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A 15\u2010year\u2010old male presented with progressive right maxillary swelling initially treated as a dental abscess. Radiographic examination revealed a poorly defined destructive lesion associated with the apices of teeth #14\u201318, exhibiting expansion and destruction of the maxillary sinus walls. Additional imaging demonstrated a contrast\u2010enhancing soft tissue mass with extensive local invasion. Histopathological examination confirmed alveolar rhabdomyosarcoma that was positive for myogenin, myoD1, CD56 and desmin, with a Ki\u201067 proliferation index of 80%\u201390%. This case highlights that approximately 3% of non\u2010endodontic periapical lesions are malignant, reinforcing the importance of thorough clinical, radiographic and histopathological examinations for lesions demonstrating atypical features. Dentists should maintain vigilance when evaluating periapical lesions, particularly those with atypical presentations or unresponsive to conventional therapy.<\/jats:p>","DOI":"10.1111\/aej.70009","type":"journal-article","created":{"date-parts":[[2025,8,11]],"date-time":"2025-08-11T11:51:07Z","timestamp":1754913067000},"page":"236-241","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report of Maxillary Alveolar Rhabdomyosarcoma Misdiagnosed as Periapical Lesion: Clinicopathological Alerts and Life\u2010Saving Lessons for Dentists"],"prefix":"10.1111","volume":"52","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2723-6502","authenticated-orcid":false,"given":"Saliha","family":"Ak\u00e7ay K\u00f6pr\u00fcc\u00fc","sequence":"first","affiliation":[{"name":"Department of Oral, Dental and Maxillofacial Radiology, Faculty of Dentistry Karadeniz Technical University  Trabzon Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Erdal Rahman","family":"K\u00f6pr\u00fcc\u00fc","sequence":"additional","affiliation":[{"name":"Department of Otorhinolaryngology, Faculty of Medicine Karadeniz Technical University  Trabzon Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aysun","family":"Atasoy S\u0131nd\u0131ra\u00e7","sequence":"additional","affiliation":[{"name":"Oral and Dental Health Hospital  Trabzon Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"\u00dcmit","family":"\u00c7obano\u011flu","sequence":"additional","affiliation":[{"name":"Department of Pathology, Faculty of Medicine Karadeniz Technical University  Trabzon Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2025,8,11]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.adaj.2016.02.018"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1111\/aej.12597"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.joen.2022.07.003"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.joen.2020.01.003"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.4103\/0971-9261.24632"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijporl.2015.06.032"},{"key":"e_1_2_9_8_1","first-page":"30","article-title":"Rhabdomyosarcoma: Updates on Classification and the Necessity of Molecular Testing Beyond Immunohistochemistry","volume":"133","author":"Dehner C. 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