{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,12]],"date-time":"2026-01-12T11:28:33Z","timestamp":1768217313050,"version":"3.49.0"},"reference-count":22,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,1,12]],"date-time":"2026-01-12T00:00:00Z","timestamp":1768176000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>Esophageal leiomyosarcoma (ELMS) is an extremely rare malignant tumor, and the possibility of malignant transformation from benign esophageal leiomyoma has historically been dismissed. This report presents, to our knowledge, the first documented case of malignant transformation within an esophageal leiomyoma and its successful treatment via endoscopic submucosal dissection (ESD), challenging the conventional belief regarding the absolute benignity of esophageal leiomyomas.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case summary<\/jats:title>\n                    <jats:p>A 52-year-old female presented with non-specific symptoms and was found to have a small (0.8 cm) esophageal submucosal nodule during gastroscopy. Endoscopic ultrasound (EUS) showed a well-defined hypoechoic lesion originating from the submucosal layer. The lesion was completely resected via ESD. Histopathological examination demonstrated a conventional leiomyoma with a focal area of malignant transformation to leiomyosarcoma, characterized by high cellularity, nuclear atypia, increased mitotic activity [5 per 2 high-power fields (HPF)], elevated Ki-67 index (40%), and loss of smooth muscle markers (SMA, desmin) in the malignant region. Surgical margins were tumor-free. The patient recovered well with no recurrence at 60-day follow-up.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3389\/fmed.2025.1716368","type":"journal-article","created":{"date-parts":[[2026,1,12]],"date-time":"2026-01-12T08:14:19Z","timestamp":1768205659000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: The leiomyoma-leiomyosarcoma sequence exists in the esophagus: first case of malignant transformation treated with endoscopic submucosal dissection"],"prefix":"10.3389","volume":"12","author":[{"given":"Xu","family":"Wang","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gen","family":"Gui","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ruixue","family":"Liu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zhaoyun","family":"Yang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guifang","family":"Xu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bin","family":"Sun","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2026,1,12]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"761","DOI":"10.1378\/chest.81.6.761","article-title":"Leiomyosarcoma of the esophagus.","volume":"81","author":"Almeida","year":"1982","journal-title":"Chest"},{"key":"B2","doi-asserted-by":"publisher","first-page":"894","DOI":"10.1016\/s0003-4975(98)00684-5","article-title":"Leiomyosarcoma of the esophagus: results of surgical treatment.","volume":"66","author":"Rocco","year":"1998","journal-title":"Ann Thorac Surg."},{"key":"B3","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1097\/00000478-200002000-00007","article-title":"Esophageal stromal tumors: a clinicopathologic, immunohistochemical, and molecular genetic study of 17 cases and comparison with esophageal leiomyomas and leiomyosarcomas.","volume":"24","author":"Miettinen","year":"2000","journal-title":"Am J Surg Pathol"},{"key":"B4","doi-asserted-by":"publisher","first-page":"436","DOI":"10.17659\/01.2013.0100","article-title":"A leiomyosarcoma of esophagus : report of two cases.","volume":"3","author":"Reddy","year":"2013","journal-title":"J Case Rep."},{"key":"B5","doi-asserted-by":"publisher","first-page":"547","DOI":"10.1111\/j.1442-2050.2012.01444.x","article-title":"Esophageal leiomyosarcoma: clinical analysis and surgical treatment of 12 cases.","volume":"27","author":"Zhang","year":"2014","journal-title":"Dis Esophagus"},{"key":"B6","doi-asserted-by":"publisher","first-page":"401","DOI":"10.1007\/s002689910065","article-title":"Tumors of the esophagus.","volume":"24","author":"Hatch","year":"2000","journal-title":"World J Surg"},{"key":"B7","doi-asserted-by":"publisher","first-page":"27","DOI":"10.2214\/ajr.167.1.8659399","article-title":"Leiomyosarcoma of the esophagus: radiographic findings in 10 patients.","volume":"167","author":"Levine","year":"1996","journal-title":"AJR Am J Roentgenol"},{"key":"B8","doi-asserted-by":"publisher","first-page":"e163","DOI":"10.1055\/s-0039-1696729","article-title":"Thoracoscopic esophagectomy for a huge leiomyosarcoma.","volume":"5","author":"Manipadam","year":"2019","journal-title":"Surg J"},{"key":"B9","doi-asserted-by":"publisher","first-page":"49","DOI":"10.4993\/acrt.28.49","article-title":"Giant leiomyosarcoma in the upper third of esophagus, a case report.","volume":"28","author":"Yusupbekov","year":"2020","journal-title":"Ann Cancer Res Ther."},{"key":"B10","doi-asserted-by":"publisher","first-page":"2422","DOI":"10.3892\/ol.2015.3065","article-title":"Radiotherapy treatment of large esophageal leiomyosarcoma: a case report.","volume":"9","author":"Ma","year":"2015","journal-title":"Oncol Lett"},{"key":"B11","doi-asserted-by":"publisher","first-page":"1303","DOI":"10.1038\/modpathol.2009.96","article-title":"Molecular and immunohistochemical evidence for the origin of uterine leiomyosarcomas from associated leiomyoma and symplastic leiomyoma-like areas.","volume":"22","author":"Mittal","year":"2009","journal-title":"Mod Pathol"},{"key":"B12","doi-asserted-by":"publisher","first-page":"136","DOI":"10.1016\/j.jamcollsurg.2003.08.015","article-title":"Current management of esophageal leiomyoma.","volume":"198","author":"Lee","year":"2004","journal-title":"J Am Coll Surg"},{"key":"B13","doi-asserted-by":"publisher","first-page":"300","DOI":"10.1159\/000542778","article-title":"Endoscopic submucosal dissection in a patient with gastric leiomyosarcoma: an alternative to surgery.","volume":"32","author":"Correia Gomes","year":"2025","journal-title":"GE Port J Gastroenterol"},{"key":"B14","doi-asserted-by":"publisher","first-page":"699","DOI":"10.1111\/pin.13165","article-title":"The first case of gastric leiomyosarcoma developed through malignant transformation of leiomyoma.","volume":"71","author":"Yamamoto","year":"2021","journal-title":"Pathol Int"},{"key":"B15","doi-asserted-by":"publisher","first-page":"256","DOI":"10.1186\/s40792-020-00998-4","article-title":"Leiomyosarcoma of the transverse colon with lymph node metastasis and malignant transformation: a case report.","volume":"6","author":"Tago","year":"2020","journal-title":"Surg Case Rep"},{"key":"B16","doi-asserted-by":"publisher","first-page":"362","DOI":"10.1097\/eus.0000000000000001","article-title":"Identification of sentinel lymph nodes in esophageal cancer patients using contrast-enhanced EUS with peritumoral injections.","volume":"12","author":"Liu","year":"2023","journal-title":"Endosc Ultrasound"},{"key":"B17","doi-asserted-by":"publisher","first-page":"1089","DOI":"10.7863\/ultra.16.01032","article-title":"Ultrasound strain elastography for breast lesions: computer-aided evaluation with quantifiable elastographic features.","volume":"36","author":"Xiao","year":"2017","journal-title":"J Ultrasound Med"},{"key":"B18","doi-asserted-by":"publisher","first-page":"156","DOI":"10.4103\/2303-9027.201086","article-title":"Role of endoscopic ultrasound and endoscopic-ultrasound-guided fine-needle aspiration in endoscopic biopsy negative gastrointestinal lesions.","volume":"6","author":"Okasha","year":"2017","journal-title":"Endosc Ultrasound"},{"key":"B19","doi-asserted-by":"publisher","first-page":"2479","DOI":"10.2169\/internalmedicine.2219-18","article-title":"Esophageal leiomyosarcoma diagnosed by endoscopic ultrasound-guided fine-needle aspiration biopsy and cured with surgical resection.","volume":"58","author":"Ebi","year":"2019","journal-title":"Intern Med"},{"key":"B20","doi-asserted-by":"publisher","first-page":"700","DOI":"10.1111\/den.12437","article-title":"Polypoid leiomyosarcoma of the esophagus treated by endoscopic submucosal dissection.","volume":"27","author":"Yamamoto","year":"2015","journal-title":"Dig Endosc"},{"key":"B21","doi-asserted-by":"publisher","first-page":"5385","DOI":"10.3748\/wjg.v19.i32.5385","article-title":"A case of rapidly progressing leiomyosarcoma combined with squamous cell carcinoma in the esophagus.","volume":"19","author":"Jang","year":"2013","journal-title":"World J Gastroenterol"},{"key":"B22","doi-asserted-by":"publisher","first-page":"194","DOI":"10.1002\/jso.2930480311","article-title":"Esophageal sarcomas.","volume":"48","author":"Perch","year":"1991","journal-title":"J Surg Oncol"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1716368\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,12]],"date-time":"2026-01-12T08:14:44Z","timestamp":1768205684000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1716368\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,12]]},"references-count":22,"alternative-id":["10.3389\/fmed.2025.1716368"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2025.1716368","relation":{},"ISSN":["2296-858X"],"issn-type":[{"value":"2296-858X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,12]]},"article-number":"1716368"}}