{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,16]],"date-time":"2026-07-16T15:10:59Z","timestamp":1784214659980,"version":"3.55.0"},"reference-count":20,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,3,6]],"date-time":"2026-03-06T00:00:00Z","timestamp":1772755200000},"content-version":"vor","delay-in-days":64,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100007397","name":"Univerzita Karlova v Praze","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007397","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003042","name":"Else Kr\u00f6ner-Fresenius-Stiftung","doi-asserted-by":"publisher","award":["2022_EKSE.185"],"award-info":[{"award-number":["2022_EKSE.185"]}],"id":[{"id":"10.13039\/501100003042","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002347","name":"Bundesministerium f\u00fcr Bildung und Forschung","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002347","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100005713","name":"Technische Universit\u00e4t M\u00fcnchen","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100005713","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Neurological Medicine"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Mixed movement disorder due to pathogenic variants in the\n                      <jats:italic>ADCY5<\/jats:italic>\n                      gene (MxMD\u2010\n                      <jats:italic>ADCY5<\/jats:italic>\n                      ) is a rare condition characterized predominantly by paroxysmal involuntary choreiform movements involving the limbs, face, and neck, often accompanied by dystonia and myoclonus. Speech disturbances, delayed motor development, cognitive impairment, axial hypotonia, and episodic exacerbations of dyskinesia are also common features. Currently, treatment strategies remain limited; however, several studies indicate a beneficial effect of caffeine in the management of dyskinesia.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>\n                      This clinical report aims to present a case of MxMD\u2010\n                      <jats:italic>ADCY5<\/jats:italic>\n                      showing a high therapeutic response to caffeine. To our knowledge, this is the first reported Ukrainian patient with this condition demonstrating dramatic improvement in paroxysmal dyskinesia after treatment with high\u2010dose caffeine.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      In our clinical observation, administration of caffeine at a total daily dose of 600\u2009mg resulted in significant clinical improvement in an adult patient with MxMD\n                      <jats:italic>-ADCY5<\/jats:italic>\n                      over a follow\u2010up period of at least 6\u2009months. No adverse events or side effects were reported.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1155\/crnm\/4661314","type":"journal-article","created":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T18:40:45Z","timestamp":1772995245000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Mixed Movement Disorder Caused by\n                    <i>ADCY5<\/i>\n                    Pathogenic Variant Successfully Treated With Caffeine: A Case From Ukraine"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0788-2961","authenticated-orcid":false,"given":"Eugenia","family":"Tsoma","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6564-1552","authenticated-orcid":false,"given":"Taras","family":"Studeniak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9732-8947","authenticated-orcid":false,"given":"Robert","family":"Jech","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8112-9153","authenticated-orcid":false,"given":"Michael","family":"Zech","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7731-5129","authenticated-orcid":false,"given":"Petra","family":"Havrankova","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4893-9661","authenticated-orcid":false,"given":"Ev\u017een","family":"R\u016f\u017ei\u010dka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0006-6781-2902","authenticated-orcid":false,"given":"Lukas","family":"Kunc","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3875-7530","authenticated-orcid":false,"given":"Yuriy","family":"Chomolyak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,3,6]]},"reference":[{"key":"e_1_2_11_1_2","doi-asserted-by":"publisher","DOI":"10.1186\/s42466-022-00204-w"},{"key":"e_1_2_11_2_2","doi-asserted-by":"publisher","DOI":"10.1002\/ana.24119"},{"key":"e_1_2_11_3_2","doi-asserted-by":"publisher","DOI":"10.1002\/mdc3.12816"},{"key":"e_1_2_11_4_2","doi-asserted-by":"publisher","DOI":"10.1002\/mds.30170"},{"key":"e_1_2_11_5_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pcbi.1007382"},{"key":"e_1_2_11_6_2","doi-asserted-by":"publisher","DOI":"10.5603\/pjnns.97024"},{"key":"e_1_2_11_7_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000002058"},{"key":"e_1_2_11_8_2","doi-asserted-by":"publisher","DOI":"10.1212\/NXG.0000000000000193"},{"key":"e_1_2_11_9_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2019.02.039"},{"key":"e_1_2_11_10_2","doi-asserted-by":"publisher","DOI":"10.1002\/mdc3.13796"},{"key":"e_1_2_11_11_2","volume-title":"Genereviews","author":"Hisama F. 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