{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,23]],"date-time":"2026-08-23T18:18:33Z","timestamp":1787509113731,"version":"build-2736575974"},"reference-count":24,"publisher":"MDPI AG","issue":"13","license":[{"start":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T00:00:00Z","timestamp":1783036800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Background: MEIS1::NCOA1\/2 fusion sarcomas are a recently described molecular entity arising predominantly in the genitourinary and gynecologic tracts. Their clinical presentation is often misleading, and no standardized treatment guidelines currently exist. Methods: A literature review was conducted using PubMed to identify all reported cases of molecularly confirmed MEIS1::NCOA1\/2 fusion sarcomas. Clinicopathological, molecular, treatment, and outcome data were extracted for comparative analysis. Case: We report the case of a 38-year-old nulliparous woman who presented with a right vulvar induration clinically consistent with a Bartholin gland cyst. Surgical excision revealed a spindle cell mesenchymal tumor harboring a MEIS1::NCOA2 fusion transcript and a CTNNB1 exon 3 mutation, with probable incomplete resection margins. A local recurrence was documented by MRI and PET\u2013CT at eight months. Surgical re-excision revealed diffuse involvement and complete excision was considered uncertain. Adjuvant external beam radiotherapy followed by an MR-Linac boost was administered. Discussion: This case highlights the diagnostic challenge of Bartholin gland masses. We provide a review of the literature on MEIS1::NCOA1\/2 fusion sarcomas and examine the potential aggressiveness of tumors that additionally harbor a CTNNB1 mutation. Given the nonspecific immunophenotype of this entity, this case underscores the indispensable role of RNA-based molecular sequencing in the diagnosis of low-grade spindle cell tumors when immunohistochemistry proves inconclusive. We further discuss the surgical challenges inherent to this anatomical region and explore intention-to-treat radiotherapy as a potential therapeutic option. Conclusions: We report a rare case of a MEIS1::NCOA2 fusion-positive sarcoma arising in the Bartholin gland region, and, to our knowledge, the first case in which radiotherapy with curative intent has been explored for this entity. This observation expands the limited literature on this emerging clinicopathological entity.<\/jats:p>","DOI":"10.3390\/diagnostics16132098","type":"journal-article","created":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T15:15:04Z","timestamp":1783091704000},"page":"2098","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["MEIS1::NCOA2 Fusion Sarcoma of the Bartholin Gland: A Case Report and Review of the Literature"],"prefix":"10.3390","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-7775-528X","authenticated-orcid":false,"given":"Pauline","family":"Dumonceaux","sequence":"first","affiliation":[{"name":"Department of Gynecology, Cliniques Universitaires Saint-Luc, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lor\u00e9ane","family":"Sims","sequence":"additional","affiliation":[{"name":"Department of Gynecology, Cliniques Universitaires Saint-Luc, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aline","family":"Francois","sequence":"additional","affiliation":[{"name":"Department of Anatomical Pathology, Saint-Luc University, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sabrina","family":"Croce","sequence":"additional","affiliation":[{"name":"Department of Biopathology, Institut Bergoni\u00e9, 33000 Bordeaux, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Latifa","family":"Fellah","sequence":"additional","affiliation":[{"name":"Department of Radiology, Saint-Luc University, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sophie","family":"Cvilic","sequence":"additional","affiliation":[{"name":"Department of Radiation, Oncology Saint-Luc University, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Charlotte","family":"Maillard","sequence":"additional","affiliation":[{"name":"Department of Gynecology, Cliniques Universitaires Saint-Luc, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pascale","family":"Jadoul","sequence":"additional","affiliation":[{"name":"Department of Gynecology, Cliniques Universitaires Saint-Luc, 1200 Brussels, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,7,3]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1097\/00128360-200407000-00006","article-title":"The Bartholin Gland Cyst: Past, Present, and Future","volume":"8","author":"Marzano","year":"2004","journal-title":"J. Low. Genit. Tract Dis."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"395","DOI":"10.1097\/OGX.0b013e31819f9c76","article-title":"Management of Bartholin Duct Cysts and Abscesses: A Systematic Review","volume":"64","author":"Wechter","year":"2009","journal-title":"Obstet. Gynecol. Surv."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"351","DOI":"10.1097\/LGT.0000000000000016","article-title":"Lesions of the Bartholin Gland: A Review","volume":"18","author":"Heller","year":"2014","journal-title":"J. Low. Genit. Tract Dis."},{"key":"ref_4","first-page":"243","article-title":"Carcinomas and Sarcomas of Bartholin Gland. A Report of Nine Cases and Review of the Literature","volume":"35","author":"Kozakiewicz","year":"2014","journal-title":"Eur. J. Gynaecol. Oncol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"171","DOI":"10.1007\/s00404-008-0652-1","article-title":"Vulvar Leiomyosarcoma in Bartholin\u2019s Gland Area: A Case Report and Literature Review","volume":"279","author":"Bartha","year":"2009","journal-title":"Arch. Gynecol. Obstet."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"393","DOI":"10.1006\/gyno.1994.1231","article-title":"Epithelioid Sarcoma of the Bartholin\u2019s Gland Primarily Diagnosed as Vulvar Carcinoma","volume":"54","author":"Konefka","year":"1994","journal-title":"Gynecol. Oncol."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"1562","DOI":"10.1097\/PAS.0000000000001140","article-title":"Novel MEIS1-NCOA2 Gene Fusions Define a Distinct Primitive Spindle Cell Sarcoma of the Kidney","volume":"42","author":"Argani","year":"2018","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1203","DOI":"10.1038\/s41379-021-00744-7","article-title":"Recurrent MEIS1-NCOA2\/1 Fusions in a Subset of Low-Grade Spindle Cell Sarcomas Frequently Involving the Genitourinary and Gynecologic Tracts","volume":"34","author":"Kao","year":"2021","journal-title":"Mod. Pathol."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"227","DOI":"10.1177\/10668969221098081","article-title":"Spindle Cell Sarcoma of the Uterus Harboring MEIS1::NCOA1 Fusion Gene and Mimicking Endometrial Stromal Sarcoma","volume":"31","author":"Mejbel","year":"2023","journal-title":"Int. J. Surg. Pathol."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"417","DOI":"10.1097\/PGP.0000000000000803","article-title":"Spindle Cell Sarcoma of the Uterine Corpus With Adipose Metaplasia: Expanding the Morphologic Spectrum of Neoplasms With MEIS1-NCOA2 Gene Fusion","volume":"41","author":"Kommoss","year":"2022","journal-title":"Int. J. Gynecol. Pathol."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"460","DOI":"10.1097\/PGP.0000000000000937","article-title":"Aggressive High-Grade Uterine Sarcoma Harboring MEIS1-NCOA2 Fusion and Amplification of Multiple 12q13-15 Genes: A Case Report With Morphologic, Immunohistochemical, and Molecular Analysis","volume":"42","author":"Niu","year":"2023","journal-title":"Int. J. Gynecol. Pathol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1097\/PGP.0000000000000777","article-title":"Primary Fallopian Tube Mullerian Adenosarcoma With Sarcomatous Overgrowth and a Previously Unreported MEIS1-NCOA2 Gene Fusion","volume":"41","author":"Ribeiro","year":"2022","journal-title":"Int. J. Gynecol. Pathol."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"47","DOI":"10.1097\/PGP.0000000000000951","article-title":"Uterine MEIS1::NCOA2 Fusion Sarcoma With Lung Metastasis: A Case Report and Review of the Literature","volume":"43","author":"Xing","year":"2024","journal-title":"Int. J. Gynecol. Pathol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"843","DOI":"10.1007\/s00428-025-04079-5","article-title":"Pelvic Spindle Cell Sarcomas Harboring MEIS1::NCOA2 Fusion and Novel Gene Amplifications in 10q23-26 Region: A Potential Predictor for Tumor Progression","volume":"486","author":"Gao","year":"2025","journal-title":"Virchows Arch. Int. J. Pathol."},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"117","DOI":"10.1111\/his.14345","article-title":"Distant Metastatic Potential in Primary Renal Sarcoma with MEIS1-NCOA2 Gene Fusion","volume":"79","author":"Lajara","year":"2021","journal-title":"Histopathology"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"620","DOI":"10.1097\/PAS.0000000000002386","article-title":"MEIS1::NCOA1 Primitive Spindle Cell Sarcoma of the Kidney: Report of 7 Cases of a Distinctive Clinicopathologic Entity","volume":"49","author":"Argani","year":"2025","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"e153","DOI":"10.5001\/omj.2020.74","article-title":"Leiomyosarcoma of the Vulva Mimicking as Chronic Bartholin Cyst: A Case Report","volume":"35","author":"Saquib","year":"2020","journal-title":"Oman Med. J."},{"key":"ref_18","first-page":"e21674","article-title":"Vulvar Leiomyosarcoma Masquerading as a Bartholin\u2019s Gland Cyst in an Adolescent","volume":"14","author":"Reinicke","year":"2022","journal-title":"Cureus"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"201","DOI":"10.21037\/tcr-22-612","article-title":"Bartholin\u2019s Gland Carcinoma-the Diagnostic and Management Challenges of a Rare Malignancy-a Case Report and Review of Current Literature","volume":"12","author":"Addley","year":"2023","journal-title":"Transl. Cancer Res."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1186\/s13000-025-01656-9","article-title":"A Locally Aggressive Pelvic MEIS1::NCOA1 Fusion Sarcoma in a Young Adult Female: A Case Report and Review of the Literature","volume":"20","author":"Ramineni","year":"2025","journal-title":"Diagn. Pathol."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"695","DOI":"10.1097\/PAS.0000000000001227","article-title":"Expanding the Spectrum of Intraosseous Rhabdomyosarcoma: Correlation Between 2 Distinct Gene Fusions and Phenotype","volume":"43","author":"Agaram","year":"2019","journal-title":"Am. J. Surg. Pathol."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1186\/s13000-024-01484-3","article-title":"Low-Grade Undifferentiated Sarcoma with MEIS1::NCOA2-Rearrangement Primary to the Lung: A Case Report","volume":"19","author":"Carter","year":"2024","journal-title":"Diagn. Pathol."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1499","DOI":"10.1136\/ijgc-2024-005823","article-title":"ESGO\/EURACAN\/GCIG Guidelines for the Management of Patients with Uterine Sarcomas","volume":"34","author":"Casali","year":"2024","journal-title":"Int. J. Gynecol. Cancer"},{"key":"ref_24","doi-asserted-by":"crossref","first-page":"1348","DOI":"10.1016\/j.annonc.2021.07.006","article-title":"Soft Tissue and Visceral Sarcomas: ESMO-EURACAN-GENTURIS Clinical Practice Guidelines for Diagnosis, Treatment and Follow-Up\u2606","volume":"32","author":"Gronchi","year":"2021","journal-title":"Ann. 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