Case Report
Copyright ©The Author(s) 2024. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 6, 2024; 12(10): 1778-1784
Published online Apr 6, 2024. doi: 10.12998/wjcc.v12.i10.1778
Cellular angiofibroma arising from the rectocutaneous fistula in an adult: A case report
Hao-En Chen, Yu-Yang Lu, Ruei-Yu Su, Hong-Hau Wang, Chao-Yang Chen, Je-Ming Hu, Jung-Cheng Kang, Kuan-Hsun Lin, Ta-Wei Pu
Hao-En Chen, Department of Surgery, Taichung Armed Forces General Hospital, Taichung 411, Taiwan
Hao-En Chen, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
Yu-Yang Lu, Department of Pediatrics, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
Ruei-Yu Su, Department of Pathology, Tri-Service General Hospital, National Defense Medical Center, Taipei 105, Taiwan
Ruei-Yu Su, Department of Pathology and Laboratory Medicine, Taoyuan Armed Forces General Hospital, Taoyuan 32551, Taiwan
Hong-Hau Wang, Department of Radiology, Tri-Service General Hospital Songshan Branch, National Defense Medical Center, Taipei 105, Taiwan
Chao-Yang Chen, Je-Ming Hu, Division of Colon and Rectal Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
Jung-Cheng Kang, Division of Colon and Rectal Surgery, Department of Surgery, Taiwan Adventist Hospital, Taipei 105, Taiwan
Kuan-Hsun Lin, Division of Thoracic Surgery, Department of Surgery, Tri-Service General Hospital, National Defense Medical Center, Taipei 114, Taiwan
Ta-Wei Pu, Division of Colon and Rectal Surgery, Department of Surgery, Tri-Service General Hospital Songshan Branch, National Defense Medical Center, Taipei 105, Taiwan
Author contributions: Chen HE, Lu YY, Su RY, and Wang HH wrote and edited the manuscript; Chen CY and Hu JM collected data; Kang JC, Lin KS, and Pu TW contributed to the conceptualization and supervision of the study; All authors read and approved the final manuscript.
Informed consent statement: Informed written consent was obtained from the patients for the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors have no conflicts of interest to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Ta-Wei Pu, MD, Lecturer, Division of Colon and Rectal Surgery, Department of Surgery, Tri-Service General Hospital Songshan Branch, National Defense Medical Center, No. 131 Jiankang Road, Taipei 105, Taiwan. tawei0131@gmail.com
Received: November 2, 2023
Peer-review started: November 2, 2023
First decision: January 15, 2024
Revised: February 3, 2024
Accepted: March 5, 2024
Article in press: March 5, 2024
Published online: April 6, 2024
Abstract
BACKGROUND

Rectocutaneous fistulae are common. The infection originates within the anal glands and subsequently extends into adjacent regions, ultimately resulting in fistula development. Cellular angiofibroma (CAF), also known as an angiomyofibroblastoma-like tumor, is a rare benign soft tissue neoplasm predominantly observed in the scrotum, perineum, and inguinal area in males and in the vulva in females. We describe the first documented case CAF that developed within a rectocutaneous fistula and manifested as a perineal mass.

CASE SUMMARY

In the outpatient setting, a 52-year-old male patient presented with a 2-year history of a growing perineal mass, accompanied by throbbing pain and minor scrotal abrasion. Physical examination revealed a soft, well-defined, non-tender mass at the left buttock that extended towards the perineum, without a visible opening. The initial assessment identified a soft tissue tumor, and the laboratory data were within normal ranges. Abdominal and pelvic computed tomography (CT) revealed swelling of the abscess cavity that was linked to a rectal cutaneous fistula, with a track-like lesion measuring 6 cm × 0.7 cm in the left perineal region and attached to the left rectum. Rectoscope examination found no significant inner orifices. A left medial gluteal incision revealed a thick-walled mass, which was excised along with the extending tract, and curettage was performed. Histopathological examination confirmed CAF diagnosis. The patient achieved total resolution during follow-up assessments and did not require additional hospitalization.

CONCLUSION

CT imaging supports perineal lesion diagnosis and management. Perineal angiofibromas, even with a cutaneous fistula, can be excised transperineally.

Keywords: Angiofibroma, Perineal mass, Rectocutaneous fistula, Anorectal fistula, Anal fistula, Case report

Core Tip: Although uncommon, certain typical clinical conditions, such as rectal cutaneous fistulas, may manifest in complex presentations, as demonstrated in this patient. Notably, recurrence and complications such as incontinence are frequently associated with complex fistulas and incomplete mapping. Therefore, establishing a definitive diagnosis through a precise mapping of the fistula is imperative before surgical intervention. Computed tomography imaging is useful for detecting, understanding and managing perineal lesions. Perineal angiofibromas can be removed via transperineal excision, even if linked to a rectal cutaneous fistula. In such complex cases, a personalized and careful diagnostic approach is crucial.