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Journal of Indian Association of Pediatric Surgeons
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Year : 2023  |  Volume : 28  |  Issue : 5  |  Page : 357-368

Can neonatal pull-through replace staged pull-through for the management of anorectal malformation? A systematic review and meta-analysis

1 Department of Paediatric Surgery, Chacha Nehru Bal Chikitsalaya, New Delhi, India
2 Department of Paediatric Surgery, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India
3 Department of Community Medicine and Family Medicine, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India

Date of Submission08-Feb-2023
Date of Decision26-May-2023
Date of Acceptance31-May-2023
Date of Web Publication05-Sep-2023

Correspondence Address:
Vivek Manchanda
Department of Paediatric Surgery, Chacha Nehru Bal Chikitsalaya, Geeta Colony, New Delhi - 110 031
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jiaps.jiaps_28_23

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Anorectal malformations (ARMs) are managed classically in three stages – colostomy at birth, anorectal pull-through after 2–3 months, and stoma closure. Single-stage pull-through has been contemplated in neonatal age aimed to reduce the number of procedures, better long-term continence, the better psycho-social status of the child, and reduced cost of treatment, especially in resource-strained countries. We conducted a systematic review comparing neonatal single-stage pull-through with stage pull-through and did a meta-analysis for the outcome and complications. Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines were followed. PubMed and Scopus databases were searched and RevMan 5.4.1 was used for the meta-analysis. Fourteen comparative studies including one randomized controlled trial were included in the systematic review for meta-analysis. The meta-analysis included 1845 patients including 866 neonates undergoing single-stage pull-through. There was no statistically significant difference for the occurrence of surgical site infection (odds ratio [OR] 0.82, 95% confidence interval [CI]: 0.24–2.83), urinary tract injury (OR 1.82, 95% CI: 0.85–3.89), rectal prolapse (OR 0.98, 95% CI: 0.21–5.04), anal stenosis/stricture, voluntary bowel movements (OR 0.97, 95% CI: 0.25–3.73), constipation (OR 1.01, 95% CI: 0.61–1.67), soiling (OR 0.89, 95% CI: 0.52–1.51), mortality (OR 1.19, 95% CI: 0.04–39.74), or other complications. However, continence was seen to be better among patients undergoing neonatal pull-through (OR 1.63, 95% CI: 1.12–2.38). Thus, we can recommend single-stage pull-through for managing patients with ARMs in the neonatal age.

Keywords: Abdominoperineal pull-through, continence, imperforate anus, laparoscopy-assisted anorectoplasty, outcome, posterior sagittal anorectoplasty

How to cite this article:
Manchanda V, Kumar P, Jadhav A, Goel AD. Can neonatal pull-through replace staged pull-through for the management of anorectal malformation? A systematic review and meta-analysis. J Indian Assoc Pediatr Surg 2023;28:357-68

How to cite this URL:
Manchanda V, Kumar P, Jadhav A, Goel AD. Can neonatal pull-through replace staged pull-through for the management of anorectal malformation? A systematic review and meta-analysis. J Indian Assoc Pediatr Surg [serial online] 2023 [cited 2023 Sep 24];28:357-68. Available from: https://www.jiaps.com/text.asp?2023/28/5/357/385145

   Introduction Top

Anorectal malformation (ARM) is the most common gastrointestinal congenital anomaly seen in approximately 1 in 4000–5000 live births.[1],[2] The management of ARM has changed four decades back – from (single-staged) “blind” pull-through to staged pull-through under colostomy cover. Posterior sagittal anorectoplasty (PSARP) as described by Pena and de Vries involves staged management of the anomaly comprises of colostomy at birth, definitive pull-through, and stoma closure.[3],[4] Similar guidelines were adopted by a consortium of pediatric surgeons after a conference at Krickenbeck[5] and reinforced by a European consensus meeting of ARM-Net members.[6] In addition, the consortium suggested that neonatal primary pull-through or delayed primary pull-through can be performed by an “experienced surgeon” if the rectal pouch is found ending within 1 cm from covering skin or vestibular fistula where painless dilatation can be performed by Hegar dilator >5 mm in size to avoid the morbidity associated with additional anesthesia for neonatal colostomy, improve behavioral aspects of continence training at age 4–5 years, and potential psychosocial damage.[6] However, the level of evidence for arriving at the consensus was not mentioned.

Neonatal pull-through (NPT) is being offered as the primary mode of management for ARM at many centers, even in absence of convincing literature proving it to be better than staged pull-through (SPT). We conducted this systematic review to collate the data present in the English literature about the outcome of NPT. The current systematic review was planned to compare the incidence of surgical site infection (SSI), rectal prolapse, anal stenosis/stricture, voluntary bowel movements, constipation, soiling, and continence after pull-through procedure either in neonatal age as single-staged procedure or later using staged approach.

   Materials and Methods Top

The systematic review and meta-analysis were performed as per Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines.[7] The protocol was registered with International prospective register of systematic reviews, CRD No. 42022310353.[8] Rayyan® was used for the cataloging and review of studies by different reviewers.[9]

   Systematic Review Top

PubMed and SCOPUS search was planned using predefined MeSH keywords. A Boolean search was performed using the keywords - “neonate” AND “anorectal malformations,” “neonate” AND “PSARP,” “neonate” AND “LAARP,” and “neonate” AND “primary pull-through” [Supplementary File 1]. Case reports, opinion articles, editorials, articles in a non-English language, experimental studies, and gray literature publications (i.e., reports, theses, conference proceedings, bibliographies, commercial documentation, and official documents not published commercially) were excluded from the database. Duplicates were excluded. Full text of articles potentially eligible for inclusion in the study was retrieved and independently assessed by three authors (VM, AJ, and PK) for inclusion in the systematic review, with blinding maintained with help of the Rayyan® platform. Any conflict thereafter was resolved through consensus.

Only articles that included the data about the objectives of the study were included in the final analysis. The outcome measures included were the incidence of SSI, rectal prolapse, anal stenosis/stricture, voluntary bowel movements, constipation, soiling, and continence in either group. We could not find enough data for rescue colostomy after primary pull-through and complications in form of anorectal stenosis and redo pull-through for systematic review in the literature.

   Meta-analysis Top

The meta-analysis was done using RevMan 5.4.1.[10] Data from various studies have been pooled using pooled odds ratio (OR). We produced I2 values to assess heterogeneity and quantify the dispersion of effect sizes. For the parameters where the heterogeneity was low (<40%), the fixed effect model was used to calculate the OR and in those with high heterogeneity (I2 > 40%), the OR was calculated using the random effects model for categorical variables, and standardized mean differences for continuous variables, along with 95% confidence intervals (CIs).

   Quality Assessment Top

Risk of bias assessment was done independently by three authors (VM, AJ, and PK) using the Newcastle-Ottawa score (NOS) for case-control studies.[11] Differences between the two authors were resolved through consensus. After the meta-analysis, the quality of evidence was assessed using the GradePro guideline development tool.[12] A Measurement Tool to Assess Systematic Reviews (AMSTAR) was used to appraise the current systematic review to complete the PRISMA checklist.[13]

   Results Top

A total of 430 titles were found on PubMed and SCOPUS searches using predefined criteria for inclusion in the study. One hundred and sixty-one among these were excluded as duplicates. The rest 269 titles were screened by the first three authors independently for inclusion in the study. Twenty-eight of the selected articles were screened using full-text articles. Ten further studies were added to the database from references of the screened articles. A total of 14 studies were included in the systematic review.[14],[15],[16],[17],[18],[19],[20],[21],[22],[23],[24],[25],[26],[27] PRISMA chart is given in [Figure 1].
Figure 1: PRISMA flow diagram. PRISMA: Preferred Reporting Items for Systematic Reviews and Meta-Analysis

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Thirteen of the studies were case − control studies, while in one study, the cases and controls were chosen randomly (no blinding or allocation concealment). The majority were retrospective studies, with only 2 prospective. The surgery done in 7 studies was Pena's PSARP, semi-PSARP, anterior sagittal anorectoplasty (ASARP) and SSARP in one each, and LAARP in three while one study included patients undergoing pull-through through various routes/techniques. Most of the studies were from Asia (China – 4, India – 3, USA – 3, Japan – 1, Nepal – 1, Malaysia – 1, and Saudi Arabia - 1). The studies reported results from 1845 patients including 866 patients undergoing single-staged NPT. The largest study included 948 patients including 490 NPT [Table 1].
Table 1: Summary of studies included in the systematic review

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   Risk of Bias Assessment Top

The risk of bias assessment was done using NOS for case-control studies.[11] All but one study had a score of 2/4 among selection scores, as all studies included hospital controls with the same disease and different approach. The sample included all the patients managed by the authors using the given approach, but selection criteria regarding decision for choosing a given approach were not mentioned in any studies. Thus, selection bias might be a confounding factor in all the studies. Only five studies compared the two groups for possible confounding factors including the age, birth weight, gestational age, and type of malformation (high vs. low). Thus, the possibility of confounding due to noncomparability of the case and control groups cannot be ruled out. The exposure criteria were presented in most of the studies except one.[16] The data collected were from patient records and secured, and the methodology remained same for cases and controls. The data presented to be complete with comparable dropout rates in most of the studies. However, blinding was not followed in any of the studies.

Overall, nine of the studies had a score ≤5, while 5 studies had a score of 6 or more. The maximum score achieved was 7 [Supplementary File 2].

   Surgical Site Infection Top

All except one study provided data regarding SSI among the patients undergoing pull-through for ARM. One study reported zero SSI in both groups. Fifty-six among 798 (7.01%) NPT had infections, while 143 children had SSI among 898 children undergoing SPT (15.92%) [Table 2]. The latter included infective complications after stoma creation and stoma closure. Different studies reported variable outcomes with six studies reporting more SSI among SPT and four reporting increased incidence of SSI after NPT. Colostomy was required after NPT in five patients to rescue the repair. No study reported data on the requirement to redo pull-through procedures among patients undergoing SPT.
Table 2: Findings of various studies included in the systematic review

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In the meta-analysis, there was high heterogeneity with an I2 = 85%. The overall OR was 0.82 (CI 95%, 0.24–2.83) in favor of NPT [Figure 2]. Due to high heterogeneity, subgroup analysis and sensitivity analysis were undertaken.
Figure 2: Meta-analysis for Surgical Site Infection after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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Subgroup analysis based on the period of the study showed reduced heterogeneity among relatively recent studies <15 year old but it could not achieve any statistical significance. However, sensitivity analysis using a fixed effect model instead of a random effect model favoured NPT among recent studies with OR 0.39 (95% CI 0.18–0.84) [Supplementary File 3].

A sensitivity analysis was done based on NOS. The analysis using random effect model reduced heterogeneity (I2 = 0%) among studies with NOS >5 but could not achieve statistical significance (OR 1.82, CI 95% 0.85–3.89) [Supplementary File 4].

Another subgroup analysis was done based on whether the studies included LAARP or open (PSARP/ASARP/SSARP/others) procedures. The subgroup analysis did not affect the results significantly [Supplementary File 5].

   Urinary Tract Injury Top

The urethral injury was reported in one patient (out of 14) by Mishra et al.[15] and 13 patients (out of 490) by Gangopadhyay et al.[18] during NPT. Only Gangopadhyay et al. reported urethral injury in 26 patients (out of 458) among patients during SPT. Opposing results resulted in a high I2 = 46%. The combined OR of 0.81 (CI 95%, 0.11–5.75) was obtained in the meta-analysis [Figure 3].
Figure 3: Meta-analysis for Urinary Tract Injury during pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Rectal Prolapse Top

Data on rectal prolapse was measured in five studies only. With one study reporting zero incidences, 16/348 patients had a rectal prolapse. The studies had moderate heterogeneity with I2 = 40%. No significant difference was noted on meta-analysis [OR 1.02, CI 95% 0.21–5.04, [Figure 4]]. Sensitivity analysis did not alter the overall results.
Figure 4: Meta-analysis for rectal prolapse after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Anal Stenosis/Stricture Top

Only three studies reported on the incidence of anal stenosis/stricture among patients operated for ARM. While one study reported zero incidences, other studies reported one case each in either group. Thus, no effort for meta-analysis was done.

   Voluntary Bowel Movements Top

Voluntary bowel movements were reported in three studies. Patients having it after pull-through procedures were found to be similar in two groups – 94.37% (67/71) and 94.44% (85/90) in patients undergoing NPT and SPT respectively. The meta-analysis included only two studies as in one study all patients had voluntary bowel movements. The heterogeneity index was low with an I2 = 0%. However, no definite association could be assigned [OR 0.97, CI 95% 0.25–3.73, [Figure 5]].
Figure 5: Meta-analysis for voluntary bowel movements after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Constipation Top

It was reported in 5 studies. 34.72% (50/144) of patients undergoing NPT and 32.41% (47/145) of patients undergoing SPT had significant constipation requiring laxatives and/or enemas. In the meta-analysis, the heterogeneity was low with I2 = 0%. Overall, the two groups had comparable incidence of constipation with OR of 1.01 [CI 95% 0.61–1.67, [Figure 6]].
Figure 6: Meta-analysis for constipation after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Soiling Top

It was reported in four studies. Forty-seven among 136 patients (34.56%) undergoing NPT and 45 among 138 patients (32.61%) undergoing SPT had soiling. In the meta-analysis, the heterogeneity was low with I2 = 0%. None of the studies was conclusive regarding the incidence of soiling after the pull-through procedure as was the pooled estimate. Overall OR was 0.89 [CI 95%, 0.52–1.51, [Figure 7]].
Figure 7: Meta-analysis for soiling after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Continence Top

Continence reporting was variable among the different studies concerning the criteria and parameters used for assessing continence. The minimum follow-up period was also variable, ranging from 1 to 4 years. Continence rates were reported only for SPT in one study and thus were excluded from the analysis.[19] Continence rates were reported for both groups in 6 studies. Good, intermediate, and poor continence rates were reported in 58.76% (275/468), 27.78% (130/468), and 13.46% (63/468) children following NPT as compared to 50.83% (215/423), 32.39% (137/423), and 20.09% (85/423) after SPT.

In the meta-analysis, we combined good and intermediate continence as acceptable continence as against poor continence after the pull-through procedure. While Liu et al reported that 100% of patients had good or intermediate continence scores after either procedure,[17] Mishra et al favoured SPT to achieve acceptable continence,[15] while four other studies favoured NPT to achieve acceptable continence.[14],[18],[21],[27] In the meta-analysis, the heterogeneity was low (I2 = 0%) and a fixed effect model was used to pool the results. Overall risk ratio was 1.63 [CI 95%, 1.12-2.38] in favor of NPT [Figure 8].
Figure 8: Meta-analysis for “good + intermediate continence” after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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Another meta-analysis was done to include only good continence as the outcome. The outcome had moderate heterogeneity (I2 = 44%), thus fixed effect model was used for pooling studies. The meta-analysis showed significant improvement in continence for NPT [OR 1.69, CI 95% 1.27–2.21, [Figure 9]].
Figure 9: Meta-analysis for good continence after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Other Complications Top

Colostomy complications including stomal necrosis, stomal prolapse, peri-stomal excoriation, burst abdomen, anastomotic leak, and postoperative adhesive obstruction were reported in five studies.

   Mortality Top

Six studies reported mortality in the patients operated on for anorectal malformation. Three of them reported zero deaths in the patients undergoing pull-through procedure regardless of age at pull-through. Thirty-eight expired after NPT (5.50%) while 187 died among SPT (27.21%). Menon et al also reported a 48% loss to follow-up after initial colostomy as a neonate. Mortality appears to be comparable in the study by Gangopadhyay et al after a definitive pull-through procedure (4.49% in primary PSARP vs 4.7% in SPT). Two studies showed more mortality among patients undergoing SPT while one study showed it to be higher among patients undergoing NPT. Heterogeneity was considered very high with I2 95%. No group fared better than the other with overall odds ratio of 1.19 [CI 95%, 0.04-39.74] [Figure 10]. Any meaningful sensitivity analysis was not possible for the outcome.
Figure 10: Meta-analysis for mortality after pull-through for anorectal malformation. CI: Confidence interval, OR: Odds ratio, PSARP: Posterior Sagittal Anorectoplasty

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   Quality Assessment of Evidence Top

The quality of evidence produced in the systematic review was assessed by GRADEPro. As the evidence was created using retrospective case-control studies, the evidence was rated as very low for all outcomes [Table 3].
Table 3: Summary of findings

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   Quality Assessment of the Systematic Review Top

It was analyzed using AMSTAR 2. The results are available in supplementary files [Supplementary File 6].

   Discussion Top

The data regarding the incidence of SSI was heterogeneous among various studies, with some studies highlighting the reduced infection after NPT[18],[20] while others warning increased incidence of SSI and chances of wound dehiscence among neonates undergoing definitive pull-through.[14],[17] On pooling the results from 13 studies, there was no statistically significant difference between the two groups. The subgroup analysis based on the period of study, NOS and sensitivity analysis by excluding the studies with only LAARP cases did not affect the association. The variation among studies can be due to the inclusion of all stages in some studies while others include only definitive pull-through procedures. Another reason could be variable definition of SSI. It may be submitted that dehiscence after pull-through could be due to inadequate mobilization of the rectum with anastomosis under tension in certain patients rather than true infection. Overall, the NPT procedures had lesser infections than SPT procedures.

Colostomy has the advantage to protect definitive repair, reduce SSI, and reduce damage to the perineal muscles.[17] In NPT, tapering of terminal rectum (not ectatic) is not required and sterile meconium reduces the chances of SSI.[14],[18],[22],[28],[29] Babies may be nursed in a prone position to prevent the soiling of the wound from meconium spillage and reduce SSI.[17] Other mechanisms to reduce SSI after PSARP are protecting tissue and preventing fat liquefaction, thorough hemostasis, obliterating dead cavity, keeping transanal tube, colon irrigation, and keeping the baby nil per oral.[17],[22],[27],[30] Although it is expected that wound infection would adversely affect the long-term outcome of continence, studies have demonstrated similar long-term results.[17] Moreover, delayed primary pull-through is a well-accepted technique for rectoperineal and vestibular fistula without covering colostomy.[1],[26]

As a marker for operative accuracy, we explored the incidence of urinary injuries in the literature. The incidence of urinary tract injury has been minimal in most of the studies. Adherence to the principles of the surgery rather than the timing of surgery is required to avoid urinary tract injury.[29] The accurate delineation of pathological anatomy increases safety by minimizing avoidable surgical trauma or injuries to the urinary and genital tract.[31] Urinary tract injury may be predisposed to either by improperly performed distal colostogram providing incomplete information or to variation in the technique that the surgeons employ for pull-through.[18]

Understanding the anatomy of the anorectal defect is of paramount importance for a safe anorectal pull-through.[14] Adequate exposure of the defect as guided by Stephens' and Pena's work has made such a surgery even in neonatal age a safe procedure.[14] Cross table prone lateral X-ray, cystoscopy, voiding cystourethrography, and magnetic resonance imaging can also provide reliable knowledge about the level of the distal rectal pouch.[14],[17],[29],[31],[32],[33],[34],[35] The stoma allows the pediatric surgeon to perform pressure augmented distal colostogram for better delineation of the anomaly.[17] albeit an operator-dependent investigation and carries a risk of radiation exposure and a bowel perforation.[31],[32] Some authors believe as the approach during LAARP is from the pelvic side, the knowledge of the exact level of the fistula may be not required by preoperative radiological investigations, as the diagnosis can be safely made intra-operatively under direct vision.[27],[36],[37]

Rectal prolapse, anal stenosis or stricture also was similar among patients undergoing pull-through at neonatal age or later. Adequate mobilization of the rectum with preservation of intramural blood supply and fixation in the centre of the sphincter complex ensures minimal postoperative complications.

Continence was reported in six studies as documented by scoring systems or using voluntary bowel movements, constipation, and soiling as markers in other studies. Continence scoring was found to be significantly better among the patients undergoing NPT. Albanese et al. identified that the early restoration of gastrointestinal continuity would train the perineal muscles and allow better long-term faecal continence. Early correction of ARM would allow neuronal networks and synapses to be formed to provide near-normal anorectal function.[17],[18],[27] Literature also demonstrates poorer continence rates in neonatal colostomy due to defunctioning of the rectum and persistence of meconium in the rectum distal to stoma.[27],[38] Single-stage LAARP has been successfully used and shown to be safe with comparable short-term complications and long-term continence rates.[23],[27],[39] The continence rates after traditional SPT vary from 2% to 84%.[21] Continence rate is affected by the type of malformation, level of fistula, and quality of sacrum and spine.[23] However, none of the studies has done subgroup analysis for known confounders.[40]

Only three studies reported mortality among patients undergoing pull-through procedures for ARM. While two studies reported on data for mortality after definitive pull-through only, one study combined data for all stages in a SPT group. In a study, the mortality in SPT operation was reported to be 40% overall, as compared to 4.5% in the NPT.[18] The high mortality in this study has been reported to be due to the historical control group. In another large study, the mortality in NPT has been reported to be 15%, attributable to associated complex heart disease and sepsis.[21] The increase in mortality after NPT is associated with poor nursing conditions and a lack of infrastructure in certain centres.[15] Less than one-third of the patients for whom stoma was created at neonatal age had their definitive pull-through procedure done.[15] Another large study reported only 52% of neonates completed all stages of correction of ARM.[21] The lost patients could be attributed to mortality due to associated malformations, infective or nutritional complications associated with colostomy or social reasons and disinclination to continue treatment and even abandonment of patients after stoma creation at birth.[20],[21]

Inaccurate identification of the fistula preoperatively is likely to make it a matter of intra-operative judgement of the operating surgeon to use laparotomy for mobilization of the rectum. Technically, the NPT is easier as the rectal pouch is filled with meconium and air and absence of peri-rectal fibrosis secondary to pouchitis.[18],[20],[22] Among female neonates, the vagina is found to be thicker (under the influence of maternal hormones) facilitating its dissection away from the rectum easier.[22],[25] Some surgeons find the surgery difficult due to small tissues and identify anal sphincter in neonates, but the use of magnification makes surgery easier.[17],[20],[25] It is expected as the experience and expertise of surgeons should guide the preference of age at surgery.[21] NPT took less time although statistical significance was not achieved.[20]

Single-staged NPT reduces the physiologic, psychological, social, and economic burden of the malformation on the patients and parents by avoidance of stoma and its complications and reducing surgery episodes and associated costs.[17],[20],[25] Furthermore, all three stages may take months to years to complete. In a study reported from India, the mean age at stoma closure was 34.4 months, with probable reasons for delayed procedures such as low socio-economic status, associated malnutrition and colostomy complications, poor follow-up, and management of associated anomalies.[18] Colostomy-related issues are a major factor resulting in dissatisfaction among parents.[25] Colostomy related complications (28%–74%) may include hypertrophy of the rectum, stoma prolapse, stoma retraction, stomal stricture, part of intestinal loop necrosis, bleeding, wound infection, adhesive obstruction, skin rash, peristomal erosion, urinary tract infection, leaked anastomosis at the time of stoma reversal, incisional hernia, and multiple scars.[17],[23],[25],[27] Pena reported 616 complications in 464 children (33%) with stomas created outside his institute, and four among 50 (8%) stomas created at his institute. He emphasized that these are a result of poor technique while performing a colostomy and if proper care is taken most could be avoided.[32]

The potential advantages of single-staged NPT are the need for only one operation, shorter total hospital stay, and avoidance of colostomy-related complications.[14],[23] While waiting for a delayed primary pull-through the patient may develop a megarectum with a poorer outcome.[41] Reducing waiting time for definitive surgery helps in improved care for children where resources are stressed as in many developing countries.[21] The one-staged NPT allows caregivers to spend their resources on the management of associated anomalies.[14] NPT is also expected to reduce urinary tract infection after disconnection of the recto-urinary fistula.[18]

Goon reported contraindications for NPT as fatal associated anomalies, very ill babies with high defects, complete sacral agenesis, cystic dilatation of the colon, and cloacal anomaly.[14]

   Limitation of Evidence in the Review Top

All but three studies included in the meta-analysis were retrospective case − control studies reducing the level of evidence. In the only randomized controlled trial, the subject's method of allocation and blinding was not provided. It must be noted that none of the studies has done subgroup analysis for known confounders for the continence after the pull-through surgeries namely type of ARM and sacral ratio. The analysis in the studies presumes the two groups to be similar with respect to possible confounders. The definition of the outcome measures such as SSI was also not defined. The methodology of the data collection was not defined in any of the studies reducing the reliability of the findings.

   Potential Bias in the Review Process Top

The review included only comparative studies (randomized controlled trial or case-control studies) in the review. The search was limited to only PubMed and SCOPUS (and cross-references of the articles included) as the source of publications. We did not run a dedicated search for nonpublished literature or that in trial/study registries. Some useful data might have been missed from the case series published.

We also could not evaluate for publication bias as nonpublished data was not included in the review.

   Conclusions and Recommendations Top

It can thus be recommended that NPT is an acceptable procedure for ARM in hands of experienced surgeon. Strict adherence to the principles of pull-through (PSARP/LAARP) and avoiding urinary injury should be paramount. NPT have the benefits of reducing costs, waiting time, loss to follow-up, and reducing poor continence among patients undergoing pull-through. Improvement of the quality of evidence by performing more randomized controlled trials is required.

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Conflicts of interest

There are no conflicts of interest.

   Supplementary Files Top

Supplementary File 1: Search Strategy

PubMed: n = 195

(“Infant, Newborn” [Mesh] OR Neonate OR Neonat*) AND (“Posterior sagittal anorectoplasty” OR “PSARP” OR “Laparoscopic-assisted anorectoplasty” OR “LAARP” OR “Primary Pull Through”)

Scopus: n = 235

(TITLE-ABS-KEY [neonate OR neonatal OR newborn] AND TITLE-ABS-KEY [“Posterior sagittal anorectoplasty” OR “PSARP” OR “Laparoscopic-assisted anorectoplasty” OR “LAARP” OR “Primary Pull Through”])

   References Top

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]

  [Table 1], [Table 2], [Table 3]


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