Huang Yanbing,He Yuanbin,Zhuang Xiaojin,et al.Anal canal duplication in children: a report of two cases with a literature review[J].Journal of Clinical Pediatric Surgery,2024,(02):184-189.[doi:10.3760/cma.j.cn101785-202211022-016]
儿童肛管重复畸形2例并文献复习
- Title:
- Anal canal duplication in children: a report of two cases with a literature review
- Keywords:
- Anal Canal; Abnormalities; Congenital Abnormalities; Diagnosis; Therapy; Child
- 摘要:
- 目的 探讨儿童肛管重复畸形的临床特征及治疗方法。方法 回顾性分析福建省儿童医院2例肛管重复畸形(anal canal duplication,ACD)患儿临床资料;同时检索中国知网、万方医学网、PubMed、Medline、Web of Science 1992—2022年相关文献,中英文检索词分别为:"肛管/肛门重复畸形"、"anal canal duplication"。收集本院以及符合纳排标准文献中ACD患儿一般资料(性别、诊断年龄)、临床特征(开口位置、类型、长度、与肛管直肠之间的关系、临床症状、合并畸形)、治疗方案及随访情况等进行总结分析。结果 本院2例于新生儿期发现正常肛门正后方异常开口,无肛周感染病史及不适,影像学检查未发现其他合并畸形。异常开口造影提示2例均为管状结构,1例管腔近端与直肠相通,均采用后矢状入路小切口ACD剥离术,术后病理结果显示ACD与正常肛管具有类似病理学特征,术后2个月随访肛门外观及功能恢复良好。文件检索共获得符合要求文献23篇,共74例ACD患儿,包括本院2例在内共76例,男女比例为1∶7.4;诊断年龄3.5(0~16)岁;71例(71/76,93%)ACD位于正常肛门截石位6点方向;91%(69/76)为管状型,9%(7/76)为囊肿型;约87%(66/76)的患儿(平均年龄2.8岁)无临床症状或仅有轻度症状,约13%(10/76)的患儿(平均年龄为7.7岁)出现严重并发症,主要表现为骶前感染;约33%(25/76)的患儿合并其他异常,以骶前包块最常见(11/25,44%)。70例予手术切除ACD(其中34例经会阴入路,36例取后矢状入路),术后并发症发生率约为4% (3/70)。76例ACD患儿平均随访时间3.6年,肛门功能均正常。结论 ACD多见于女性,当发现肛门正后方异常开口时应考虑ACD,予进一步影像学检查以排除其他合并异常。一旦诊断ACD应早期手术治疗,建议采取后矢状入路ACD剥离术,手术安全,疗效良好。
- Abstract:
- Objective To explore the clinical characteristics and treatments of anal canal duplication (ACD) in children.Methods The relevant clinical data were retrospectively reviewed for two ACD children.More related cases were retrieved from the relevant Chinese and English literature reports of the databases of PubMed,Medline,Web of Science,CNKI and Wanfang from 1993 to 2022.Results Two neonates presented with sagittal anal orifice discovered by their mothers.There were no other associated anomalies.Fistulography revealed a tubular structure in two cases and proximal end of lumen communicated with rectum in case 2.Dissection was performed via a posterior sagittal approach.Histological examination hinted at squamous epithelium with smooth muscle bundles.Both cases achieved excellent cosmetics with normal sphincter control.A total of 76 cases of ACD have been reported.There were 9 boys and 67 girls with a mean diagnostic age of 3.5(0-16) year; 93%(71/76) of lesion were located at 6 o’clock behind anus; 91%(69/76) was tubular and 9%(69/76) cystic.And 87%(66/76) were asymptomatic or mildly symptomatic (a mean age of 2.8 year); 13%(10/76) developed presacral abscesses (a mean age of 7.7 year); 33%(25/76) presented associated anomalies,including presacral mass (44%); 70 children underwent surgical removal (perineal approach,n=34; posterior sagittal approach,n=36) and postoperative complications occurred (4%,3/70).Bowel function normalized during a mean follow-up period of 3.6 years (1 month to 19 years).Conclusions A clinical diagnosis of ACD should be suspected for an opening in posterior anal midline,particularly in girls.Associated anomalies should be ruled out.Early surgery is recommended.ACD removal via a posterior sagittal approach is both safe and effective.
参考文献/References:
[1] Ailhaud A,Alao O,Sole Cruz E,et al.Anal canal duplication in children:a monocentric experience of 12 cases[J].Pediatr Surg Int,2021,37(8):1007-1012.DOI:10.1007/s00383-021-04910-5.
[2] Trecartin AC,Pe?a A,Lovell M,et al.Anal duplication:is surgery indicated? A report of three cases and review of the literature[J].Pediatr Surg Int,2019,35(9):971-978.DOI:10.1007/s00383-019-04509-x.
[3] Connor SJ,Brisighelli G,Patel N,et al.Clinical quiz-a rare case of anal canal duplication in the context of currarino syndrome[J].European J Pediatr Surg Rep,2021,9(1):e68-e71.DOI:10.1055/s-0041-1735595.
[4] Van Biervliet S,Maris E,Vande Velde S,et al.Anal canal duplication in an 11-year-old-child[J].Case Rep Gastrointest Med,2013,2013:503691.DOI:10.1155/2013/503691.
[5] ?zbey H.Anal canal duplication in a 12-year-old girl[J].J Pediatr Gastroenterol Nutr,2021,72(1):e21.DOI:10.1097/MPG.0000000000002850.
[6] Li DD,Liu SB,Feng JX,et al.Anal canal duplication mimicking recurrent abscess:a case report and review of the literature[J].Front Surg,2022,9:908390.DOI:10.3389/fsurg.2022.908390.
[7] Honda S,Minato M,Miyagi H,et al.Anal canal duplication presenting with abscess formation[J].Pediatr Int,2017,59(4):500-501.DOI:10.1111/ped.13222.
[8] Dukes CE,Galvin C.Colloid carcinoma arising within fistulae in the anorectal region[J].Ann R Coll Surg Engl,1956,18(4):246-261.
[9] Palazon P,Julia V,Saura L,et al.Anal canal duplication and triplication:a rare entity with different presentations[J].Pediatr Surg Int,2017,33(5):609-617.DOI:10.1007/s00383-017-4074-7.
[10] Akova F,Altinay S,Aydin E.The controversy of surgical intervention for anal canal duplication in children[J].Pak J Med Sci,2020,36(6):1330-1333.DOI:10.12669/pjms.36.6.1832.
[11] Hamada Y,Sato M,Hioki K.Anal canal duplication in childhood[J].Pediatr Surg Int,1996,11(8):577-579.DOI:10.1007/BF00626072.
[12] Ochiai K,Umeda T,Murahashi O,et al.Anal-canal duplication in a 6-year-old child[J].Pediatr Surg Int,2002,18(2/3):195-197.DOI:10.1007/s003830100691.
[13] Kratz JR,Deshpande V,Ryan DP,et al.Anal canal duplication associated with presacral cyst[J].J Pediatr Surg,2008,43(9):1749-1752.DOI:10.1016/j.jpedsurg.2008.05.026.
[14] Choi SO,Park WH.Anal canal duplication in infants[J].J Pediatr Surg,2003,38(5):758-762.DOI:10.1016/jpsu.2003.50161.
[15] Mateescu T,Tarta C,Stanciu P,et al.Anal canal duplication in an adult female-case report and pathology guiding[J].Medicina (Kaunas),2021,57(11):1205.DOI:10.3390/medicina57111205.
[16] van der Putte SCJ.Normal and abnormal development of the anorectum[J].J Pediatr Surg,1986,21(5):434-440.DOI:10.1016/S0022-3468(86)80515-2.
[17] Nievelstein RAJ,van der Werff JFA,Verbeek FJ,et al.Normal and abnormal embryonic development of the anorectum in human embryos[J].Teratology,1998,57(2):70-78.DOI:10.1002/(SICI)1096-9926(199802)57:2<70::AID-TERA5>3.0.CO;2-A.
[18] Koga H,Okazaki T,Kato Y,et al.Anal canal duplication:experience at a single institution and literature review[J].Pediatr Surg Int,2010,26(10):985-988.DOI:10.1007/s00383-010-2653-y.
[19] Narc? A,Dilek FH,?etinkur?un S.Anal canal duplication[J].Eur J Pediatr,2010,169(5):633-635.DOI:10.1007/s00431-009-1094-x.
[20] Lisi G,Illiceto MT,Rossi C,et al.Anal canal duplication:a retrospective analysis of 12 cases from two European pediatric surgical departments[J].Pediatr Surg Int,2006,22(12):967-973.DOI:10.1007/s00383-006-1801-x.
[21] Lippert SJ,Hartin CWJr,Ozgediz DE.Communicating anal canal duplication cyst in an adolescent girl[J].Colorectal Dis,2012,14(5):e270-e271.DOI:10.1111/j.1463-1318.2011.02870.x.
[22] Nakata M,Mitsunaga T,Terui E,et al.Recurrence of anal canal duplication with abscess formation[J].J Pediatr Surg Case Rep,2018,33:68-71.DOI:10.1016/j.epsc.2018.04.007.
[23] Mirzaei R,Mahjubi B,Alvandipoor M,et al.Late presentation of anal canal duplication in adults:a series of four rare cases[J].Ann Coloproctol,2015,31(1):34-36.DOI:10.3393/ac.2015.31.1.34.
[24] Karamatzanis I,Kosmidou P,Harmanis S,et al.Early diagnosis of anal canal duplication:the importance of a physical examination[J].Cureus,2022,14(5):e25040.DOI:10.7759/cureus.25040.
[25] 陈希琳,冯六泉,姜国丹,等.肛瘘的诊治专家共识(2020版)[J].实用临床医药杂志,2020,24(17):1-7.DOI:10.7619/jcmp.202017001.Chen XL,Feng LQ,Jiang GD,et al.Expert Consensus on Diagnosing and Treating Anal Fistula (2020 Edition)[J].J Clin Med Pract,2020,24(17):1-7.DOI:10.7619/jcmp.202017001.
[26] Nosek M,Golonka A,Kalińska-Lipert A,et al.Rectal duplication with sciatic hernia[J].Wideochir Inne Tech Malo Inwazyjne,2015,10(2):282-285.DOI:10.5114/wiitm.2015.52708.
[27] 孙琳,王燕霞,刘予.小儿直肠重复畸形(附7例报告)[J].中国普外基础与临床杂志,1996,3(4):214-216.Sun L,Wang YX,Liu Y.Rectal duplications in children:a report of 7 cases[J].Chin J Bas Clin Gen Surg,1996,3(4):214-216.
[28] Tiryaki T,Senel E,Atayurt H.Anal canal duplication in children:a new technique[J].Pediatr Surg Int,2006,22(6):560-561.DOI:10.1007/s00383-006-1654-3.
[29] Jacquier C,Dobremez E,Piolat C,et al.Anal canal duplication in infants and children-a series of 6 cases[J].Eur J Pediatr Surg,2001,11(3):186-191.DOI:10.1055/s-2001-15482.
[30] Sinnya S,Curtis K,Walsh M,et al.Late presentation of anal canal duplication in an adolescent female:a rare diagnosis[J].Int J Colorectal Dis,2013,28(8):1175-1176.DOI:10.1007/s00384-012-1608-2.
[31] Carpentier H,Maizlin I,Bliss D.Anal canal duplication:case reviews and summary of the world literature[J].Pediatr Surg Int,2009,25(10):911-916.DOI:10.1007/s00383-009-2474-z.
[32] 高群,黄河,潘祝彬,等.肛门重复畸形一例[J].中华小儿外科杂志,2010,31(5):403-404.DOI:10.3760/cma.j.issn.0253-3006.2010.05.027.Gao Q,Huang H,Pan ZB,et al.Anal canal duplication:one case report[J].Chin J Pediatr Surg,2010,31(5):403-404.DOI:10.3760/cma.j.issn.0253-3006.2010.05.027.
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备注/Memo
收稿日期:2022-11-11。
通讯作者:林宇,Email:171433750@qq.com