2010score in the motor domain of the Qol questionnaire at T2 as compared to T1. In contrast, seven children continued to have a severe developmental delay at T2. No significant difference in any of the variables was found between NBL survivors and OMS patients without NBL.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Cognitive and behavioral impairments and not a motor impairment, such as ataxia, appear to be predominant at long\u2010term in children with OMS. Pediatr Blood Cancer 2009;53:1048\u20131053. \u00a9 2009 Wiley\u2010Liss, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/pbc.22226","type":"journal-article","created":{"date-parts":[[2009,8,11]],"date-time":"2009-08-11T17:52:36Z","timestamp":1250013156000},"page":"1048-1053","source":"Crossref","is-referenced-by-count":32,"title":["Improvement of neurological status and quality of life in children with opsoclonus myoclonus syndrome at long\u2010term follow\u2010up"],"prefix":"10.1002","volume":"53","author":[{"given":"Coriene E.","family":"Catsman\u2010Berrevoets","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Femke K.","family":"Aarsen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marloes L.C.","family":"van Hemsbergen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Max M.","family":"van Noesel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederique G.A.J.","family":"Hakvoort\u2010Cammel","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marry M.","family":"van den Heuvel\u2010Eibrink","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2009,8,11]]},"reference":[{"key":"e_1_2_6_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.canlet.2005.01.051"},{"key":"e_1_2_6_3_2","doi-asserted-by":"publisher","DOI":"10.1097\/00002826-199206000-00002"},{"key":"e_1_2_6_4_2","doi-asserted-by":"publisher","DOI":"10.1002\/ana.20539"},{"key":"e_1_2_6_5_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM196808292790907"},{"key":"e_1_2_6_6_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(97)70272-5"},{"key":"e_1_2_6_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneuroim.2005.08.018"},{"key":"e_1_2_6_8_2","doi-asserted-by":"publisher","DOI":"10.1067\/mpd.2001.118200"},{"key":"e_1_2_6_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(06)80167-8"},{"key":"e_1_2_6_10_2","doi-asserted-by":"publisher","DOI":"10.1002\/mpo.1138"},{"key":"e_1_2_6_11_2","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1096-911X(199704)28:4<284::AID-MPO7>3.0.CO;2-E"},{"key":"e_1_2_6_12_2","first-page":"895","volume-title":"Principles and practice of pediatric oncology","author":"Brodeur G","year":"2002"},{"key":"e_1_2_6_13_2","doi-asserted-by":"publisher","DOI":"10.1016\/0887-8994(95)00083-R"},{"key":"e_1_2_6_14_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF01953945"},{"key":"e_1_2_6_15_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0929-693X(00)80129-3"},{"key":"e_1_2_6_16_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1995.tb11944.x"},{"key":"e_1_2_6_17_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-006-0247-4"},{"key":"e_1_2_6_18_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0887-8994(01)00245-4"},{"key":"e_1_2_6_19_2","doi-asserted-by":"publisher","DOI":"10.1093\/eurpub\/9.3.188"},{"key":"e_1_2_6_20_2","doi-asserted-by":"publisher","DOI":"10.1023\/A:1008848218806"},{"key":"e_1_2_6_21_2","doi-asserted-by":"publisher","DOI":"10.1044\/jshr.1202.246"},{"key":"e_1_2_6_22_2","doi-asserted-by":"publisher","DOI":"10.1002\/1097-0142(19880815)62:4<723::AID-CNCR2820620413>3.0.CO;2-U"},{"key":"e_1_2_6_23_2","doi-asserted-by":"publisher","DOI":"10.1177\/088307389000500217"},{"key":"e_1_2_6_24_2","doi-asserted-by":"publisher","DOI":"10.1177\/1043454204272560"},{"key":"e_1_2_6_25_2","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/121.4.561"},{"key":"e_1_2_6_26_2","doi-asserted-by":"publisher","DOI":"10.1176\/jnp.16.3.367"},{"key":"e_1_2_6_27_2","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000120549.77188.36"},{"key":"e_1_2_6_28_2","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awg195"},{"key":"e_1_2_6_29_2","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/123.5.1051"}],"container-title":["Pediatric Blood &amp; 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