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. 2022 Jul 24;9(8):1276–1288. doi: 10.1002/acn3.51629

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© 2022 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association.

This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc-nd/4.0/ License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.

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Functional modeling of ANKLE2 variants recapitulates microcephaly phenotypes in Drosophila melanogaster. (A–F) Bright field images of third instar larval brains from (A) Ankle2 ^A , da‐hANKLE2 wt, (B) Ankle2 ^A, (C) Ankle2 ^A , da‐hANKLE2 p.Val229Gly, (D) Ankle2 ^A , da‐hANKLE2 p.Arg236*, (E) Ankle2 ^A , da‐hANKLE2 p.Arg536Cys, (F) Ankle2 ^A , da‐hANKLE2 c.1421‐1G>C. (G) Brain volume quantification of animals from (A–F). One‐way ANOVA with post hoc test. *p < 0.05, **p < 0.01, ****p < 0.0001.