Results of anoplasty can be evaluated by objective and subjective methods. The objective method is less favorable now because this method is expensive and may be unavailable, especially in developing countries. Objective methods include manometry, computed tomography, MRI, anal sphincter myography, and intra-anal sonography 1. There are several scoring systems suggested by Kelly 2, Templeton and Ditesheim 3, Kiesewetter and Chang 4, Holschneider 5, and Rintala and Lindah 6. The Kelly score 2 requires rectal examination by hand and the Holschneider 5 score requires anorectal manometry. The Rintala score does not require physical examination and is a purely subjective method 6. The aim of this study was to evaluate the outcome and quality of life among patients with low or high anorectal malformation (ARM) who underwent posterior sagittal anorectoplasty (PSARP).
Patients and methods
Sixty patients (male 32, female 28) who underwent standard PSARP were included in this study. These patients underwent surgery during the past 1–15 years. Patients with mental retardation were excluded from our study. We used low and high ARM definitions according to the relationship of the terminal colon to the levator muscles of the pelvic floor 7. After surgery, parents were educated on the dilatation program. Dilatation was performed for each patient by parents according to the age of the patient after 2 weeks of repair. The protocol of dilatation used for patients is shown in Table 18. The outcome of cases was assessed using the validated bowel function score (Table 2).
Patients were classified according to their scores into four categories: poor (6–9); fair (9–11); good (12–17); and normal (18–20). We used a questionnaire introduced by Rintala and Lindah 6. Voluntary control, sensation, soiling, stool frequency, constipation, and the social impact of constipation were assessed by the Rintala score 9. The Student t-test, the Pearson χ2-test, one-way analysis of variance, and the Levine test were used for data analysis using SPSS (ver. 13.0; SPSS Inc., Chicago, Illinois, USA). The person who filled the questionnaire was not a member of the medical team. This study was approved by the ethical committee of the university.
Sixty patients aged 3–17 years (13.63±3.27 years) were included in this study (Table 3). Of the patients, 32 (53.33%) were male and 28 (46.67%) were female (P=0.46). Of them, 29 patients (48.4%) were 3–12 years old and 31 patients were more than 12 years old. The mean of age was 10.77±2.94 years. Out of 60 patients, 29 (48.33%) had low anomaly and 31 (51.67%) had high anomaly (P=0.71).
The mean of scores among all patients was 13.63±3.27. The minimum and the maximum of scores were 7 and 18, respectively. The mean of scores in patients with low-type ARM was 14.5±2.6 and in patients with high-type ARM was 13.19±3.75. There was no significant difference between the two groups (P=0.28).
The mean of scores was 13.34±3.5 among male patients and 13.94±2.9 among female patients. There was no statistically significant difference (P=0.46).
There was no significant correlation between the score and the age of the patients (Pearson coefficient=0.13). The score was significantly higher among patients with fistula (n=51, 13.94±3.19) than among patients without fistula (n=9, 11.8±3.3; P=0.03). Excluding two cases with scrotal-type fistula and rectal atresia, there was no significant difference between the two groups (P=0.06). The average of scores among different types of ARMs is shown in Table 4. The minimum (score=7) score was seen in a patient with rectal atresia and the maximum (score=16) was seen in a patient with scrotal fistula (Table 4).
Poor prognostic cases were seen in 16.12% of patients with high ARM and 3.45% of patients with low ARM (P=0.22). Of 31 patients with high anomaly, 17 (54.83%) had good prognosis. Of 29 patients with low anomaly, 21 (72.41%) had good prognosis (P=0.15, χ2=1.99). There was no significant difference between high and low ARM regarding their prognosis evaluated by the Rintala score (Table 5).
Fecal soling and some degree of fecal incontinence were seen in 67 and 31.7% of the cases, respectively.
In our study, the male/female ratio was 1.14/1. In the study from South Africa, the male/female ratio was 1.6/1 10. In the study from Pakistan, of 100 neonates with ARM, 77 were male and 23 were female (male/female=3.4/1) 11. Hence, boys are affected more often than girls in these studies.
In the study carried out by Goyal et al. 12, the best prognosis was reported in cases with perineal fistula and poor prognosis was seen in vesical fistula. These findings were similar to our study. In the study by Hassett et al. 13, the best prognosis was reported in cases with perineal followed by vestibular and urethral fistula. In their study, a posterior sagittal approach was used for treatment. A questionnaire was used for the scoring system.
In the study of Kaselas et al. 14, the highest score was seen in patients with perinanal fistula. In our study, the highest score was seen in cases with vaginal fistula (15.1±2.3) followed by perineal fistula (14.6±2.3). Vesical fistula had the poorest prognosis in the study of Kaselas et al. 14 and in our study.
Rintala et al. 9 compared children with low ARM with normal healthy children. They concluded that only half of the children with a low ARM have age-appropriate normal bowel function.
In our study, there is no significant score improvement with increasing age. In some studies, as age increased, there was improvement in their score 9.
Hassink 15 conducted a study on patients with low-type anomaly. Of the patients, 83% showed good prognosis and 15% had fair and poor prognosis. Our findings are similar. In the study of Hassink, there was a positive correlation between prognosis improvement and increasing age 15. In our study, there was no significant correlation between age and prognosis improvement.
In our study, the most common complication after surgery was constipation. Other studies also reported constipation as the major postoperative complication in patients who underwent PSARP 16,17. The incidence of constipation has been reported to be from 10 to 73% in patients who underwent PSARP 16,18. In the study of Rintala et al. 9, constipation was present in 42% of the patients.
In our study, 31.7% of the patients had fecal incontinence. Elhalaby 19 reported that incontinence was present in 33.3% of their 38 patients. In the study of Elhalaby, 18 out of 38 patients were more than 3 years old. In our study, all patients were more than 3 years old. Improvement in constipation in patients with increasing age was reported by Rintala and Linadhl 20. Hence, constipation is expected to resolve in our patients when they reach adolescence. In the study by Ibrahim 21, of 23 neonates whose follow-up periods were longer than 3 years, 21 neonates had a good score using the Kiesewetter score 4.
Constipation is higher in our study than in other studies. The number of patients with normal sphincteric function is lower than in other studies. There was no significant difference between boys and girls. There was no significant difference between low-type and high-type ARM. The mean of scores was significantly higher among patients with fistula than among patients without fistula. Careful follow-up and parent education are also recommended to achieve a better outcome.
The source of data used in this paper was the general surgery residency thesis of Dr Seyed-Masood Mousavi; Thesis No: D-218 financial support was provided by research affairs of Ahvaz Jundishapur University of Medical Sciences.
Conflicts of interest
There are no conflicts of interest.
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