Single-staged Anterior versus Posterior Sagittal Anorectoplasty for Low Female Arms

ABSTRACT

Background:

Anorectal malformations (ARMs) are among the predominant birth anomalies seen in pediatric surgery.

Aims:

The aim of the study was to create a detailed comparison of short-term (3–6 months) outcomes in female patients having rectovestibular fistula (RVF) operated through a single stage by both techniques, posterior sagittal anorectoplasty (PSARP) versus anterior sagittal anorectoplasty (ASARP).

Materials and Methods:

Our prospective cohort study involved 36 females whose ages fell between 3 and 12 months and who developed congenital ARM in RVF or rectoperineal fistula. Participants went through an equal random categorization into two groups: Group 1: patients with RVF or rectoperineal fistula who underwent single-staged PSARP and Group 2: patients with RVF or rectoperineal fistula who underwent single-staged ASARP.

Results:

There was a notable association between the incidence of wound infection and the hospital stay period, as with total parenteral nutrition (TPN) duration and the start of enteral feeding. 7 (63.4%) patients developed dehiscent wounds, and their hospital stay was significantly longer as opposed to the others with (P < 0.001). Wound infection was significantly associated with longer TPN duration among infected wounds compared to another group (P < 0.001). Wound dehiscence was significantly associated with wound infection among both groups (P = 0.025 and P = 0.002). Hospitalization was significantly prolonged between the two groups, with the PSARP group longer than ASARP (P = 0.003 and P < 0.001).

Conclusions:

ASARP may be a better option in low-resource settings, as it is associated with shorter hospital stays, fewer wound infections, and fewer complications than PSARP. Patients who underwent PSARP experienced more extended hospital stays, more infections, and longer TPN durations.

INTRODUCTION

Anorectal malformations (ARM) represent congenital birth abnormalities associated with the anorectaum as well as the pelvis in which the anus is missing or deformed. The prevalence of ARMs is equal in both sexes and ranges from 1 in 3500 to 1 in 5000 live birth.[1]

A rectovestibular fistula (RVF) represents the predominant anomaly type among girls. It is recognized as low malformation characterized by the anal hole being positioned between the vagina and the vestibule’s fourchette.[2]

Multiple operative approaches were adopted to manage RVF cases. However, posterior sagittal anorectoplasty (PSARP) emerged as a primary surgical approach, and de Vries and Pena developed it at the beginning of the 1980s.[3,4,5]

Classically, this operation is performed through three stages: colostomy, PSARP, and colostomy closure. On the other hand, different research addresses that PSARP could be accomplished through only one stage with no need for colostomy, exhibiting comparable acceptable findings; several medical institutes are implementing the anterior sagittal anorectoplasty (ASARP) as a potential substitute for PSARP repair. In addition, the outcomes reported are encouraging.[6,7,8]

PSARP is widely regarded as the gold standard due to its comprehensive approach and better functional outcomes; it requires advanced surgical skills, specialized equipment, and extensive postoperative care, making it challenging to implement in low-resource environments. In contrast, ASARP offers a less complex surgical option, with shorter procedure times and reduced postoperative care requirements, making it a more feasible solution in settings with limited healthcare resources.

Therefore, this work aimed to compare the short-term consequences among female patients with RVFs operated through a single stage using PSARP and ASARP techniques.

MATERIALS AND METHODS

Our prospective randomized, single-blind clinical study involved 36 females aged 3–12 months with a congenital ARM in the RVF or rectoperineal fistula who underwent single-staged repair, whether ASARP or PSARP. Customized cases having RVF were excluded, as well as cases with severe vertebral anomalies and sacral agenesis.

Randomization and blindness

A random list was generated using an online randomization application (http://www.randomizer.org), and the codes for each patient were stored in an opaque sealed envelope. Using a 1:1 allocation ratio, the patients were divided into two equal groups:

Group 1 patients with RVF or rectoperineal fistula who underwent single-staged PSARP [Figure 1].

Figure 1.

Posterior sagittal anorectoplasty (a) catheterization while patient supine, (b) patient position, (c) division of muscles, (d) dissection of rectum from the vagina, (e) dissection of rectum from vagina, (f) anoplasty and (g) end of repair

Group 2 patients with RVF or rectoperineal fistula underwent single-staged ASARP [Figure 2].

Figure 2.

Anterior sagittal anorectoplasty (a) position, (b) position and stay sutures, (c) midline incision, (d) mobilization of rectum in (d1) on left, (d2) on right, (e1) dissection of rectum from vagina, (e2) separating the rectum from vagina, (f) anoplasty, (g) closure of perineal body and anoplasty and (h) end of repair

All participants went through a comprehensive medical history, including family history of previous babies with the same anomalies, type of labor and drug intake during pregnancy, proper general examination to detect any associated anomalies such as VACTREL anomalies, and local examination to allocate the position of the fistula in the fourchette or the perineum.

Preoperative investigations were ordered in the form of blood workup abdominal pelvic US to detect the associated urological anomalies and the presence of presacral masses. Echocardiography was obtained to detect any associated cardiac anomalies, its assessment, follow-up and need for specific therapy, and X-ray spine for detection of sacral bone anomalies. Magnetic resonance imaging spine is usually requested in case of the presence of severe sacral anomalies to exclude tethered cord anomaly.

After fulfilling the previous workup, the first case allocation was done randomly. All participants went through an allocation alternatively (each participant was recruited within a treatment group) till 18 participants were involved within each group. Only one pediatric surgeon with 11 years of expertise performed all surgical procedures. Regarding Group 1 who underwent PSARP, it is done with GA utilizing a caudal block. All participants were positioned in a prone jackknife posture with the pelvis raised. The precise anus, as well as the anal musculature complex, was ascertained with the use of electrical stimulation. An incision was made in the skin and subcutaneous tissue of the perineum along the midline, starting at the coccyx tip and extending toward and around the fistula. Subsequently, the muscles, rectum, as well as vagina were detached from each other. Initially, the posterior wall was freed, followed by the lateral sides, and ultimately, the anterior rectal wall adjacent to the vagina was carefully detached. The anus, as well as the rectum, were anatomically positioned and secured in place utilizing sutures. Subsequently, the surgical procedure was completed with a perineal as well as incisional line repair.

Regarding group 2 who underwent ASARP, the procedure is done under general anesthesia with a caudal block. A tiny sheet was positioned under the buttocks to provide a good view of the surgical area (perineal region). Soft cotton rolls were utilized to support the legs, thus avoiding any harm to the hip joint or nerve impairment. Urinary catheterization was carried out. The anus location was confirmed through the presence of the anal dimple, then marked utilizing electrical stimulation as well as silk sutures. An incision was performed along the middle line, starting from the fistula posterior margin, extending to the proposed anus posterior limit. The incision was deepened, and all tissues, involving the perineal muscles and anterior fibers of the external sphincter complex, were divided precisely at the midline. The posterior and lateral rectal walls were carefully freed, and the anterior rectal wall was separated meticulously from the vagina. The rectum was mobilized completely circumferentially until the fistulous opening reached the proposed neoanus location without tension. The rectum was then positioned in the center of the muscle complex. In addition, the perineum as well as the incision line was repaired completely.

All participants were not allowed oral feeding for about 5 days after the surgical procedure. In addition, total parenteral nutrition (TPN) began on the 1^st day postoperatively and was continuous for a few days until oral feeding commenced.

We did some bowel preparation by keeping the patients NPO for 2 days preoperatively with IV fluids and antibiotics, flagyl, and ciprofloxacin; some of our patients developed minor accepted wound dehiscence and managed conservatively, and there was no major wound dehiscence that needed surgical repair or affected the postoperative continence.

All participants within the two groups were closely observed during the surgical procedure, postoperatively within the hospital, and during intermittent outpatient visits in the clinic.

Regarding anal dilatation, patients underwent anal calibration, and the parents were taught the method of dilatation as well as allowed to carry out dilatation two times daily, passing the dilator to the designated mark and keeping it inside for 30 s. Increasing the dilator’s size is accomplished every 2 weeks until the appropriate size for the age, and then we gradually decrease the dilatation frequency according to Pena protocol.

All subjects underwent a 6–12-month follow-up following the treatment. The outcomes and adverse events were then compared among both groups. The complications involving surgical site infection (SSL) as well as anorectoplasty site dehiscence, anal stenosis, rectal prolapse, rectovaginal fistula, then following bowel habits such as constipation, incontinence, and pseudo incontinence. The acquired data were gathered utilizing a checklist tailored for the research. The data underwent a statistical analysis using SPSS v26 (IBM Inc., Chicago, IL, USA).

Statistical analysis

Data underwent a statistical analysis utilizing SPSS v26 (IBM Inc., Chicago, IL, USA). The Shapiro–Wilks test and histograms were used to assess the normality of data distribution. Quantitative nonparametric data were displayed as the median and interquartile range, analyzed using the Mann–Whitney test, and compared between preoperative and postoperative within groups using the Wilcoxon signed-rank test. Qualitative variables were displayed as frequency and percentage (%) and then analyzed using the Chi-square or Fisher’s exact test when appropriate. A two-tailed P < 0.05 was deemed to show a statistical significance.

RESULTS

Sociodemographic data are shown in Table 1.

Table 1.

Sociodemographic data of all cases (n=36)

Variable	n (%)
Weight (kg)	7 (6.5–7.25)
Age (months)	7 (6–8)
Diagnosis
RVF	33 (91.7)
Rectoperineal fistula	3 (8.3)

Data exhibited as n (%). RVF: Rectovestibular fistula

As elucidated in Table 2, no statistically significant variation was documented among both groups concerning weight and age during operation time.

Table 2.

The difference between both groups concerning the sociodemographic characteristics, the feeding details and postoperative (complications, bowel control,and hospital stays) of the participants (n=36), and between wound infection and (wounddehiscence, hospital stays, total parenteral nutritionduration, and parenteral feeding start)

Value	ASARP (n=18)	PSARP (n=18)	P
Weight (kg)	6.79±0.49	6.97±0.61	-
Age at operation (months)	6.95±0.95	6.69±1.25	-
At the feeding details
Enteral feeding start	7 (7–7)	7 (7–7)	-
TPN	8 (8–8)	8 (8–8)	-
Postoperative complications
Wound infection	6 (33.3)	5 (27.8)	1.00
Wound dehiscence	3 (16.7)	4 (22.2)	1.00
Postoperative bowel control
Normal	4 (20)	7 (43.8)	0.323
Soiling	1 (5)	2 (12.5)
Constipation	13 (65)	6 (37.5)
Constipation with overflow	2 (10)	1 (6.3)
Postoperative hospital stays
Hospital stays (days)	11 (11–11)	11 (11–13)	0.05
Wound infection	Clean	Infected	P
Wound dehiscence
Intact (n=29)	25 (63.6)	4 (36.4)	<0.001*
Dehiscent (n=7)	0	7 (63.6)
Hospital stays (days)	11 (11–11)	15 (14–16)	<0.001*
TPN duration	8 (8–8)	11 (11–11)	<0.001*
Parenteral feeding start	7 (7–7)	10 (10–10)	<0.001*

*Significant P value <0.05. Data exhibited as n (%) and median (IQR). Mean±SD, n (%) or median (IQR). TPN: Total parenteral nutrition, ASARP: Anterior sagittal anorectoplasty, PSARP: Posterior sagittal anorectoplasty, IQR: Interquartile range, SD: Standard deviation

All patients in both groups were kept TPN dependent for 5 days and allowed oral feeding on day 6 postoperatively, as elucidated in Table 2.

Regarding complications occurring postoperatively, involving wound infection as well as dehiscence, no statistical variation was documented among both groups with a P = 1, as mentioned in Table 2.

Along with these data, no statistical significance was reported among both groups as regards bowel habits and bowel control, as elucidated in Table 2.

There was a notable amelioration between the incidence of wound infection and the hospital stay period as with TPN duration and the start of enteral feeding, as elucidated in Table 3; in our study, seven patients (63.4%) developed dehiscent wounds, and their hospital stay was significantly more extended as opposed to the others with (P < 0.001).

Table 3.

Correlation between wound infection and (wound dehiscence, hospital stays, total parenteral nutrition duration, and parenteral feeding start)

	Clean	Infected	P
Wound dehiscence, n (%)
Intact (n=29)	25 (63.6)	4 (36.4)	<0.001*
Dehiscent (n=7)	0	7 (63.6)
Hospital stays (days)	11 (11–11)	15 (14–16)	<0.001*
TPN duration	8 (8–8)	11 (11–11)	<0.001*
Parenteral feeding start	7 (7–7)	10 (10–10)	<0.001*

*Significant P value <0.05. Data exhibited as n (%) and median (IQR). TPN: Total parenteral nutrition, IQR: Interquartile range

In line with this, wound infection was significantly associated with longer TPN duration among infected wounds compared to the other group (P < 0.001). Children with infected wounds started their enteral feeding later than those with clean wounds. This was statistically significant (P < 0.001) [Table 3].

Wound dehiscence was significantly associated with wound infection among both groups (P = 0.025 and P = 0.002) for the ASARP and PSARP groups, respectively. No statistically significant variation was documented among clean and infected wounds regarding the participants’ age within both groups (P = 0.362 and 0.508) for ASARP and PSARP groups, respectively. Hospitalization was significantly prolonged between the two groups, with the PSARP group longer than ASARP (P = 0.003 and P < 0.001), respectively. Infected wounds were associated with longer TPN duration among both groups, with both groups equally affected (P < 0.001 and P < 0.001, respectively). These findings were the same when comparing both groups (P < 0.001 and P < 0.001) for ASARP and PSARP, respectively [Table 4].

Table 4.

Relation of wound infection to dehiscence, age, hospital stay, total parenteral nutrition duration, and start of oral feeding in each group

	ASARP (n=18)			PSARP (n=18)
	Clean	Infected	P	Clean	Infected	P
Dehiscence, n (%)
Intact	12 (100)	0	0.025*	13 (100)	0	0.002*
Dehiscent	3 (50)	3 (50)		1 (20)	4 (80)
Age	7 (7–8)	7 (5.75–7.25)	0.362	6 (5–8)	7 (6–8)	0.508
Hospital stays (days)	11 (11–11)	14.5 (11.75–16.25)	0.003*	11 (11–11)	16 (15–17)	<0.001*
TPN duration	8 (8–8)	11 (11–11)	<0.001*	8 (8–8)	11 (11–13)	<0.001*
Enteral feeding start	7 (7–7)	10 (10–10)	<0.001*	7 (7–7)	10 (10–10)	<0.001*

*Significant P value <0.05. Data exhibited as n (%) and median (IQR). ASARP: Anterior sagittal anorectoplasty, PSARP: Posterior sagittal anorectoplasty, TPN: Total parenteral nutrition, IQR: Interquartile range

Anal size for age at the end of the follow-up period of the participants is shown in Table 5.

Table 5.

Anal size for age at end of follow-up period of the participants (n=36)

Anal size	Anal calibration	Postoperative
	Below normal	Below normal	Normal	Above normal
Preoperative below normal	36 (100)	18 (50)	17 (47.2)	1 (2.8)

Data are presented as n (%)

DISCUSSION

RVF, also known as vestibular anus, represents the predominant deformity among female ARMs seen by pediatric surgeons. Furthermore, its importance in surgical repair remains often undervalued.[9]

Despite the long clinical success of three stages of PSARP repair for RVF introduced by deVries and Peña,[10] growing evidence has shown great success for a PSARP.

Along with this assertion, requiring a diverting colostomy when correcting RVF is debatable. One-stage repair for RVF without colostomy has gained acceptance among many authors as a widely known approach to managing RVF, particularly in newborns. Such an approach involves a single main surgical procedure in neonates that is safe with minimum adverse events.[11] In contrast, some experts have suggested a multistage repair procedure involving colostomy to prevent wound complications that might potentially impact the functional result.[12] Adeniran[13] showed that the repair of RVF could be a safe surgical procedure when carried out through a single-stage ASARP.[14,15,16]

Osifo et al.[17] showed that one-stage repair is feasible in all ARMs, and with meticulous selection, primary PSARP in neonates is safe. The virtually sterile meconium during the 1^st week of life reduces the risk of infection from faecal contamination. Many centres in developed countries have recorded multiple successes with primary PSARP in neonates.[18]

Regarding the current research, we performed 36 cases divided equally into two groups with 18 cases in each group, all performed without covering colostomy over 3 months and followed up over another 3 months at least. Kumar et al.[19] where 123 patients underwent primary repair either through ASARP (34) or PSARP (89) approaches, then a follow-up period ranging from 3 months to 7 years.

The classic techniques described by Okada et al.[7] and others were followed meticulously in which the anterior rim of the external anal sphincter is cut, as well as keeping the puborectalis sling untouched. However, some authors tried to modify such technique with equally acceptable results, including Elsawaf and Hashish[20] who introduced a new technical innovation that combines the advantages of ASARP with sphincter preservation and adequately positioning the rectum at the center of the anal sphincter under direct vision.

In our study, all patients included had the repair done at ages 3–12, with no significant variation among both groups, with a mean age of almost 7 months in both groups.

Conversely, research by Adeniran[13] involved four girls whose ages fell between 3 days and 1.5 years. In contrast, Mirshemirani et al. addressed[21] that all cases fell between 1 and 20 days during the neonatal period.

Kulshrestha et al.[9] decided to operate at 3 months of age for those who presented in the neonatal period; the surgery median age exhibited 10.5 months, regarding another research by Kuijper and Aronson.[16] The surgery median age exhibited 4 months (range: 0–73 months); about 34% were carried out within the neonatal period.

The adverse events of the surgical procedure were compared among our research groups; the predominant complication involved wound infection, with around 11 cases within one stage of repair.

In the ASARP group, there were 6 cases, about (33,3%) versus 5 patients (27,8%) within the PSARP group, addressing no significant variation among both groups.

Regarding Adeniran’s[14] research, a case undergoing PSARP developed a superficial wound infection around once. However, Mirshemirani et al.[21] addressed three cases of developing wound infections postoperatively.

Our result revealed that wound infection was significantly associated with longer TPN duration among infected wounds compared to the other group. Wound dehiscence was significantly associated with wound infection among both groups.

Some bowel preparation was done by keeping the patients NPO for 2 days preoperatively with IV fluids and antibiotics, flagyl, and ciprofloxacin; some of our patients developed minor accepted wound dehiscence and managed conservatively, and there was no major wound dehiscence that needed surgical repair or affected the postoperative continence.

In our case, wound dehiscence occurred slightly more in patients with PSARP than in ASARP cases, with four versus three instances. Kuijper and Aronson[16] mentioned that seven children (4 rectoperineal and three RVF) out of 22 girls in his study eventually healed after minor wound dehiscence. Amanollahi’s and Ketabchian[6] study showed that SSI rate and dehiscence reached (30%) within the stage limb, which was the same in total of both groups in our study.

Hospital stays did not differ significantly between both groups in our study. It was mainly influenced by wound infection and dehiscence, with the mean being 11 and 15 days in clean and infected wounds, respectively.

Regarding the prevalence of constipation, in our study, 19 patients developed postoperative constipation, 13 of them in the PSARP 65% group and 6 of them in the ASARP group 37.5%.

However, Peña and Devries[8] mentioned that constipation in the vestibular fistula can reach up to 50% of cases, much more than other types of ARM. The prevalence of constipation in the ASARP group (13 cases, 65%) was more frequent than in the PSARP group (6 cases: 33.3%) in contrast to Shehata[22] and Kulshrestha et al.,[9] which favors ASARP in regard of long-term complications rate.

Constipation with overflow was also recorded in 3 cases, 2 in ASARP and 1 in PSARP groups. There is no significant difference between both groups. Such cases were differentiated from incontinence or soiling by an abdomen plain X-ray to exclude fecal accumulation and managed by bowel management protocol by Peña.[12]

A total of three cases (8.3%) developed soiling, one within the ASARP group as well as two within the PSARP group, with no significant differences between both groups; however, one patient developed soiling in the research conducted by Mirshemirani et al.,[21] along with one patient in Kumar et al.[19]

To our knowledge, there is no objective study that demarcates the link between anal dilatation and the development of anal stricture postoperative. We found in our dissertation, to clarify this point, we found that more than half of the patients had standard anal size for age during the conclusion of the 3-month follow-up. None showed anal stenosis, indicating that anal dilatation is essential for prevention of anal stricture.

The limitations of our study included the fact that the sample size was relatively small. It was a single-center study. The period of follow-up was short. Hence, we recommended a long follow-up period to offer more robust insights into the effectiveness of ASARP versus PSARP.

CONCLUSIONS

The clinical implications of comparing PSARP and ASARP in treating RVF in female patients. ASARP may be a better option in low-resource settings, as it is associated with shorter hospital stays, fewer wound infections, and fewer complications than PSARP. Patients who underwent PSARP experienced more extended hospital stays, more diseases, and longer TPN durations. These findings support using ASARP as a viable alternative for achieving similar outcomes while reducing the burden on healthcare systems and families, particularly in resource-limited environments.

Conflicts of interest

There are no conflicts of interest.

Funding Statement

Nil.