biostudies-literatureUnknown14Storck A•<i>VPS11</i> is associated with hypomyelinated leukodystrophy affecting children.•A single case of adult-onset generalized dystonia was reported.•We provide the first replication by describing a second case of complex dystonia related to biallelic variants in <i>VPS11</i>.Clinical parkinsonism & related disorders100419https://www.ebi.ac.uk/biostudies/studies/S-EPMC12808586biostudies-literatureConfirmation of biallelic &lt;i&gt;VPS11&lt;/i&gt; variants as a cause of complex dystonic syndrome.PMC12808586Antal MCAbiwarde MTLebre ASHardy GAnheim MScheidecker SStorck AWirth TfalseConfirmation of biallelic &lt;i&gt;VPS11&lt;/i&gt; variants as a cause of complex dystonic syndrome.•<i>VPS11</i> is associated with hypomyelinated leukodystrophy affecting children.•A single case of adult-onset generalized dystonia was reported.•We provide the first replication by describing a second case of complex dystonia related to biallelic variants in <i>VPS11</i>.2026-01-01T00:00:00Z20262026-06-06T14:45:04.689Z2026-06-01T03:07:15.689ZS-EPMC128085864155106910.1016/j.prdoa.2025.100419