{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T16:38:05Z","timestamp":1789663085471,"version":"4.0.1"},"reference-count":55,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"4","license":[{"start":{"date-parts":[[2025,1,24]],"date-time":"2025-01-24T00:00:00Z","timestamp":1737676800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2025,1,24]]},"abstract":"<jats:sec>\n            <jats:title>Rationale:<\/jats:title>\n            <jats:p>Aggressive angiomyxoma (AAM) is an exceptionally rare mesenchymal tumor that predominantly manifests in the female genital organs during the reproductive age. Its rarity alone makes it a fascinating subject for study. The diagnosis of AAM necessitates differentiation from other benign or mesenchymal tumors and can be confirmed through immunohistochemistry (IHC) staining. Surgical resection is the primary treatment, and adjuvant treatment can be used as hormonal therapy with gonadotropin-releasing hormone agonists, selective estrogen receptor modulators, and aromatase inhibitors.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Patient concerns:<\/jats:title>\n            <jats:p>A 44-year-old premenopausal Korean woman presented with a growing perineal mass and frequent urination.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Diagnoses:<\/jats:title>\n            <jats:p>Histopathological findings confirmed AAM, with IHC staining showing estrogen receptor, progesterone receptor, actin and desmin positivity, and CD34 and S100 negativity.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Interventions:<\/jats:title>\n            <jats:p>The mass was excised transvaginally under general anesthesia.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Outcomes:<\/jats:title>\n            <jats:p>The patient showed no signs of recurrence 6 months postoperatively.<\/jats:p>\n          <\/jats:sec>\n          <jats:sec>\n            <jats:title>Lessons:<\/jats:title>\n            <jats:p>AAM in the vagina is a rare tumor that requires differential diagnosis using IHC staining. Previously, we reviewed reported cases and confirmed the feasibility and effectiveness of surgery as the main treatment. This might reassure us about the potential successful treatment of AAM. Adjuvant hormonal therapy with gonadotropin-releasing hormone agonists, selective estrogen receptor modulators, and aromatase inhibitors can further reduce the risk of recurrence.<\/jats:p>\n          <\/jats:sec>","DOI":"10.1097\/md.0000000000041287","type":"journal-article","created":{"date-parts":[[2025,1,24]],"date-time":"2025-01-24T23:01:19Z","timestamp":1737759679000},"page":"e41287","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":7,"title":["Aggressive angiomyxoma of the vagina: A case report and literature review"],"prefix":"10.1097","volume":"104","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7558-5363","authenticated-orcid":false,"given":"Bo Ram","family":"Yu","sequence":"first","affiliation":[{"name":"Department of Obstetrics and Gynecology, Jeonbuk National University Hospital, Jeonbuk National University Medical School, Jeonju, Republic of Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4891-0241","authenticated-orcid":false,"given":"Won Ku","family":"Choi","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Jeonbuk National University Hospital, Jeonbuk National University Medical School, Jeonju, Republic of Korea"},{"name":"Research Institute of Clinical Medicine, Jeonbuk National University, Biomedical Research Institute of Jeonbuk National University Hospital, Jeonju, Republic of Korea."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1557-9575","authenticated-orcid":false,"given":"Dong Hyu","family":"Cho","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Jeonbuk National University Hospital, Jeonbuk National University Medical School, Jeonju, Republic of Korea"},{"name":"Research Institute of Clinical Medicine, Jeonbuk National University, Biomedical Research Institute of Jeonbuk National University Hospital, Jeonju, Republic of Korea."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6236-9319","authenticated-orcid":false,"given":"Na-Ri","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Hematology and Oncology, Jeonbuk National University Hospital, Jeonbuk National University Medical School, Jeonju, Republic of Korea"},{"name":"Research Institute of Clinical Medicine, Jeonbuk National University, Biomedical Research Institute of Jeonbuk National University Hospital, Jeonju, Republic of Korea."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","reference":[{"key":"R1-20250124","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1016\/j.urology.2020.05.028","article-title":"Aggressive angiomyxoma in an 11-year-old boy \u2013 diagnostic and therapeutic dilemmas: an unusual case report and review of the literature.","volume":"144","author":"Korecka","year":"2020","journal-title":"Urology"},{"key":"R2-20250124","doi-asserted-by":"crossref","first-page":"603","DOI":"10.1136\/jcp.53.8.603","article-title":"Aggressive angiomyxoma of pelvic parts exhibits oestrogen and progesterone receptor positivity.","volume":"53","author":"McCluggage","year":"2000","journal-title":"J Clin Pathol"},{"key":"R3-20250124","first-page":"1","article-title":"Characteristics and treatment strategies of aggressive angiomyxoma in women: a retrospective review of 87 cases.","volume":"10","author":"Wang","year":"2023","journal-title":"Front Surg"},{"key":"R4-20250124","doi-asserted-by":"crossref","first-page":"164","DOI":"10.5468\/ogs.2014.57.2.164","article-title":"Aggressive angiomyxoma of the vulva: a case report.","volume":"57","author":"Lee","year":"2014","journal-title":"Obstet Gynecol Sci"},{"key":"R5-20250124","doi-asserted-by":"crossref","first-page":"505","DOI":"10.1097\/PGP.0b013e318211d56c","article-title":"Aggressive angiomyoma of the female genital tract and pelvis \u2013 clinicopathologic features with immunohistochemical analysis.","volume":"30","author":"Bigby","year":"2011","journal-title":"Int J Gynecol Pathol"},{"key":"R6-20250124","doi-asserted-by":"crossref","first-page":"981","DOI":"10.1002\/gcc.20483","article-title":"Frequency and characterization of HMGA2 and HMGA1 rearrangements in mesenchymal tumors of the lower genital tract.","volume":"46","author":"Medeiros","year":"2007","journal-title":"Genes Chromosomes Cancer"},{"key":"R7-20250124","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1097\/PGP.0000000000000689","article-title":"HMGA2 is a useful marker of vulvovaginal aggressive angiomyxoma but may be positive in other mesenchymal lesions at this site.","volume":"40","author":"Harkness","year":"2021","journal-title":"Int J Gynecol Pathol"},{"key":"R8-20250124","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1136\/bcr-2018-227475","article-title":"Novel HMGA2-YAP1 fusion gene in aggressive angiomyxoma.","volume":"12","author":"Lee","year":"2019","journal-title":"BMJ Case Rep"},{"key":"R9-20250124","doi-asserted-by":"crossref","first-page":"838","DOI":"10.1007\/s00428-006-0186-5","article-title":"Deregulation of HMGA2 in an aggressive angiomyxoma with t (11; 12)(q23; q15).","volume":"448","author":"Micci","year":"2006","journal-title":"Virchows Arch"},{"key":"R10-20250124","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/1752-1947-7-153","article-title":"Inverted Y incision and trans-sacral approach in retroperitoneal aggressive angiomyxoma: a case report.","volume":"7","author":"Hong","year":"2013","journal-title":"J Med Case Rep"},{"key":"R11-20250124","doi-asserted-by":"crossref","first-page":"101204","DOI":"10.1016\/j.gore.2023.101204","article-title":"A tale of two vulvar angiomyxomas: two cases and review of literature.","volume":"47","author":"Navitski","year":"2023","journal-title":"Gynecol Oncol Rep"},{"key":"R12-20250124","first-page":"514","article-title":"Aggressive angiomyxoma of the pelvis: 35-year experience.","volume":"67","author":"Cardenas Lara","year":"2024","journal-title":"Dis Colon Rectum"},{"key":"R13-20250124","doi-asserted-by":"crossref","first-page":"e536","DOI":"10.1634\/theoncologist.2018-0338","article-title":"Treatment outcomes and sensitivity to hormone therapy of aggressive angiomyxoma: a multicenter, international, retrospective study.","volume":"24","author":"Fuc\u00e0","year":"2018","journal-title":"Oncologist"},{"key":"R14-20250124","doi-asserted-by":"crossref","first-page":"335","DOI":"10.1016\/j.ejso.2009.11.006","article-title":"Aggressive angiomyxoma: a case series and literature review.","volume":"36","author":"Haldar","year":"2010","journal-title":"Eur J Surg Oncol"},{"key":"R15-20250124","first-page":"e12023","article-title":"Aggressive angiomyxoma of the posterior wall of the uterus.","volume":"12","author":"Alosaimi","year":"2020","journal-title":"Cureus"},{"key":"R16-20250124","doi-asserted-by":"crossref","first-page":"82","DOI":"10.5603\/GP.a2020.0183","article-title":"Long-term response to hormone therapy in a young woman with aggressive pelvic angiomyxoma.","volume":"92","author":"Walczak","year":"2021","journal-title":"Ginekol Pol"},{"key":"R17-20250124","doi-asserted-by":"crossref","first-page":"172","DOI":"10.1016\/j.ygyno.2011.06.013","article-title":"Leuprolide acetate depot plus high-dose raloxifene hydrochloride before and after surgery for recurrent vaginal aggressive angiomyxoma: a case report.","volume":"123","author":"Palomba","year":"2011","journal-title":"Gynecol Oncol"},{"key":"R18-20250124","doi-asserted-by":"crossref","first-page":"e23","DOI":"10.1016\/j.athoracsur.2017.01.118","article-title":"Successful surgical treatment of an aggressive pelvic angiomyxoma with intracardiac extension.","volume":"104","author":"Husso","year":"2017","journal-title":"Ann Thorac Surg"},{"key":"R19-20250124","doi-asserted-by":"crossref","first-page":"e59","DOI":"10.1308\/rcsann.2020.7008","article-title":"Aggressive angiomyxoma of the ischioanal fossa in a post-menopausal woman.","volume":"103","author":"Peterknecht","year":"2021","journal-title":"Ann R Coll Surg Engl"},{"key":"R20-20250124","doi-asserted-by":"crossref","first-page":"819","DOI":"10.3109\/01443615.2016.1157151","article-title":"Treatment of aggressive angiomyxoma of the female perineum: Combined operative and hormone therapy.","volume":"36","author":"Im","year":"2016","journal-title":"J Obstet Gynaecol"},{"key":"R21-20250124","doi-asserted-by":"crossref","first-page":"120","DOI":"10.1006\/gyno.2000.6119","article-title":"Primary medical management of recurrent aggressive angiomyxoma of the vulva with a gonadotropin-releasing hormone agonist.","volume":"81","author":"Fine","year":"2001","journal-title":"Gynecol Oncol"},{"key":"R22-20250124","first-page":"111","article-title":"Aggressive angiomyxoma of the female urethra.","volume":"35","author":"Elsaqa","year":"2022","journal-title":"Proc (Bayl Univ Med Cent)"},{"key":"R23-20250124","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1186\/s13256-022-03284-z","article-title":"Aggressive (deep) angiomyxoma of the vulva: a case report.","volume":"16","author":"Goyal","year":"2022","journal-title":"J Med Case Rep"},{"key":"R24-20250124","doi-asserted-by":"crossref","first-page":"100765","DOI":"10.1016\/j.gore.2021.100765","article-title":"A case series of aggressive angiomyxoma: using morphologic type and hormonal modification to tailor treatment.","volume":"36","author":"Kooy","year":"2021","journal-title":"Gynecol Oncol Rep"},{"key":"R25-20250124","doi-asserted-by":"crossref","first-page":"105756","DOI":"10.1016\/j.ijscr.2021.105756","article-title":"Aggressive angiomyxoma of the pelvis surgical management in a case with delayed diagnosis.","volume":"81","author":"Akhavan","year":"2021","journal-title":"Int J Surg Case Rep"},{"key":"R26-20250124","first-page":"102706","article-title":"Aggressive angiomyxoma of vulva in 28-years old patient: a case report of second recurrence.","volume":"69","author":"Zamani","year":"2021","journal-title":"Ann Med Surg (Lond)"},{"key":"R27-20250124","doi-asserted-by":"crossref","first-page":"e241550","DOI":"10.1136\/bcr-2021-241550","article-title":"Aggressive angiomyxoma of left buttock.","volume":"14","author":"Akram","year":"2021","journal-title":"BMJ Case Rep"},{"key":"R28-20250124","doi-asserted-by":"crossref","first-page":"rjaa330","DOI":"10.1093\/jscr\/rjaa330","article-title":"Aggressive angiomyxoma persistently misdiagnosed as an obturator hernia managed with resection and hormonal therapy: case report.","volume":"2020","author":"Alomary","year":"2020","journal-title":"J Surg Case Rep"},{"key":"R29-20250124","doi-asserted-by":"crossref","first-page":"300060520936414","DOI":"10.1177\/0300060520936414","article-title":"Aggressive angiomyxoma in pregnancy: a case report and literature review.","volume":"48","author":"Xu","year":"2020","journal-title":"J Int Med Res"},{"key":"R30-20250124","first-page":"1816","article-title":"Clinicopathologic and treatment features of aggressive angiomyxoma: a case report.","volume":"12","author":"Lin","year":"2019","journal-title":"Int J Clin Exp Pathol"},{"key":"R31-20250124","doi-asserted-by":"crossref","first-page":"e226973","DOI":"10.1136\/bcr-2018-226973","article-title":"Aggressive angiomyxoma: giant recurrence successfully treated with wide excision and adjuvant therapy with GnRH analogue.","volume":"11","author":"Aguilar-Frasco","year":"2018","journal-title":"BMJ Case Rep"},{"key":"R32-20250124","doi-asserted-by":"crossref","first-page":"885","DOI":"10.1097\/AOG.0000000000002254","article-title":"Aggressive angiomyxoma of the vulva and bladder.","volume":"130","author":"Song","year":"2017","journal-title":"Obstet Gynecol"},{"key":"R33-20250124","doi-asserted-by":"crossref","first-page":"E55","DOI":"10.1097\/LGT.0b013e3182a22019","article-title":"Hormonal therapy for aggressive angiomyxoma: a case report and proposed management algorithm.","volume":"18","author":"Schwartz","year":"2014","journal-title":"J Low Genit Tract Dis"},{"key":"R34-20250124","doi-asserted-by":"crossref","first-page":"657","DOI":"10.1016\/j.diii.2013.01.009","article-title":"Aggressive angiomyxoma.","volume":"94","author":"Gay","year":"2013","journal-title":"Diagn Interventional Imaging"},{"key":"R35-20250124","first-page":"686","article-title":"Aggressive angiomyxoma of the vulva treated by using a gonadotropin-releasing hormone agonist: a case report.","volume":"32","author":"Lee","year":"2011","journal-title":"Eur J Gynaecol Oncol"},{"key":"R36-20250124","doi-asserted-by":"crossref","first-page":"110","DOI":"10.1016\/j.eucr.2017.03.019","article-title":"Aggressive angiomyxoma involving penis and urethra\u2013a case report.","volume":"13","author":"Damodaran","year":"2017","journal-title":"Urol Case Rep"},{"key":"R37-20250124","doi-asserted-by":"crossref","first-page":"589","DOI":"10.4103\/0019-509X.178398","article-title":"Aggressive angiomyxoma perineum: a rare soft tissue neoplasm in males.","volume":"52","author":"Mathur","year":"2015","journal-title":"Indian J Cancer"},{"key":"R38-20250124","doi-asserted-by":"crossref","first-page":"e240687","DOI":"10.1136\/bcr-2020-240687","article-title":"Vulvar aggressive angiomyxoma: a surgical challenge.","volume":"14","author":"Srivastava","year":"2021","journal-title":"BMJ Case Rep"},{"key":"R39-20250124","first-page":"9256749","article-title":"A giant aggressive angiomyxoma of the pelvis misdiagnosed as incarcerated femoral hernia: a case report and review of the literature.","volume":"2016","author":"Sozutek","year":"2016","journal-title":"Case Rep Surg"},{"key":"R40-20250124","doi-asserted-by":"crossref","first-page":"1097","DOI":"10.1136\/ijgc-00009577-200511000-00013","article-title":"Aggressive angiomyxoma of the perineum.","volume":"15","author":"Abu","year":"2005","journal-title":"Int J Gynecol Cancer"},{"key":"R41-20250124","doi-asserted-by":"crossref","first-page":"207","DOI":"10.1016\/j.jsbmb.2014.03.003","article-title":"An overview of current and emerging SERMs.","volume":"143","author":"Komm","year":"2014","journal-title":"J Steroid Biochem Mol Biol"},{"key":"R42-20250124","doi-asserted-by":"crossref","first-page":"82","DOI":"10.1016\/j.steroids.2014.07.012","article-title":"Ospemifene: a first-in-class, non-hormonal selective estrogen receptor modulator approved for the treatment of dyspareunia associated with vulvar and vaginal atrophy.","volume":"90","author":"DeGregorio","year":"2014","journal-title":"Steroids"},{"key":"R43-20250124","doi-asserted-by":"crossref","first-page":"3403","DOI":"10.3390\/cancers15133403","article-title":"Aggressive angiomyxoma of the lower female genital tract in pregnancy: a review of the MITO rare tumors group.","volume":"15","author":"Cicogna","year":"2023","journal-title":"Cancers (Basel)"},{"key":"R44-20250124","doi-asserted-by":"crossref","first-page":"1375","DOI":"10.3390\/cancers16071375","article-title":"Aggressive angiomyxoma of the lower female genital tract: a review of the MITO rare tumors group.","volume":"16","author":"Dellino","year":"2024","journal-title":"Cancers (Basel)"},{"key":"R45-20250124","doi-asserted-by":"crossref","first-page":"1726","DOI":"10.3390\/jcm12051726","article-title":"Aggressive angiomyxoma of the vulva: which is the best management strategy? Description of a case report and review of literature of the last ten years.","volume":"12","author":"Gulino","year":"2023","journal-title":"J Clin Med"},{"key":"R46-20250124","doi-asserted-by":"crossref","first-page":"107","DOI":"10.3390\/medicina58010107","article-title":"Genital prolapse in pregnant woman as a presentation of aggressive angiomyxoma: case report and literature review.","volume":"58","author":"Espejo-Reina","year":"2022","journal-title":"Medicina (Kaunas)"},{"key":"R47-20250124","doi-asserted-by":"crossref","first-page":"107313","DOI":"10.1016\/j.ijscr.2022.107313","article-title":"A deep giant aggressive angiomyxoma of the labia majora: a case report.","volume":"96","author":"Ayati","year":"2022","journal-title":"Int J Surg Case Rep"},{"key":"R48-20250124","first-page":"5604460","article-title":"A paraurethral aggressive (deep) angiomyxoma.","volume":"2022","author":"York","year":"2022","journal-title":"Case Rep Obstet Gynecol"},{"key":"R49-20250124","doi-asserted-by":"crossref","first-page":"105948","DOI":"10.1016\/j.ijscr.2021.105948","article-title":"Deep (aggressive) angiomyxoma of the vagina misdiagnosed as Gartner cyst: a case report.","volume":"83","author":"Djusad","year":"2021","journal-title":"Int J Surg Case Rep"},{"key":"R50-20250124","doi-asserted-by":"crossref","first-page":"2050313X19843391","DOI":"10.1177\/2050313X19843391","article-title":"Aggressive angiomyxoma of the perineal region.","volume":"7","author":"Raptin","year":"2019","journal-title":"SAGE Open Med Case Rep"},{"key":"R51-20250124","first-page":"292304","article-title":"Aggressive angiomyxoma of the vulva: a bizarre perineal lesion.","volume":"2015","author":"Zizi-Sermpetzoglou","year":"2015","journal-title":"Case Rep Oncol Med"},{"key":"R52-20250124","doi-asserted-by":"crossref","first-page":"bcr2014207287","DOI":"10.1136\/bcr-2014-207287","article-title":"Paravaginal aggressive angiomyxoma.","volume":"2015","author":"Saadeh","year":"2015","journal-title":"BMJ Case Rep"},{"key":"R53-20250124","doi-asserted-by":"crossref","first-page":"525","DOI":"10.5468\/ogs.2015.58.6.525","article-title":"Alternative surgical approaches for aggressive angiomyxoma at different sites in the pelvic cavity.","volume":"58","author":"Choi","year":"2015","journal-title":"Obstet Gynecol Sci"},{"key":"R54-20250124","first-page":"465","article-title":"Aggressive angiomyxoma of the female genital tract: report of two cases.","volume":"35","author":"Ki","year":"2014","journal-title":"Eur J Gynaecol Oncol"},{"key":"R55-20250124","first-page":"1","article-title":"Aggressive angiomyxoma of the pelvis and abdominal wall: dramatic response to chemical ablation therapy.","volume":"13","author":"Li","year":"2023","journal-title":"Front Oncol"}],"container-title":["Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.lww.com\/10.1097\/MD.0000000000041287","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,24]],"date-time":"2025-01-24T23:01:33Z","timestamp":1737759693000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/10.1097\/MD.0000000000041287"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,1,24]]},"references-count":55,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025]]}},"URL":"https:\/\/doi.org\/10.1097\/md.0000000000041287","relation":{},"ISSN":["1536-5964"],"issn-type":[{"value":"1536-5964","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,1,24]]}}}