{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,14]],"date-time":"2026-09-14T12:03:08Z","timestamp":1789387388107,"version":"build-2803163510"},"reference-count":16,"publisher":"MDPI AG","issue":"3","license":[{"start":{"date-parts":[[2026,9,14]],"date-time":"2026-09-14T00:00:00Z","timestamp":1789344000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Reports"],"abstract":"<jats:p>Background and Clinical Significance: Lymphatic malformations (cystic lymphangiomas) are rare benign tumors in adults, with retroperitoneal and ischioanal involvement being exceptionally uncommon. These lesions often present significant surgical challenges because of their infiltrative growth pattern and high risk of recurrence following incomplete excision. We present a rare case of a giant recurrent retroperitoneal lymphangioma with extension into the pelvic and perineal regions after two previous unsuccessful surgical interventions; Case Presentation: A 46-year-old Hispanic female presented with progressive pelvic and perineal swelling associated with discomfort and functional limitation. The patient had a history of two prior surgeries for lymphangioma recurrence. Imaging studies revealed a large multicystic retroperitoneal mass extending through the pelvis into the ischioanal fossa and perineal region. Given the lesion\u2019s size and complex anatomical involvement, a multidisciplinary surgical approach was planned. The patient underwent a combined abdominal\u2013perineal en bloc resection, achieving complete removal of a 20 \u00d7 8 \u00d7 9.5 cm mass while preserving anal sphincter integrity and function. Histopathological examination confirmed the diagnosis of cystic lymphangioma. The postoperative course was uneventful, and no recurrence was observed for 6 months follow-up; Conclusions: Giant recurrent pelvic lymphangiomas require complete en bloc excision and a multidisciplinary approach to ensure disease clearance and prevent recurrence.<\/jats:p>","DOI":"10.3390\/reports9030312","type":"journal-article","created":{"date-parts":[[2026,9,14]],"date-time":"2026-09-14T10:19:41Z","timestamp":1789381181000},"page":"312","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Giant Recurrent Retroperitoneal and Ischioanal Lymphangioma in an Adult: A Case Report"],"prefix":"10.3390","volume":"9","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7111-5030","authenticated-orcid":false,"given":"Edy","family":"Quizhpe","sequence":"first","affiliation":[{"name":"Facultad de Ciencias de la Salud \u201cEugenio Espejo\u201d, Universidad UTE, Quito 170527, Ecuador"},{"name":"Hospital General del Sur de Quito, Instituto Ecuatoriano de Seguridad Social (IESS), Quito 170132, Ecuador"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3245-5937","authenticated-orcid":false,"given":"Alex Pa\u00fal","family":"Guachilema Rivadeneira","sequence":"additional","affiliation":[{"name":"Hospital General Enrique Garces, Ministerio de Salud P\u00fablica (MSP), Quito 170606, Ecuador"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jaime Eduardo","family":"Ort\u00edz Flores","sequence":"additional","affiliation":[{"name":"Hospital General Enrique Garces, Ministerio de Salud P\u00fablica (MSP), Quito 170606, Ecuador"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Melany Micaela","family":"Montalvo Cevallos","sequence":"additional","affiliation":[{"name":"Facultad de Salud y Bienestar, Pontificia Universidad Cat\u00f3lica del Ecuador, Quito 170525, Ecuador"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2026,9,14]]},"reference":[{"key":"ref_1","unstructured":"International Society for the Study of Vascular Anomalies (2026, February 18). ISSVA Classification of Vascular Anomalies (2025 Update). ISSVA. Available online: https:\/\/www.issva.org\/classification."},{"key":"ref_2","first-page":"e69209","article-title":"Reviving the Rarity of Cecal Lymphangioma: A Case Report","volume":"16","author":"Eppley","year":"2024","journal-title":"Cureus"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"1006","DOI":"10.18203\/2349-2902.isj20251543","article-title":"A rare case of cystic lymphangioma of the ascending colon mesentery in an adult woman: Clinical, radiological, and histopathological findings","volume":"12","author":"Vermylen","year":"2025","journal-title":"Int. Surg. J."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"027","DOI":"10.17352\/2455-2283.000125","article-title":"Intra-Abdominal Multi Cystic Lymphangiomas: A Case Series with Adult and Pediatric Literature Review","volume":"10","author":"Avani","year":"2024","journal-title":"Arch. Clin. Gastroenterol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"171","DOI":"10.4291\/wjgp.v1.i5.171","article-title":"Retroperitoneal cystic lymphangioma in an adult: A case report and review of the literature","volume":"5","author":"Bhavsar","year":"2010","journal-title":"World J. Gastrointest. Pathophysiol."},{"key":"ref_6","first-page":"e103836","article-title":"Pelvic Lymphangioma: The Purpose of a Clinical Case","volume":"2","author":"Baiherych","year":"2026","journal-title":"Cureus"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"102328","DOI":"10.1016\/j.jvsv.2025.102328","article-title":"Surgical treatment of perineal lymphatic malformations: A single-center retrospective study of 24 years","volume":"14","author":"Liu","year":"2025","journal-title":"J. Vasc. Surg. Venous Lymphat. Disord."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"715","DOI":"10.3349\/ymj.2005.46.5.715","article-title":"Recurrent retroperitoneal cystic lymphangioma","volume":"5","author":"Ozdemir","year":"2005","journal-title":"Yonsei Med. J."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1097\/JS9.0000000000000373","article-title":"The SCARE 2023 guideline: Updating consensus Surgical CAse REport (SCARE) guidelines","volume":"109","author":"Sohrabi","year":"2023","journal-title":"Int. J. Surg."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"91","DOI":"10.4166\/kjg.2023.057","article-title":"Cystic Lymphangioma of Rectum-A Case Report and Review of Literature","volume":"82","author":"Dessai","year":"2023","journal-title":"Korean J. Gastroenterol."},{"key":"ref_11","doi-asserted-by":"crossref","unstructured":"Zhao, N., Fu, Y., Wang, Z., An, Q., and Jia, W. (2022). Case report: Submucosal cavernous lymphangioma causing jejuno-jejunal intussusception in an adult. Front. Surg., 9.","DOI":"10.3389\/fsurg.2022.953840"},{"key":"ref_12","first-page":"154","article-title":"Sclerotherapy for lymphatic malformations of head and neck: Systematic review and meta-analysis","volume":"1","author":"Balakrishnan","year":"2020","journal-title":"J. Vasc. Surg. Venous Lymphat. Disord."},{"key":"ref_13","first-page":"e932607","article-title":"Cavernous lymphangioma of the small bowel mesentery: A case report and literature review","volume":"22","author":"Drachneris","year":"2021","journal-title":"Am. J. Case Rep."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"15","DOI":"10.14740\/wjon1561","article-title":"Adult Primary Retroperitoneal Lymphangioma: Updated Facts","volume":"14","author":"Mansour","year":"2023","journal-title":"World J. Oncol."},{"key":"ref_15","first-page":"e92002","article-title":"CT and MRI Characterization of a Retroperitoneal Lymphatic Malformation in an Adult","volume":"17","author":"Saucedo","year":"2025","journal-title":"Cureus"},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1760065","DOI":"10.3389\/fsurg.2026.1760065","article-title":"Clinical features and outcomes of adult primary retroperitoneal lymphangioma: Insights from a high-volume sarcoma center","volume":"13","author":"Li","year":"2026","journal-title":"Front. 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