|Year : 2017 | Volume
| Issue : 2 | Page : 83-86
Anorectal malformations in males: Pros and cons of neonatal versus staged reconstruction for high and intermediate varieties
Prema Menon, Katragadda Lakshmi Narasimha Rao, Amit Kumar Sinha, K Lokesha, Ram Samujh, Jai Kumar Mahajan, Ravi Prakash Kanojia, Monika Bawa
Department of Pediatric Surgery, Advanced Pediatrics Centre, Postgraduate Institute of Medical Education and Research, Chandigarh, India
|Date of Web Publication||22-Mar-2017|
Katragadda Lakshmi Narasimha Rao
Department of Pediatric Surgery, Advanced Pediatrics Centre, Post Graduate Institute of Medical Education and Research, Chandigarh - 160 012
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Background: High and intermediate types of anorectal malformations (ARMs) in male neonates may be managed either by primary neonatal reconstruction without colostomy cover or by traditional policy of staged reconstruction after neonatal colostomy. Posterior sagittal anorectoplasty (PSARP) is the current widely practiced reconstructive technique with varied results.
Aim: To assess our functional results of PSARP without colostomy in male neonates with high and intermediate ARMs compared to 3-stage (neonatal colostomy – PSARP – colostomy closure) methodology in a high volume tertiary care institution of a developing country.
Patients and Methods: The number of colostomies performed for male high/intermediate anomalies and the number of babies who completed 3-stage reconstruction during a 10-year period is analyzed. The outcome of primary neonatal PSARPs during the same period was analyzed. Eighty primary PSARPs were compared to 81 staged reconstructions for outcome analysis, using Kelly score.
Results: A total of 453 colostomies were performed, but only 253 of them completed all stages of reconstruction (52%). Good continence was achieved in 45% of cases of primary PSARP versus 26% in 3-staged surgery.
Conclusions: Primary PSARP in neonatal period without colostomy is a good option for high and intermediate ARMs in males if the treating surgeon is reasonably skilled in neonatal surgery and PSARP procedures.
Keywords: Anorectal malformations, colostomy, fecal continence, posterior sagittal anorectoplasty
|How to cite this article:|
Menon P, Rao KL, Sinha AK, Lokesha K, Samujh R, Mahajan JK, Kanojia RP, Bawa M. Anorectal malformations in males: Pros and cons of neonatal versus staged reconstruction for high and intermediate varieties. J Indian Assoc Pediatr Surg 2017;22:83-6
|How to cite this URL:|
Menon P, Rao KL, Sinha AK, Lokesha K, Samujh R, Mahajan JK, Kanojia RP, Bawa M. Anorectal malformations in males: Pros and cons of neonatal versus staged reconstruction for high and intermediate varieties. J Indian Assoc Pediatr Surg [serial online] 2017 [cited 2023 Nov 30];22:83-6. Available from: https://www.jiaps.com/text.asp?2017/22/2/83/202675
| Introduction|| |
Anorectal malformations (ARMs) are one of the most common conditions in pediatric surgical practice. A lot has been written about the techniques of management and currently Pena's posterior sagittal anorectoplasty (PSARP) occupies the center stage., There are many scoring systems which are in vogue to measure the continence results and quality of life scores.,,,,,,,, The anomalies may be treated by conventional three stages  (neonatal colostomy – PSARP or abdominoperineal pull-through [APPT] – colostomy closure) or accomplished by reconstruction in neonatal period without colostomy.,,,,,,,,, There are many proponents of single stage reconstruction in the literature in female anomalies also., The aim of this study is to present a large series of single stage reconstruction in the neonatal period without colostomy and compare the results with 3-stage reconstruction performed during the same period in the same circumstances, in male ARMs.
| Patients and Methods|| |
The number of neonatal high divided sigmoid colostomies for male high/intermediate ARMs performed in our department over a 10-year period from 2006 to 2016 was counted. The number of patients completing subsequent 2 stages of management (PSARP or APPT and colostomy closure) during the same period was documented. The neonates who underwent primary PSARP without colostomy cover after initial cross table prone lateral (CTPL) radiograph  were analyzed during this period (Group A). It is the senior authors' practice to perform primary PSARP in all neonates whose CTPL radiograph shows air in the presacral region. Survival figures and functional results (Kelly score) in this group were compared with all babies who underwent 3-stage reconstruction by 2 consultant surgeons (PM, KLNR) (Group B). Continence achieved in different locations of recto-urinary communication was analyzed. Three years follow up for neonatal PSARP and 2 years follow up after the completion of all 3 stages was accepted as minimum requirement for assessing continence. Statistical significance for continence results between two groups was calculated using Chi-square test (Pearson value) with a P< 0.05 taken as significant. Congenital pouch colons , and low ARMs referred after initial colostomy were excluded from this study.
| Results|| |
A total of 453 colostomies were performed for high and intermediate variety of ARMs in males in our department during the study period [Table 1]. However, all of them did not translate into definitive reconstruction with only 253 (52%) patients completing all the stages of definitive surgery. Group A comprised 80 neonates and Group B had 81 babies. [Table 2] outlines continence scores in both groups. Significant “good” scores were achieved in primary PSARP group. The location of the recto-urinary tract fistula did not affect the continence rates significantly [Table 3].
|Table 1: Number of colostomies versus posterior sagittal anorectoplasty/abdominoperineal pull-throughs performed for high and intermediate anorectal malformations in males|
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|Table 2: Anorectal malformation: Comparison of fecal continence in primary versus staged surgery groups|
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| Discussion|| |
Despite considerable interest of surgeons, the ultimate results of high and intermediate ARMs as measured by continence results are not completely satisfactory. The reported continence rates vary from 2% to 84% (!).,,,, Neonatal high divided sigmoid colostomy in a distended abdomen of a sick baby is not devoid of complications and various authors have confirmed this.,, In developing countries, especially patients from low economic strata, the conventional policy of colostomy - PSARP – colostomy closure effectively means long duration of treatment, significant costs in terms of time and resources, difficult social environment, mental trauma to parents and ultimately, questionable continence results. Patients not coming for follow up after a colostomy are well known to all pediatric surgeons. This may be due to loss of life due to diarrhea, associated anomalies or dis-inclination to continue treatment. In our center, this happened in 219 of 453 colostomies over a 10-year period (48% loss) despite most babies going home initially. It may be argued that some of these babies may be undergoing subsequent reconstruction elsewhere, but it is also true that being a tertiary care center, we perform secondary reconstructive surgery for many babies whose initial colostomy had been performed elsewhere. These 2 factors should reasonably neutralize each other for statistical purpose.
We presented here one of the largest series of male neonatal PSARPs for high and intermediate varieties in the literature (Group A). The initial mortality figures of 15%, mainly due to congenital heart disease, ventilator related issues or sepsis appear to be a drawback; however, cognizance has to be taken of the ultimate loss of 48% of babies in staged reconstruction. The ethical, philosophical, or reputational issues may have to be taken into consideration. Counseling of the family is essential and may help. Apart from providing the family, a one-time surgery solution, the continence rate is better in this group (45% good continence compared to 26% in staged reconstruction; P = 0.045). The authors do not underestimate the requirement of technical surgical skills required, logistics of neonatal reconstruction in terms of hospital resources (timings of available operation theaters for the neonates etc.). But once accomplished, this policy would conserve hospital resources especially valuable operation theater time of a busy hospital for other needy patients. The surgeon's skill is of paramount importance.
The advantages of neonatal single stage PSARP may be summarized as follows: For the parents, avoidance of two additional anesthesia/surgeries, burden of colostomy management under difficult social conditions and lesser time spent in the hospital and economic cost. For a busy hospital, saving resources of theatre and bed occupancy time is significant. For the patient, better continence rates and avoidance of entry into abdomen for colostomy and colostomy closure may enhance the quality of life. Better continence rates may be explained by better brain-anal reflexes development right from the time of birth., Technically, it is quite easy to find and dissect the meconium filled, distended rectal pouch at birth during primary PSARP procedure.
What are the reasons for not so complimentary results in these high ARMs? Poorly developed muscles of continence and neural deficiency are primary factors. It is possible that the puborectalis muscle, once divided and even if adequately reconstructed may end in fibrosis and may not function optimally. Disuse atrophy for many months or years of waiting for the reconstructive process to complete may partly be responsible. High incidence of constipation may be due to excess use of diathermy on the rectum resulting in denervation or due to colonic motility problems. Laparoscopy assisted pull through has been proposed in the recent years., However, the authors' personal exposure to the patients treated by this method did not inspire confidence in this technique. The “master” of the technique, Peña and Hong, in the world's largest series of 1192 patients  has reported 37.5% full continence, 37.5% soiling and 25% total incontinence. As a rule of thumb, 1/3 of these children may achieve good continence, 1/3 fair and 1/3 may be fully incontinent. Ultimately for achieving “social continence”, bowel management program or antegrade colonic enemata are a necessary part of rehabilitation in these children. Rintala and Lindahl  have reported the improvement of continence at adolescence as constipation disappears.
| Conclusion|| |
If the treating surgeon is reasonably skilled in neonatal surgery and PSARP procedures, the authors would recommend consideration of primary neonatal PSARP in these anomalies. Although there is higher initial mortality, ultimately more babies survive with better continence rates in the long term. Availability of operation theater logistics for performing these emergency procedures on an elective basis is essential.
Limitations of the study: It is not a randomized study. Most of the primary neonatal PSARPs were done by senior experienced surgeons and some amount of surgical skill bias cannot be eliminated.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
De Vries PA, Pena A. Posterior sagittal anorectoplasty. J Pediatr Surg 1982;15:638-43.
Peña A, Hong A. Advances in the management of anorectal malformations. Am J Surg 2000;180:370-6.
Ure BM, Rintala RJ, Holschneider AM. Scoring postoperative results. In: Holschneider AM, Hutson JM, editors. Anorectal Malformations in Children. Berlin: Springer; 2006. p. 351-60.
Scott JE, Swenson O, Fisher JH. Some comments on the surgical treatment of imperforate anus. Am J Surg 1960;99:137-43.
Kiesewetter WB, Turner CR, Sieber WK. Imperforate anus. Review of a sixteen year experience with 146 patients. Am J Surg 1964;107:412-21.
Kelly JH. The clinical and radiological assessment of anal continence in childhood. Aust N
Z J Surg 1972;42:62-3.
Kiesewetter WB, Chang JH. Imperforate anus: A five to thirty year follow-up perspective. Prog Pediatr Surg 1977;10:111-20.
Ditesheim JA, Templeton JM Jr. Short-term vs. long-term quality of life in children following repair of high imperforate anus. J Pediatr Surg 1987;22:581-7.
Iwai N, Yanagihara J, Tokiwa K, Takahashi T. Rectoanal pressure studies and postoperative continence in imperforate anus. Prog Pediatr Surg 1989;24:115-20.
Rintala RJ, Lindahl H. Is normal bowel function possible after repair of intermediate and high anorectal malformations? J Pediatr Surg 1995;30:491-4.
Holschneider AM, Jesch NK, Stragholz E, Pfrommer W. Surgical methods for anorectal malformations from Rehbein to Peña – Critical assessment of score systems and proposal for a new classification. Eur J Pediatr Surg 2002;12:73-82.
Peña A. Anorectal malformations. Semin Pediatr Surg 1995;4:35-47.
Freeman NV, Burge DM, Soar JS, Dedgewick EM, Soar GI. Anal evoked potentials. Z Kinderchir 1980;31:22-30.
Freeman NV, Bulut M “High” anorectal anomalies treated by early (neonatal) operation. J Pediatr Surg 1986;21:218-20.
Moore TC. Advantages of performing the sagittal anoplasty operation for imperforate anus at birth. J Pediatr Surg 1990;25:276-7.
Goon HK. Repair of anorectal anomalies in neonatal period. Pediatr Surg Int 1990;5:216-9.
Albanese CT, Jennings RW, Lopoo JB, Bratton BJ, Harrison MR. One-stage correction of high imperforate anus in the male neonate. J Pediatr Surg 1999;34:834-6.
Mishra BN, Narasimhan KL, Chowdhary SK, Samujh R, Rao KL. Neonatal PSARP versus staged PSARP – A comparative analysis. J Indian Assoc Pediatr Surg 2000;5:10-3.
Liu G, Yuan J, Geng J, Wang C, Li T. The treatment of high and intermediate anorectal malformations: One stage or three procedures? J Pediatr Surg 2004;39:1466-71.
Adeniran JO, Abdur-Rahman L. One-stage correction of intermediate imperforate anus in males. Pediatr Surg Int 2005;21:88-90.
Gangopadhyay AN, Gopal SC, Sharma S, Gupta DK, Sharma SP, Mohan TV. Management of anorectal malformations in Varanasi, India: A long-term review of single and three stage procedures. Pediatr Surg Int 2006;22:169-72.
Osifo OD, Osagie TO, Udefiagbon EO. Outcome of primary posterior sagittal anorectoplasty of high anorectal malformation in well selected neonates. Niger J Clin Pract 2014;17:1-5.
] [Full text]
Adeniran JO. One-stage correction of imperforate anus and rectovestibular fistula in girls: Preliminary results. J Pediatr Surg 2002;37:E16.
Menon P, Rao KL. Primary anorectoplasty in females with common anorectal malformations without colostomy. J Pediatr Surg 2007;42:1103-6.
Narasimharao KL, Prasad GR, Katariya S, Yadav K, Mitra SK, Pathak IC. Prone cross-table lateral view: An alternative to the invertogram in imperforate anus. AJR Am J Roentgenol 1983;140:227-9.
Narasimharao KL, Yadav K, Mitra SK, Pathak IC. Congenital short colon with imperforate anus (pouch colon syndrome). Ann Paediatr Surg 1984;1:159-67.
Rao KL, Menon P. Congenital pouch colon associated with anorectal agenesis (pouch colon syndrome). Pediatr Surg Int 2005;21:125-6.
Langemeijer RA, Molenaar JC. Continence after posterior sagittal anorectoplasty. J Pediatr Surg 1991;26:587-90.
Gangopadhyay AN, Pandey V, Gupta DK, Sharma SP, Kumar V, Verma A. Assessment and comparison of fecal continence in children following primary posterior sagittal anorectoplasty and abdominoperineal pull through for anorectal anomaly using clinical scoring and MRI. J Pediatr Surg 2016;51:430-4.
Nagdeve NG, Bhingare PD, Naik HR. Neonatal posterior sagittal anorectoplasty for a subset of males with high anorectal malformations. J Indian Assoc Pediatr Surg 2011;16:126-8.
] [Full text]
Pratap A, Tiwari A, Kumar A, Adhikary S, Singh SN, Paudel BH, et al.
Sphincter saving anorectoplasty (SSARP) for the reconstruction of Anorectal malformations. BMC Surg 2007;7:20.
Leva E, Macchini F, Arnoldi R, Di Cesare A, Gentilino V, Fumagalli M, et al.
Single-stage surgical correction of anorectal malformation associated with rectourinary fistula in male neonates. J Neonatal Surg 2013;2:3.
Nour S, Beck J, Stringer MD. Colostomy complications in infants and children. Ann R Coll Surg Engl 1996;78:526-30.
Patwardhan N, Kiely EM, Drake DP, Spitz L, Pierro A. Colostomy for anorectal anomalies: High incidence of complications. J Pediatr Surg 2001;36:795-8.
Chowdhary SK, Chalapathi G, Narasimhan KL, Samujh R, Mahajan JK, Menon P, et al.
An audit of neonatal colostomy for high anorectal malformation: The developing world perspective. Pediatr Surg Int 2004;20:111-3.
van der Steeg HJ, Schmiedeke E, Bagolan P, Broens P, Demirogullari B, Garcia-Vazquez A, et al.
European consensus meeting of ARM-Net members concerning diagnosis and early management of newborns with anorectal malformations. Tech Coloproctol 2015;19:181-5.
Georgeson KE, Inge TH, Albanese CT. Laparoscopically assisted anorectal pull-through for high imperforate anus – A new technique. J Pediatr Surg 2000;35:927-30.
De Vos C, Arnold M, Sidler D, Moore SW. A comparison of laparoscopic-assisted (LAARP) and posterior sagittal (PSARP) anorectoplasty in the outcome of intermediate and high anorectal malformations. S Afr J Surg 2011;49:39-43.
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;49:1635-42.
Peña A, Guardino K, Tovilla JM, Levitt MA, Rodriguez G, Torres R. Bowel management for fecal incontinence in patients with anorectal malformations. J Pediatr Surg 1998;33:133-7.
Rintala RJ, Lindahl HG. Fecal continence in patients having undergone posterior sagittal anorectoplasty procedure for a high anorectal malformation improves at adolescence, as constipation disappears. J Pediatr Surg 2001;36:1218-21.
[Table 1], [Table 2], [Table 3]
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