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AIS affects \u223c3% of children worldwide and is the most common musculoskeletal diagnosis in pediatric populations, posing a significant physiological, psychosocial and financial burden to patients. Genetic predisposition is a clear and major contributor to AIS, and insights from genomic discoveries are inspiring translational studies ultimately aimed at developing novel diagnostics and therapies. Pre-clinical animal models of AIS are now essential to validate human genetic findings, understand gene-by-environment interactions, and speed etiologic and therapeutic discovery. In this Perspective, we highlight the current status of pre-clinical models of AIS and discuss the challenges posed by the nature of the disorder combined with the limitations of standard approaches. Current research suggests that straightforward genetic targeting of orthologous AIS disease genes in vertebrates may not necessarily yield equivalent physiological phenotypes but nevertheless can be utilized to understand disease mechanisms. Longer-term, appropriately complex models are needed to fully recapitulate the human AIS phenotype arising from genetic, physiological and mechanical interactions.<\/jats:p>","DOI":"10.1242\/dmm.052438","type":"journal-article","created":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T07:45:53Z","timestamp":1756712753000},"source":"Crossref","is-referenced-by-count":0,"title":["The importance of imperfect pre-clinical models in adolescent idiopathic scoliosis"],"prefix":"10.1242","volume":"18","author":[{"given":"Diane S.","family":"Sepich","sequence":"first","affiliation":[{"name":"Washington University School of Medicine 1 Department of Developmental Biology , , St. Louis, MO 63110 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryan S.","family":"Gray","sequence":"additional","affiliation":[{"name":"Dell Pediatric Research Institute, University of Texas at Austin Dell Medical School 2 Department of Pediatrics , , Austin, TX 78723 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nadav","family":"Ahituv","sequence":"additional","affiliation":[{"name":"University of California San Francisco 3 Department of Bioengineering and Therapeutic Sciences , , San Francisco, CA 94158 ,","place":["USA"]},{"name":"Institute for Human Genetics, University of California San Francisco 4 , San Francisco, CA 94158 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christina A.","family":"Gurnett","sequence":"additional","affiliation":[{"name":"Washington University School of Medicine 5 Department of Neurology , , St. Louis, MO 63110 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jonathan J.","family":"Rios","sequence":"additional","affiliation":[{"name":"University of Texas Southwestern Medical Center 6 Department of Orthopaedic Surgery , , Dallas, TX 75235 ,","place":["USA"]},{"name":"McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center 7 , Dallas, TX 75235 ,","place":["USA"]},{"name":"University of Texas Southwestern Medical Center 8 Department of Pediatrics , , Dallas, TX 75235 ,","place":["USA"]},{"name":"Center for Translational Research, Scottish Rite for Children 9 , 2222 Welborn St., Dallas, TX 75219 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lila","family":"Solnica-Krezel","sequence":"additional","affiliation":[{"name":"Washington University School of Medicine 1 Department of Developmental Biology , , St. Louis, MO 63110 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6790-2194","authenticated-orcid":false,"given":"Carol A.","family":"Wise","sequence":"additional","affiliation":[{"name":"University of Texas Southwestern Medical Center 6 Department of Orthopaedic Surgery , , Dallas, TX 75235 ,","place":["USA"]},{"name":"McDermott Center for Human Growth and Development, University of Texas Southwestern Medical Center 7 , Dallas, TX 75235 ,","place":["USA"]},{"name":"University of Texas Southwestern Medical Center 8 Department of Pediatrics , , Dallas, TX 75235 ,","place":["USA"]},{"name":"Center for Translational Research, Scottish Rite for Children 9 , 2222 Welborn St., Dallas, TX 75219 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2025,9,1]]},"reference":[{"key":"2025092508161092900_DMM052438C1","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1159\/000085665","article-title":"The scoliosis (sco) mouse: a new allele of Pax1","volume":"111","author":"Adham","year":"2005","journal-title":"Cytogenet Genome Res."},{"key":"2025092508161092900_DMM052438C2","doi-asserted-by":"publisher","first-page":"2","DOI":"10.1186\/1748-7161-1-2","article-title":"Adolescent idiopathic scoliosis: natural history and long term treatment effects","volume":"1","author":"Asher","year":"2006","journal-title":"Scoliosis"},{"key":"2025092508161092900_DMM052438C3","doi-asserted-by":"publisher","first-page":"3168","DOI":"10.1038\/s41467-020-16971-6","article-title":"Piezo2 expressed in proprioceptive neurons is essential for skeletal integrity","volume":"11","author":"Assaraf","year":"2020","journal-title":"Nat. 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