Anal canal duplication in children: a monocentric experience of 12 cases

Ailhaud, Antoine; Alao, Oyekashopefoluw; Cruz, Eva Sole; Faguet, Romain; Verot, Pierre-Louis; Piolat, C.; Jacquier, C.; Rabattu, Pierre‐Yves

doi:10.1007/s00383-021-04910-5

Cited by 4 publications

(8 citation statements)

References 10 publications

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“…ACD is characterized by an extra perineal orifice located right behind the normal anus at 6 o’clock, with a blind-ending tubular structure, not opening into the anorectum. According to the literature, more than half of the cases are reported to be asymptomatic ( 4 , 9 ). Generally, the abnormality is discovered incidentally or during the careful perineum inspection at birth.…”

Section: Discussionmentioning

confidence: 99%

Anal Canal Duplication Mimicking Recurrent Abscess: A Case Report and Review of the Literature

Liu

Feng

et al. 2022

Front. Surg.

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BackgroundAnal canal duplication (ACD) is a very rare duplication of the gastrointestinal tract and is described as a secondary anal orifice along the posterior side of the normal anal canal. Early surgical removal is advisable, also in asymptomatic patients, because of the risk of inflammatory complications, such as recurrent crissum abscess, and malignant changes.Case presentationA previously healthy 2-year-old boy was evaluated in the emergency department with fever. He complained of anal pain in the absence of incentive. Physical examination and ultrasound confirmed a diagnosis of perianal abscess. He was treated with incision and drainage of the abscess and intravenous antibiotics. Two months after his discharge from the hospital, he developed fever and had intervals discharge pus and pain in the same locations. Colorectal endoscopy revealed that there was no fistula opening at the rectal wall. Intraoperative fistulography showed a fistulous tract that was connected to a subcutaneous cavity. Excision of the fistulous tract and wide drainage of the deep postanal space were performed. The patient was referred to our hospital for further evaluation 6 months later. Physical examination showed a secondary anus that had not been noticed before. MRI showed an anal fistula between 1 and 3 o’clock, and preoperative fistulography revealed a 3-cm-long tubular structure without any connection with the rectum. The diagnosis of ACD was made by intraoperative examination with a metal catheter and the postoperative pathological analysis. The duplicated anal canal was resected completely via a perianal approach without any rectal injury. Histology showed a squamous epithelium in the distal end with some smooth-muscle fibers. After a follow-up of 8 months, the patient has been doing well.ConclusionRecurrent crissum abscess should raise clinical attention to alimentary tract congenital malformations such as ACD. Prompt recognition of these unique presentations of ACD is needed, and complete excision through a perineal approach or posterior sagittal approach is recommended.

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Section: Discussionmentioning

confidence: 99%

Anal Canal Duplication Mimicking Recurrent Abscess: A Case Report and Review of the Literature

Liu

Feng

et al. 2022

Front. Surg.

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“…ACD is a congenital malformation that typically manifests early in life [4,5], but in a minority of cases such as the current case, it manifests in adulthood [1]. In a compilation of case series and case reports with a total of 72 patients, the maximum age was 14 years [6], much younger than the case we present.…”

Section: Discussionmentioning

confidence: 62%

“…ACD is one of the rarest duplications of the gastrointestinal tract [9]. To our knowledge, as Ailhaud et al, Trecartin et al, and Özbey et al had reported, no more than 90 or 100 cases had been reported in the literature to date [5,6,10]. Despite being a rare pathology, ACD should be considered in the differential diagnosis of other anorectal pathologies.…”

Section: Introductionmentioning

confidence: 77%

“…It was first described in 1956 by Dukes et al [1]; subsequently, Ochiai et al defined it as a lesion that should include "a squamous epithelium at the caudal end, a transitional epithelium at the cranial end, and smooth muscle cells in the wall of the duplicated anal canal" [3]. ACD presents and is usually diagnosed early in life [4,5]. Thirty-six percent of cases may be associated with other malformations [4] such as myelomeningocele, midline defects [6,7], or most commonly, urinary malformations in up to 45% of cases [8].…”

Section: Introductionmentioning

confidence: 99%

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Anal Canal Duplication in a 25-Year-Old Female Patient

Gutierrez-Alvarez¹,

Fiallo²,

Fuentes³

et al. 2023

Cureus

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Anal canal duplication (ACD) is a congenital malformation that typically presents and is diagnosed early in life. It can be associated with other syndromes or congenital malformations. ACD is one of the rarest duplications of the gastrointestinal tract, with no more than 90 to 100 cases reported in the literature. It can be confused with more frequent pathologies such as perianal fistula, especially when it occurs in adulthood. We present the case of a 25-year-old female patient who presents with a second orifice above the native anal orifice. An arthroscopic examination was performed, an incidental diagnosis of ACD was made, and a complete excision of the duplicated anal canal was performed. The aim of the study is to expand the information on this rare pathology in order to take it into account as a differential diagnosis in patients with abscesses, recurrent fistulous tracts, or any other anorectal pathology.

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“…The pathology criteria were set by Ochiai et al (2) in 2002, as follows: the presence of (1) squamous epithelial cells in the caudal end, (2) transitional epithelium in the cranial end, and (3) smooth muscle cells in the lesion wall. There were only about 110 cases described in the literature, with a female-to-male ratio of 15:1 (3)(4)(5)(6)(7)(8). Patients usually present with a second opening in the midline, at 6 o'clock behind the true anus (9,10).…”

Section: Introductionmentioning

confidence: 99%