biostudies-literatureLuf MDepartment of Defense Melanoma Research ProgramTisch Cancer Institute Cancer CenterNational Institutes of Health, National Institute of General Medical SciencesNational Cancer InstituteNCI NIH HHSjkaf227https://www.ebi.ac.uk/biostudies/studies/S-EPMC12693516biostudies-literatureUnknown15(12)The deubiquitinating enzyme BAP1 is the catalytic subunit of the Polycomb Repressive Deubiquitinase (PR-DUB) complex, which acts with the Polycomb Repressive Complexes 1 and 2 to regulate chromatin organization to repress homeotic genes and other developmental regulators. Loss of BAP1 is implicated in several cancers, in the familial cancer syndrome BAP1 Tumor Predisposition Syndrome, and in the neurodevelopmental disorder Küry-Isidor syndrome. In Drosophila, there are numerous reports in the literature describing developmental patterning phenotypes for several chromatin regulators, including the discovery of Polycomb itself, but corresponding adult morphological phenotypes due to developmental dysregulation of the Drosophila BAP1 ortholog calypso (caly) are less well-described. We report G3 (Bethesda, Md.)Knockdown of PR-DUB subunit calypso in the developing Drosophila eye and wing results in mis-patterned tissues with altered size and shape.PMC12693516R01GM135330P30 CA196521R01GM122995R01CA161870ME240338Bowcock AMPfleger CMBegani PLuf MfalseKnockdown of PR-DUB subunit calypso in the developing Drosophila eye and wing results in mis-patterned tissues with altered size and shape.The deubiquitinating enzyme BAP1 is the catalytic subunit of the Polycomb Repressive Deubiquitinase (PR-DUB) complex, which acts with the Polycomb Repressive Complexes 1 and 2 to regulate chromatin organization to repress homeotic genes and other developmental regulators. Loss of BAP1 is implicated in several cancers, in the familial cancer syndrome BAP1 Tumor Predisposition Syndrome, and in the neurodevelopmental disorder Küry-Isidor syndrome. In Drosophila, there are numerous reports in the literature describing developmental patterning phenotypes for several chromatin regulators, including the discovery of Polycomb itself, but corresponding adult morphological phenotypes due to developmental dysregulation of the Drosophila BAP1 ortholog calypso (caly) are less well-described. We report 2025-01-01T00:00:00Z2025 Dec2026-05-26T14:00:31.949Z2026-05-24T03:11:43.581ZS-EPMC126935164102258410.1093/g3journal/jkaf227