Perioperative Histologically Controlled Fistula Resection in Patients with Imperforate Anus and Perineal Fistula

Abstract

Introduction Postoperative constipation (PC) in patients with imperforate anus and perineal fistula (PF) has been reported in up to 60%. Histological studies of PF revealed innervation anomalies which seem to be one of the reasons for PC. Perioperative histologically controlled fistula resection (PHCFR) allows appropriate resection of PF and pull-down normoganglionic rectum at the time of posterior sagittal anorectoplasty (PSARP).

Materials and Methods A total of 665 patients with anorectal malformations underwent surgery between 1991 and 2021. Of these, 364 presented PF; 92 out of them (41 F) were studied. Patients with sacral and spinal cord anomalies, neurological disorders, and cut-back anoplasty were excluded. PSARP was done on all patients. Hematoxylin-eosin staining and NADH Tetrazolium-reductase histochemical method were used. Four and more ganglion cells in the myenteric plexus represented a sufficient length of the resection. The continence was scored according to the modified Krickenbeck scoring system. Final scores ranged from 1 to 7 points. Values are given as median.

Results A total of 65 (70.7%) patients presented an aganglionic segment in PF, and 27 patients presented hypoganglionosis. The median length of the resected fistula was 25 mm (interquartile range [IQR]: 20–30). The median total continence score was 7 (IQR: 6–7). Post-op constipation was observed in 6/92 (6.5%) patients.

Conclusion PHCFR diminished PC to 6.5% of patients.

Keywords

anorectal malformations - perineal fistula - posterior sagittal anorectoplasty - perioperative biopsy - chronic constipation - follow-up

Ethical Approval

This study received approval from the institutional ethical committee under No: EK – 646/22.

Publikationsverlauf

Eingereicht: 22. Februar 2023

Angenommen: 21. September 2023

Artikel online veröffentlicht:
28. November 2023

• References

• 1 Peña A, Hong A. Advances in the management of anorectal malformations. Am J Surg 2000; 180 (05) 370-376
• 2 Rintala RJ. Fecal incontinence in anorectal malformations, neuropathy, and miscellaneous conditions. Semin Pediatr Surg 2002; 11 (02) 75-82
• 3 Levitt M, Peña A. Update on pediatric faecal incontinence. Eur J Pediatr Surg 2009; 19 (01) 1-9
• 4 Kyrklund K, Pakarinen MP, Koivusalo A, Rintala RJ. Long-term bowel functional outcomes in rectourethral fistula treated with PSARP: controlled results after 4-29 years of follow-up: a single-institution, cross-sectional study. J Pediatr Surg 2014; 49 (11) 1635-1642
• 5 Santos-Jasso KA, Arredondo-García JL, Maza-Vallejos J, Lezama-Del Valle P. Effectiveness of senna vs polyethylene glycol as laxative therapy in children with constipation related to anorectal malformation. J Pediatr Surg 2017; 52 (01) 84-88
• 6 Rintala RJ, Pakarinen MP. Imperforate anus: long- and short-term outcome. Semin Pediatr Surg 2008; 17 (02) 79-89
• 7 Divarci E, Ergun O. General complications after surgery for anorectal malformations. Pediatr Surg Int 2020; 36 (04) 431-445
• 8 Levitt MA, Kant A, Peña A. The morbidity of constipation in patients with anorectal malformations. J Pediatr Surg 2010; 45 (06) 1228-1233
• 9 Holschneider AM, Pfrommer W, Gerresheim B. Results in the treatment of anorectal malformations with special regard to the histology of the rectal pouch. Eur J Pediatr Surg 1994; 4 (05) 303-309
• 10 Huang CF, Lee HC, Yeung CY. et al. Constipation is a major complication after posterior sagittal anorectoplasty for anorectal malformations in children. Pediatr Neonatol 2012; 53 (04) 252-256
• 11 Shandilya G, Pandey A, Pant N, Singh G, Kumar A, Rawat J. Evaluation and management of “low” anorectal malformation in male children: an observational study. Pediatr Surg Int 2022; 38 (02) 337-343
• 12 Chang PC, Duh YC, Fu YW, Hsu YJ, Wei CH, Huang H. How much do we know about constipation after surgery for anorectal malformation?. Pediatr Neonatol 2020; 61 (01) 58-62
• 13 Holschneider AM, Ure BM, Pfrommer W, Meier-Ruge W. Innervation patterns of the rectal pouch and fistula in anorectal malformations: a preliminary report. J Pediatr Surg 1996; 31 (03) 357-362
• 14 Meier-Ruge WA, Holschneider AM. Histopathologic observations of anorectal abnormalities in anal atresia. Pediatr Surg Int 2000; 16 (1–2): 2-7
• 15 Holschneider AM, Koebke J, Meier-Ruge W, Land N, Jesch NK, Pfrommer W. Pathophysiology of chronic constipation in anorectal malformations. Long-term results and preliminary anatomical investigations. Eur J Pediatr Surg 2001; 11 (05) 305-310
• 16 Lombardi L, Bruder E, Caravaggi F, Del Rossi C, Martucciello G. Abnormalities in “low” anorectal malformations (ARMs) and functional results resecting the distal 3 cm. J Pediatr Surg 2013; 48 (06) 1294-1300
• 17 Tiwari A, Naik DC, Khanwalkar PG, Sutrakar SK. Histological study of neonatal bowel in anorectal malformations. Int J Anat Res 2014; 2: 318-324
• 18 Xiao H, Huang R, Cui DX, Xiao P, Diao M, Li L. Histopathologic and immunohistochemical findings in congenital anorectal malformations. Medicine (Baltimore) 2018; 97 (31) e11675
• 19 Brisighelli G, Macchini F, Consonni D, Di Cesare A, Morandi A, Leva E. Continence after posterior sagittal anorectoplasty for anorectal malformations: comparison of different scores. J Pediatr Surg 2018; 53 (09) 1727-1733
• 20 Dudorkinová D, Skába R, Lojda Z, Dubovska M. Application of NADH tetrazolium reductase reaction in perioperative biopsy of dysganglionic large bowel. Eur J Pediatr Surg 1994; 4 (06) 362-365
• 21 Martucciello G, Favre A, Torre M, Pini Prato A, Jasonni V. A new rapid acetylcholinesterase histochemical method for the intraoperative diagnosis of Hirschsprung's disease and intestinal neuronal dysplasia. Eur J Pediatr Surg 2001; 11 (05) 300-304
• 22 Beschorner R, Mittelbronn M, Bekure K, Meyermann R. Problems in fast intraoperative diagnosis in Hirschsprung's disease. Folia Neuropathol 2004; 42 (04) 191-195
• 23 Shayan K, Smith C, Langer JC. Reliability of intraoperative frozen sections in the management of Hirschsprung's disease. J Pediatr Surg 2004; 39 (09) 1345-1348
• 24 Bischoff A, Bealer J, Peña A. Critical analysis of fecal incontinence scores. Pediatr Surg Int 2016; 32 (08) 737-741
• 25 Pakarinen MP, Koivusalo A, Lindahl H, Rintala RJ. Prospective controlled long-term follow-up for functional outcome after anoplasty in boys with perineal fistula. J Pediatr Gastroenterol Nutr 2007; 44 (04) 436-439