{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,30]],"date-time":"2025-12-30T03:45:52Z","timestamp":1767066352538,"version":"3.37.3"},"reference-count":40,"publisher":"Portland Press Ltd.","issue":"12","license":[{"start":{"date-parts":[[2022,6,29]],"date-time":"2022-06-29T00:00:00Z","timestamp":1656460800000},"content-version":"vor","delay-in-days":5,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["portlandpress.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2022,6,30]]},"abstract":"<jats:p>In cochlea, deafness-related protein PDZD7 is an indispensable component of the ankle link complex, which is critical for the maturation of inner-ear hair cell for sound perception. Ankle links, connecting the different rows of cochlear stereocilia, are essential for the staircase-like development of stereocilia. However, the molecular mechanism of how PDZD7 governs stereociliary development remains unknown. Here, we reported a novel PDZD7-binding partner, FCHSD2, identified by yeast two-hybrid screening. FCHSD2 was reported to be expressed in hair cell, where it co-operated with CDC42 and N-WASP to regulate the formation of cell protrusion. The association between FCHSD2 and PDZD7 was further confirmed in COS-7 cells. More importantly, we solved the complex structure of FCHSD2 tail with PDZD7 PDZ3 domain at 2.0\u2005\u00c5 resolution. The crystal structure shows that PDZD7 PDZ3 adopts a typical PDZ domain topology, comprising five \u03b2 strands and two \u03b1 helixes. The PDZ-binding motif of FCHSD2 tail stretches through the \u03b1B\/\u03b2B groove of PDZD7 PDZ3. Our study not only uncovers the interaction between FCHSD2 tail and PDZD7 PDZ3 at the atomic level, but also provides clues of connecting the ankle link complex with cytoskeleton dynamics for exploiting the molecular mechanism of stereociliary development.<\/jats:p>","DOI":"10.1042\/bcj20220147","type":"journal-article","created":{"date-parts":[[2022,6,13]],"date-time":"2022-06-13T11:02:56Z","timestamp":1655118176000},"page":"1393-1405","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Deafness-related protein PDZD7 forms complex with the C-terminal tail of FCHSD2"],"prefix":"10.1042","volume":"479","author":[{"given":"Huang","family":"Wang","sequence":"first","affiliation":[{"name":"1Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Ministry of Education, Bio-X Institutes, Shanghai Jiao Tong University, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dange","family":"Zhao","sequence":"additional","affiliation":[{"name":"1Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Ministry of Education, Bio-X Institutes, Shanghai Jiao Tong University, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Haibo","family":"Du","sequence":"additional","affiliation":[{"name":"2Shandong Provincial Key Laboratory of Animal Cell and Developmental Biology, School of Life Sciences, Shandong University, Qingdao, Shandong, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xiaoyan","family":"Zhai","sequence":"additional","affiliation":[{"name":"2Shandong Provincial Key Laboratory of Animal Cell and Developmental Biology, School of Life Sciences, Shandong University, Qingdao, Shandong, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shaoxuan","family":"Wu","sequence":"additional","affiliation":[{"name":"1Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Ministry of Education, Bio-X Institutes, Shanghai Jiao Tong University, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lin","family":"Lin","sequence":"additional","affiliation":[{"name":"1Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Ministry of Education, Bio-X Institutes, Shanghai Jiao Tong University, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zhigang","family":"Xu","sequence":"additional","affiliation":[{"name":"2Shandong Provincial Key Laboratory of Animal Cell and Developmental Biology, School of Life Sciences, Shandong University, Qingdao, Shandong, China"},{"name":"3Shandong Provincial Collaborative Innovation Center of Cell Biology, Shandong Normal University, Jinan, Shandong, China"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6956-3333","authenticated-orcid":false,"given":"Qing","family":"Lu","sequence":"additional","affiliation":[{"name":"1Key Laboratory for the Genetics of Developmental and Neuropsychiatric Disorders, Ministry of Education, Bio-X Institutes, Shanghai Jiao Tong University, Shanghai, China"},{"name":"4Department of Otolaryngology-Head and Neck Surgery, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China"},{"name":"5Ear Institute, Shanghai Jiao Tong University School of Medicine, Shanghai, China"},{"name":"6Shanghai Key Laboratory of Translational Medicine on Ear and Nose Diseases, Shanghai, China"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"288","published-online":{"date-parts":[[2022,6,24]]},"reference":[{"key":"2022062907233673400_BCJ-2022-0147C1","doi-asserted-by":"publisher","first-page":"9","DOI":"10.1083\/jcb.201001138","article-title":"Review series: the cell biology of hearing","volume":"190","year":"2010","journal-title":"J. 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