Rectal hemolymphangioma: a rare case report and a review of the literature

Introduction and importance: Rectal hemolymphangioma is an exceptionally rare vascular malformation, often presenting with non-specific symptoms, complicating its clinical diagnosis. Due to the overlap in clinical and imaging features with other anorectal conditions, obtaining a definitive preoperative diagnosis is challenging. Case presentation: A 28-year-old woman presented with long-standing rectal bleeding and anemia. She had a background in medical treatment for hemorrhoids, proctitis, and a rectal tumor with defunctioning colostomy before the definitive resectional surgery. Colonoscopic biopsies at this stage revealed only chronic inflammation. The preoperative biopsy at a repeated colonoscopy confirmed rectal lymphangioma and the magnetic resonance imaging revealed circular thickness of the low rectal wall. A decision was made to resect the tumor, and a laparoscopic ultra-low anterior resection was performed. Postoperative histopathologic analysis confirmed the diagnosis of rectal hemolymphangioma. Clinical discussion: Rectal hemolymphangioma is a type of lymphangioma with only four cases previously reported in the literature. The presenting symptoms are typically non-specific, which complicates establishing a definitive diagnosis. Superficial biopsies obtained via colonoscopy frequently yield inconclusive results, and imaging modalities may also lack diagnostic specificity. Level 1 evidence is lacking; however, we believe that definitive surgical treatment with resection is warranted. Conclusion: Rectal hemolymphangioma is an exceedingly rare entity, with only a limited number of cases documented in the literature. It should be considered in the differential diagnosis of anorectal lesions, particularly when distinguishing it from more common pathologies such as rectal cancer and hemorrhoids. Surgical excision remains the preferred treatment modality to prevent potential complications.

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Publication Details

Journal
International Journal of Surgery Open
Published
2025-01-09
DOI
https://doi.org/10.1097/io9.0000000000000247
Primary Topic
Vascular Malformations and Hemangiomas
Type
article
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article

Rectal hemolymphangioma: a rare case report and a review of the literature

Pham Phuc Khanh, Matthew J. F. X. Rickard, Pham Thi Thu Huyen, Le Nhat Huy et al.
International Journal of Surgery Open
Vascular Malformations and Hemangiomas
article

Rectal hemolymphangioma: a rare case report and a review of the literature

Pham Phuc Khanh, Matthew J. F. X. Rickard, Pham Thi Thu Huyen, Le Nhat Huy, Nguyen Quoc Dat, Do Tat Thanh, Nguyen Dac Thao
article en

Abstract

Introduction and importance: Rectal hemolymphangioma is an exceptionally rare vascular malformation, often presenting with non-specific symptoms, complicating its clinical diagnosis. Due to the overlap in clinical and imaging features with other anorectal conditions, obtaining a definitive preoperative diagnosis is challenging. Case presentation: A 28-year-old woman presented with long-standing rectal bleeding and anemia. She had a background in medical treatment for hemorrhoids, proctitis, and a rectal tumor with defunctioning colostomy before the definitive resectional surgery. Colonoscopic biopsies at this stage revealed only chronic inflammation. The preoperative biopsy at a repeated colonoscopy confirmed rectal lymphangioma and the magnetic resonance imaging revealed circular thickness of the low rectal wall. A decision was made to resect the tumor, and a laparoscopic ultra-low anterior resection was performed. Postoperative histopathologic analysis confirmed the diagnosis of rectal hemolymphangioma. Clinical discussion: Rectal hemolymphangioma is a type of lymphangioma with only four cases previously reported in the literature. The presenting symptoms are typically non-specific, which complicates establishing a definitive diagnosis. Superficial biopsies obtained via colonoscopy frequently yield inconclusive results, and imaging modalities may also lack diagnostic specificity. Level 1 evidence is lacking; however, we believe that definitive surgical treatment with resection is warranted. Conclusion: Rectal hemolymphangioma is an exceedingly rare entity, with only a limited number of cases documented in the literature. It should be considered in the differential diagnosis of anorectal lesions, particularly when distinguishing it from more common pathologies such as rectal cancer and hemorrhoids. Surgical excision remains the preferred treatment modality to prevent potential complications.

International Journal of Surgery OpenVol. 63(2)
The University of Sydney (AU), Hanoi Medical University (VN), Concord Repatriation General Hospital (AU), Viet Duc Hospital (VN), Macquarie University (AU)
Gender equality
Openalex Percentile: Top 21%
Vascular Malformations and Hemangiomas
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