Download PDF
Eur J Pediatr Surg 2024; 34(05): 418-422
DOI: 10.1055/s-0043-1777101
Original Article

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

Richard Skaba
1   Department of Paediatric Surgery, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
,
Vojtech Dotlacil
1   Department of Paediatric Surgery, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
,
Pavla Fuccillo
2   Department of Pathology and Molecular Medicine, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
,
Blanka Rouskova
1   Department of Paediatric Surgery, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
,
Lucie Pos
1   Department of Paediatric Surgery, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
,
Michal Rygl
1   Department of Paediatric Surgery, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic
› Author Affiliations
› Further Information
Also available at
    Permissions and Reprints

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.




Publication History

Received: 22 February 2023

Accepted: 21 September 2023

Article published online:
28 November 2023

© 2023. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Peña A, Hong A. Advances in the management of anorectal malformations. Am J Surg 2000; 180 (05) 370-376
    CrossrefPubMedSearch in Google Scholar
  • 2 Rintala RJ. Fecal incontinence in anorectal malformations, neuropathy, and miscellaneous conditions. Semin Pediatr Surg 2002; 11 (02) 75-82
    CrossrefPubMedSearch in Google Scholar
  • 3 Levitt M, Peña A. Update on pediatric faecal incontinence. Eur J Pediatr Surg 2009; 19 (01) 1-9
    Thieme ConnectPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 6 Rintala RJ, Pakarinen MP. Imperforate anus: long- and short-term outcome. Semin Pediatr Surg 2008; 17 (02) 79-89
    CrossrefPubMedSearch in Google Scholar
  • 7 Divarci E, Ergun O. General complications after surgery for anorectal malformations. Pediatr Surg Int 2020; 36 (04) 431-445
    CrossrefPubMedSearch in Google Scholar
  • 8 Levitt MA, Kant A, Peña A. The morbidity of constipation in patients with anorectal malformations. J Pediatr Surg 2010; 45 (06) 1228-1233
    CrossrefPubMedSearch in Google Scholar
  • 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
    Thieme ConnectPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 14 Meier-Ruge WA, Holschneider AM. Histopathologic observations of anorectal abnormalities in anal atresia. Pediatr Surg Int 2000; 16 (1–2): 2-7
    CrossrefPubMedSearch in Google Scholar
  • 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
    Thieme ConnectPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    PubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 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
    Thieme ConnectPubMedSearch in Google Scholar
  • 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
    Thieme ConnectPubMedSearch in Google Scholar
  • 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
    PubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar
  • 24 Bischoff A, Bealer J, Peña A. Critical analysis of fecal incontinence scores. Pediatr Surg Int 2016; 32 (08) 737-741
    CrossrefPubMedSearch in Google Scholar
  • 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
    CrossrefPubMedSearch in Google Scholar