{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T19:02:19Z","timestamp":1790622139040,"version":"4.1.0"},"reference-count":123,"publisher":"Oxford University Press (OUP)","issue":"20","license":[{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"DOI":"10.13039\/100006691","name":"Harvard Medical School","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006691","id-type":"DOI","asserted-by":"publisher"}]},{"name":"McKenzie Family Charitable Trust"},{"DOI":"10.13039\/501100004199","name":"Okinawa Institute of Science and Technology Graduate University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004199","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,9,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Spinal Muscular Atrophy (SMA) is a neuromuscular disorder associated with motor neuron degeneration, yet the role of the well-conserved Survival of Motor Neuron (SMN) protein in muscle remains insufficiently understood. Using the Drosophila neuromuscular junction (NMJ) as a model, we combined tissue-specific Smn depletion with transcriptomic profiling and prior genetic modifier screens to identify SMN-dependent genes that contribute to synaptic architecture. Functional screening of candidate genes significantly and selectively effected in muscle identified genes whose knockdown phenocopied NMJ morphometry defects characteristic of Smn loss-of-function (LOF), including FER, CAP, HDAC4, Nhe2, and Raskol. Unlike other genes in this group, analysis of postsynaptic architecture using a subsynaptic reticulum (SSR) biomarker disrupted by Smn LOF singled out the conserved tyrosine kinase FER as an SMN-dependent gene in muscle that influences this prominent feature of the NMJ. Analysis of different FER transcripts suggests that elevation of the FERp100 protein isoform in Smn LOF muscle tissue leads to a dominant-negative effect likely to disrupt signaling via the MAPK-family kinase Basket (Bsk) to regulate the downstream phosphatase Puckered (Puc).<\/jats:p>","DOI":"10.1093\/hmg\/ddag096","type":"journal-article","created":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T18:22:57Z","timestamp":1789755777000},"source":"Crossref","is-referenced-by-count":0,"title":["Tissue-specific models of spinal muscular atrophy identify muscle functions required for\n                    <i>Drosophila<\/i>\n                    neuromuscular junction architecture"],"prefix":"10.1093","volume":"35","author":[{"given":"Takakazu","family":"Yokokura","sequence":"first","affiliation":[{"name":"Department of Cell Biology, Harvard Medical School , 240 Longwood Avenue, 240 Longwood Avenue, Boston, MA 02115 ,","place":["United States"]},{"name":"Okinawa Institute of Science and Technology Graduate University , 1919-1 Tancha, Onna, Kunigami, Okinawa 9040954 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hansine","family":"Heggeness","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Harvard Medical School , 240 Longwood Avenue, 240 Longwood Avenue, Boston, MA 02115 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seiko","family":"Yoshikawa","sequence":"additional","affiliation":[{"name":"Okinawa Institute of Science and Technology Graduate University , 1919-1 Tancha, Onna, Kunigami, Okinawa 9040954 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Salvatore","family":"Alaimo","sequence":"additional","affiliation":[{"name":"Department of Clinical and Experimental Medicine, University of Catania , Piazza Universit\u00e0, 2, 95124 Catania CT ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thirada","family":"Boonrawd","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Harvard Medical School , 240 Longwood Avenue, 240 Longwood Avenue, Boston, MA 02115 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Navpreet","family":"Ahluwalia","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Harvard Medical School , 240 Longwood Avenue, 240 Longwood Avenue, Boston, MA 02115 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thi Thu","family":"Van Dihn","sequence":"additional","affiliation":[{"name":"Okinawa Institute of Science and Technology Graduate University , 1919-1 Tancha, Onna, Kunigami, Okinawa 9040954 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alfredo","family":"Ferro","sequence":"additional","affiliation":[{"name":"Department of Clinical and Experimental Medicine, University of Catania , Piazza Universit\u00e0, 2, 95124 Catania CT ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elizabeth M","family":"McNeill","sequence":"additional","affiliation":[{"name":"Department of Food Science and Human Nutrition, Iowa State University , 515 Morrill Road, Ames, IA 50011 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8156-9482","authenticated-orcid":false,"given":"David","family":"Van Vactor","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Harvard Medical School , 240 Longwood Avenue, 240 Longwood Avenue, Boston, MA 02115 ,","place":["United 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