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...omplications: inflammation and malignancy. Nevertheless they refused surgical mucosal stripping. 2. Discussion ACD is the least frequent digestive duplication. Clinically, it presents itself as an extra perineal orifice located just behind the anus. Clinically, it is difficult to differentiate ACD from a rectal or anal fistula, however, in noncomplicated ACD inflammation will be absent. Only histology gives diagnostic certainty describing 3 characteristics of ACD: squamous epithelium in the caudal end, transitional epithelium in the cranial end and smooth-muscle cells in the wall of the canal [1, 2]. It is most frequently a tubular (90%) anomaly without communication to the rectum. In 10% of cases, the lesion is cystic [3]. We found only 55 patients (including our patient) withACD inEnglish literature (Table 1). Females comprise up to 89% of the patients with ACD (Table 1). Twohypotheses concerning the origin of anal canal duplication are suggested in literature. Choi and Park postulate it as a consequence of recanalization of a cloacal membrane excess in late embryonic life [4]. Hamada et al. suggest a duplication of the dorsal cloaca in an early developmental stage [5]. Half of the pat...

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... 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplication 1 malrotation Ponson (2001) [14] 3 3 F Post 3 tub 1 asympt2 mild 23m (10m–4 y) 3 none Hamada (1996) [5] 2 2 F Post 2 tub 1 asympt1 mild 3.5 y (7m–6 y) 1 cleft lip 1 none Tagart (1977) [15] 4 1 F3M 3 right side 1 post 3 tub 1 cyst 4 complication 29 y (11m–45 y) None Aaronson (1970) [16] 1 1 F Post 1 tub 1 asympt 3m 1 anterior sacral meningocoele,covered anus Total group numbers 55 49 F5M 52 post 3 right side 48 tub 1 combined 6 cyst 26 asympt 18 mild 11 complication 4.6 y (0–45 y) 20 associated anomalies Total group percentage % 89%F 11%M 94.5% post 5.5% right side 87% tub 2% combined 11% cyst 47% asympt 33% mild 20% complications 36% associated anomalies Overview of the reported cases in English literature (first author and year of publication between brackets) with the number (No.) of reported cases, the localization (post: posterior), type of lesion (tub: tubular, cyst: c...

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...omplications: inflammation and malignancy. Nevertheless they refused surgical mucosal stripping. 2. Discussion ACD is the least frequent digestive duplication. Clinically, it presents itself as an extra perineal orifice located just behind the anus. Clinically, it is difficult to differentiate ACD from a rectal or anal fistula, however, in noncomplicated ACD inflammation will be absent. Only histology gives diagnostic certainty describing 3 characteristics of ACD: squamous epithelium in the caudal end, transitional epithelium in the cranial end and smooth-muscle cells in the wall of the canal [1, 2]. It is most frequently a tubular (90%) anomaly without communication to the rectum. In 10% of cases, the lesion is cystic [3]. We found only 55 patients (including our patient) withACD inEnglish literature (Table 1). Females comprise up to 89% of the patients with ACD (Table 1). Twohypotheses concerning the origin of anal canal duplication are suggested in literature. Choi and Park postulate it as a consequence of recanalization of a cloacal membrane excess in late embryonic life [4]. Hamada et al. suggest a duplication of the dorsal cloaca in an early developmental stage [5]. Half of the pat...

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...requent digestive duplication. Clinically, it presents itself as an extra perineal orifice located just behind the anus. Clinically, it is difficult to differentiate ACD from a rectal or anal fistula, however, in noncomplicated ACD inflammation will be absent. Only histology gives diagnostic certainty describing 3 characteristics of ACD: squamous epithelium in the caudal end, transitional epithelium in the cranial end and smooth-muscle cells in the wall of the canal [1, 2]. It is most frequently a tubular (90%) anomaly without communication to the rectum. In 10% of cases, the lesion is cystic [3]. We found only 55 patients (including our patient) withACD inEnglish literature (Table 1). Females comprise up to 89% of the patients with ACD (Table 1). Twohypotheses concerning the origin of anal canal duplication are suggested in literature. Choi and Park postulate it as a consequence of recanalization of a cloacal membrane excess in late embryonic life [4]. Hamada et al. suggest a duplication of the dorsal cloaca in an early developmental stage [5]. Half of the patients with ACD are asymptomatic. Parents or caregivers notice a perianal orifice posterior to the anus. Mild symptoms such as ...

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...the caudal end, transitional epithelium in the cranial end and smooth-muscle cells in the wall of the canal [1, 2]. It is most frequently a tubular (90%) anomaly without communication to the rectum. In 10% of cases, the lesion is cystic [3]. We found only 55 patients (including our patient) withACD inEnglish literature (Table 1). Females comprise up to 89% of the patients with ACD (Table 1). Twohypotheses concerning the origin of anal canal duplication are suggested in literature. Choi and Park postulate it as a consequence of recanalization of a cloacal membrane excess in late embryonic life [4]. Hamada et al. suggest a duplication of the dorsal cloaca in an early developmental stage [5]. Half of the patients with ACD are asymptomatic. Parents or caregivers notice a perianal orifice posterior to the anus. Mild symptoms such as anal pain, pruritus,mucous discharge and constipation are present in one third of the patients. Perineal abscess or inflammation can, however, be the presenting complication of ACD. Although ACD is present at birth, it can easily be overlooked resulting in a widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with com...

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... of the canal [1, 2]. It is most frequently a tubular (90%) anomaly without communication to the rectum. In 10% of cases, the lesion is cystic [3]. We found only 55 patients (including our patient) withACD inEnglish literature (Table 1). Females comprise up to 89% of the patients with ACD (Table 1). Twohypotheses concerning the origin of anal canal duplication are suggested in literature. Choi and Park postulate it as a consequence of recanalization of a cloacal membrane excess in late embryonic life [4]. Hamada et al. suggest a duplication of the dorsal cloaca in an early developmental stage [5]. Half of the patients with ACD are asymptomatic. Parents or caregivers notice a perianal orifice posterior to the anus. Mild symptoms such as anal pain, pruritus,mucous discharge and constipation are present in one third of the patients. Perineal abscess or inflammation can, however, be the presenting complication of ACD. Although ACD is present at birth, it can easily be overlooked resulting in a widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with complications [3]. In the reported cases, there 2 Case Reports in Gastrointestinal Medicine Table...

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...y), and number and type of associated anomalies. is a significant age difference according to the symptom severity (...

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...n one third of the patients. Perineal abscess or inflammation can, however, be the presenting complication of ACD. Although ACD is present at birth, it can easily be overlooked resulting in a widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with complications [3]. In the reported cases, there 2 Case Reports in Gastrointestinal Medicine Table 1: Summary of all reported anal canal duplication cases in English literature. Reference No. Sex Localization Type Symptoms Age Associated anomalies Our patient 1 1 F Post 1 tub 1 mild 11 y None Sinnya (2012) [7] 1 1 F Post 1 tub 1 complication 15 y Dysplastic coccyx Lippert (2012) [8] 1 1 F Post 1 cyst 1 complication 12 y None Narci (2010) [9] 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complicat...

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..., be the presenting complication of ACD. Although ACD is present at birth, it can easily be overlooked resulting in a widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with complications [3]. In the reported cases, there 2 Case Reports in Gastrointestinal Medicine Table 1: Summary of all reported anal canal duplication cases in English literature. Reference No. Sex Localization Type Symptoms Age Associated anomalies Our patient 1 1 F Post 1 tub 1 mild 11 y None Sinnya (2012) [7] 1 1 F Post 1 tub 1 complication 15 y Dysplastic coccyx Lippert (2012) [8] 1 1 F Post 1 cyst 1 complication 12 y None Narci (2010) [9] 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omph...

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...sent at birth, it can easily be overlooked resulting in a widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with complications [3]. In the reported cases, there 2 Case Reports in Gastrointestinal Medicine Table 1: Summary of all reported anal canal duplication cases in English literature. Reference No. Sex Localization Type Symptoms Age Associated anomalies Our patient 1 1 F Post 1 tub 1 mild 11 y None Sinnya (2012) [7] 1 1 F Post 1 tub 1 complication 15 y Dysplastic coccyx Lippert (2012) [8] 1 1 F Post 1 cyst 1 complication 12 y None Narci (2010) [9] 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) ...

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...widely varying age at presentation (Table 1). Diagnosis at a later age is more often associated with complications [3]. In the reported cases, there 2 Case Reports in Gastrointestinal Medicine Table 1: Summary of all reported anal canal duplication cases in English literature. Reference No. Sex Localization Type Symptoms Age Associated anomalies Our patient 1 1 F Post 1 tub 1 mild 11 y None Sinnya (2012) [7] 1 1 F Post 1 tub 1 complication 15 y Dysplastic coccyx Lippert (2012) [8] 1 1 F Post 1 cyst 1 complication 12 y None Narci (2010) [9] 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 i...

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... Sex Localization Type Symptoms Age Associated anomalies Our patient 1 1 F Post 1 tub 1 mild 11 y None Sinnya (2012) [7] 1 1 F Post 1 tub 1 complication 15 y Dysplastic coccyx Lippert (2012) [8] 1 1 F Post 1 cyst 1 complication 12 y None Narci (2010) [9] 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplicat...

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... 2 2 F Post 2 tub 2 asympt 5 y(1–9 y) None Koga (2010) [10] 10 10 F Post 10 tub 3 asympt 6 mild 1 complication 6m (24 d–4 y) 1 hypoplastic kidney 1 teratoma and thetered cord 2 anal stenosis Carpentier (2009) [2] 2 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplication 1 malrotation Ponson (2001) [14] 3 3 F Post 3 tub 1 asympt2 mild 23m (10m–4 y) 3 none Hamada (1996) [5] 2 2 F Post 2 tub 1 asympt1 mild 3.5 y (7m–6 y) 1 cleft lip 1 none Tagart (1977) [15] 4 1 F3M 3 right side 1 post 3 tub 1 cyst 4 complication 29 y ...

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... 1 F1M Post 1 tub 1 cyst 1 mild 1 complication 2.5m (2-3m) 1 spina bifida occulta, thetered cord, hydronefrosis 1 none Kratz (2008) [11] 1 1 F Post 1 cyst 1 complication 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplication 1 malrotation Ponson (2001) [14] 3 3 F Post 3 tub 1 asympt2 mild 23m (10m–4 y) 3 none Hamada (1996) [5] 2 2 F Post 2 tub 1 asympt1 mild 3.5 y (7m–6 y) 1 cleft lip 1 none Tagart (1977) [15] 4 1 F3M 3 right side 1 post 3 tub 1 cyst 4 complication 29 y (11m–45 y) None Aaronson (1970) [16] 1 1 F Post 1 tub 1 asympt 3m 1 anterior sacral meningocoele,covered anus Total group numbers 55 49 F5M 52 post 3 right side 48 tub 1 combined 6 cyst 26 asympt 18 mild 11 complic...

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...n 16 y None Lisi (2006) [3] 12 11 F1M Post 10 tub 1 cyst 6 asympt 4 mild 2 complication 17.8m (0–60m) 1 anorectal malformation 1 cleft lip, cleft palate, omphalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplication 1 malrotation Ponson (2001) [14] 3 3 F Post 3 tub 1 asympt2 mild 23m (10m–4 y) 3 none Hamada (1996) [5] 2 2 F Post 2 tub 1 asympt1 mild 3.5 y (7m–6 y) 1 cleft lip 1 none Tagart (1977) [15] 4 1 F3M 3 right side 1 post 3 tub 1 cyst 4 complication 29 y (11m–45 y) None Aaronson (1970) [16] 1 1 F Post 1 tub 1 asympt 3m 1 anterior sacral meningocoele,covered anus Total group numbers 55 49 F5M 52 post 3 right side 48 tub 1 combined 6 cyst 26 asympt 18 mild 11 complication 4.6 y (0–45 y) 20 associated anomalies Total group percentage % 89%F 11%M 94.5% post 5.5% right side 87% tub 2% combined 11% cyst 47% asympt 33% mild 20% complica...

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...phalocoele 1 presacral ependymoma 2 teratoma’s Tiryaki (2006) [12] 2 2 F Post 2 tub 1 asympt1 mild 7 y (7–7 y) 1 none 1 intrasacral meningocele Choi (2003) [4] 6 6 F Post 6 tub 6 asympt 4.5m(3–9m) 6 none Ochiai (2002) [1] 1 1 F Post 1 combined 1 mild 6 y None Jacquier (2001) [13] 6 6 F Post 6 tub 5 asympt1 mild 2.5m (0m–12 y) 1 sacral teratoma, lumbosacral meningomyelocoele 1 sacral teratoma 1 uteric duplication 1 malrotation Ponson (2001) [14] 3 3 F Post 3 tub 1 asympt2 mild 23m (10m–4 y) 3 none Hamada (1996) [5] 2 2 F Post 2 tub 1 asympt1 mild 3.5 y (7m–6 y) 1 cleft lip 1 none Tagart (1977) [15] 4 1 F3M 3 right side 1 post 3 tub 1 cyst 4 complication 29 y (11m–45 y) None Aaronson (1970) [16] 1 1 F Post 1 tub 1 asympt 3m 1 anterior sacral meningocoele,covered anus Total group numbers 55 49 F5M 52 post 3 right side 48 tub 1 combined 6 cyst 26 asympt 18 mild 11 complication 4.6 y (0–45 y) 20 associated anomalies Total group percentage % 89%F 11%M 94.5% post 5.5% right side 87% tub 2% combined 11% cyst 47% asympt 33% mild 20% complications 36% associated anomalies Overview of the reported cases in English literature (first author and year of publication between brackets) with the number ...