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PubMed1992

Anterior sagittal anorectoplasty for rectovestibular and anovestibular fistula.

Rectovestibular (or anovestibular) fistula is the most common form of anorectal anomaly in female infants. In the surgical repair of these malformations, anal transplantation has been in widespread conventional use. Based on the idea of posterior sagittal anorectoplasty, we devised a new approach, anterior sagittal anorectoplasty, in which, in the lithotomy position, the anterior portion of sphincter muscles are cut through a median perineal skin incision and then the rectum is pulled through the center of these muscles to make its opening at the normal position. The operation has been performed in 12 patients with satisfactory results.

Anal Canal↗
PubMed1992

Cloacal exstrophy: prenatal diagnosis before rupture of the cloacal membrane.

Embryologically, cloacal exstrophy is thought to result from persistence and subsequent rupture of the infraumbilical cloacal membrane during the fifth embryonic week. We report a case of cloacal exstrophy in which a prenatal diagnosis was made prior to rupture of the cloacal membrane. A routine ultrasound at 17 weeks' gestation demonstrated monoamniotic twins. One twin was normal, but the other was found to have a sacral myelomeningocele, "rocker-bottom" feet, splaying of the pubic rami, and a large cystic mass protruding from the infraumbilical anterior abdominal wall. A repeat ultrasound was performed at 22 weeks, with the same findings. At 26 weeks, further examination showed disappearance of the abdominal cyst, a small omphalocele, no demonstrable bladder, and the suggestion of prolapsed bowel inferior to the umbilical cord insertion. After delivery at 34 weeks, the abnormal twin was found to have the typical findings of cloacal exstrophy, myelomeningocele, bilateral lower limb anomalies, and extremely foreshortened small bowel. Rupture of the presumed cloacal membrane after 22 weeks in this case is inconsistent with our current understanding of the embryology of this anomaly, and should stimulate a reexamination of the current concepts. If the characteristic features are recognized, cloacal exstrophy can be diagnosed by prenatal ultrasound, permitting prenatal counseling and appropriate perinatal management.

Abnormalities, Multiple↗
PubMed1992

Tracheoesophageal fistula associated with perforated Meckel's diverticulum.

Neonates with esophageal atresia and tracheoesophageal fistula (TEF) may present with respiratory distress. Intubation and mechanical ventilation may force air from the tracheobronchial tree, through a distal fistula and into the gastrointestinal tract. We present a newborn with TEF who became moribund during mechanical ventilation. High ventilator pressures transmitted via the TEF caused over distention of the gastrointestinal tract and perforation of a Meckel's diverticulum.

Abnormalities, Multiple↗
PubMed1993

Constipation is a major functional complication after internal sphincter-saving posterior sagittal anorectoplasty for high and intermediate anorectal malformations.

Anorectal function was clinically, manometrically, and radiologically followed-up in 40 patients who underwent an internal sphincter-saving posterior sagittal anorectoplasty in 1984 to 1989. The functioning internal sphincter was manometrically verified in 83% (33/40) of the patients. Soiling related to sphincter insufficiency was found in only 4 of the 33 patients with a functioning internal sphincter; 5 of the 7 patients without an internal sphincter had soiling. Symptomatic constipation was found in 73% (24/33) of the patients with a functioning internal sphincter, but in only 28% (2/7) of the patients without this structure. Constipation was not associated with a stenotic anal outlet in any patient. On anorectal manometry, there was no statistical difference in basal and pressures and internal sphincter reflex threshold values between constipated and nonconstipated patients. Histologically, ganglion cells were found in the proximal anal canal in all cases. The radiological size of the rectal pouch before closure of the protecting colostomy had a positive correlation with the severity of constipation. Medical treatment with diet and bulk-laxatives (13 patients) or with stimulant laxatives and occasional enemas (11 patients) successfully relieved constipation in 24 patients. In 2 patients, the symptoms were refractory to medical treatment and a resection of the megarectum was required. In conclusion, the preservation of the internal sphincter in patients with high or intermediate anorectal anomalies gives a good fecal continence outcome, but is associated with a high incidence of symptomatic constipation.

Anal Canal↗
PubMed1994

Dynamic graciloplasty for anal atresia.

The aim of this study was to assess whether an electrically stimulated graciloplasty (dynamic graciloplasty) can achieve continence in nine patients with anal atresia (median age, 28 years; range, 18 to 40). As the first procedure, a gracilis muscle was transposed. Six weeks later, intramuscular electrodes were implanted and connected to a pulse generator. Eventually, the muscle was gradually trained, by electrical stimulation, to achieve fecal continence. Continence was obtained in five patients (55%). Manometry demonstrated an increase in mean anal pressure, from 36 mm Hg (without stimulation) to 52 mm Hg (with stimulation), after 8 weeks (mean increase, 16 mm Hg, [95% confidence interval, 8, 24 mm Hg; n = 9; P < .01). Failures resulted from a noncontracting distal part of the gracilis muscle (in three patients) and a nondistending rectum (in one patient). We conclude that dynamic graciloplasty can achieve continence in a substantial number of patients with thus-far untreatable incontinence after surgical correction for anal atresia.

Adult↗
PubMed1994

Rectoperineal fistula in newborn boys.

Three newborn boys presented with features suggestive of classic low anorectal abnormality. However, during surgery they were found to have an intermediate anorectal abnormality and a rectoperineal fistula rather than an anocutaneous fistula. The surgical treatment of these infants is discussed.

Anus, Imperforate↗
PubMed1995

Spinal dysraphism detected by magnetic resonance imaging in patients with anorectal anomalies: incidence and clinical significance.

Though the concept of caudal regression, suggested in 1961 by Duhamel, could explain the association between anorectal anomalies (ARA) and spinal dysraphism (SD), its real incidence may be underestimated and its clinical significance is debatable. From 1988 to 1993, 111 patients with ARA were treated at the authors' institution. Associated anomalies were present in 36% of cases, with the exception of vesicoureteral reflux, which was considered functional rather than anatomical. In view of the late (1 to 2 years after surgical treatment) onset of vesical dysfunction and/or orthopaedic symptoms in some of these patients, a screening protocol was started in 1991, using magnetic resonance imaging (MRI) in all patients with ARA. Fifty patients, 29 boys and 21 girls, underwent a spinal cord MRI, with pathological findings in 25 cases (50%), 13 boys and 12 girls. The authors did not find any significant difference in incidence with respect to high, low, or cloacal malformations. A thickened filum, with or without fibrolipoma, was the most frequent finding, but even tethered cord, syringomyelia, and sac morphological alterations were present. MRI was also able to detect osteoarticular and/or muscular anomalies. Even when a urodynamic study and a neurological and orthopaedic workup were performed in 20 patients undergoing MRI, the clinical significance of these findings remained unclear. However, accurate follow-up of these patients is mandatory in order to detect early neurological symptoms, because currently it is not advisable to refer for neurosurgery all the patients with ARA presenting with anomalies of the spinal cord.

Abnormalities, Multiple↗
PubMed1995

Psychosocial adjustment of children treated for anorectal anomalies.

The psychosocial adjustment of 160 children with anorectal malformations was assessed at 6 to 17 years of age in relation to levels of continence (Kelly score). Five measures of emotional and behavioural adjustment were used to assess a number of domains of child/adolescent functioning and to include measures from multiple perspectives. The psychiatric assessment of the child identified a disorder in 29% of the group overall. Based on parental assessments, behavioural maladjustment was shown in 27% of the children, and on the basis of a self-report questionnaire 24% of the children were depressed. Behavioural adjustment as rated by teachers was similar to the norms. The level of continence achieved (total Kelly score) did not appear to influence psychological adjustment, with the exception of the incontinent young girls (6 to 11 years) who were shown to be less well adjusted than the continent young girls. Differences between children showing positive versus negative adjustment were dependent on the perspective of the respondent and were also related to the child's age and gender, age of achieving continence, frequency of accidents, and parental factors.

Adaptation, Psychological↗
PubMed1995

Epidermolysis bullosa associated with pyloric, esophageal, and anal atresia: a case report.

The association of epidermolysis bullosa (EB) and pyloric atresia (PA) in newborns is rare but has been well described. It has been reported that in patients with EB, the esophageal, gastric, intestinal, and colonic mucosae are affected. The authors report a case of EB and PA associated with esophageal and anal atresia in a newborn. To our knowledge, these associations had not been reported in the English-language literature.

Abnormalities, Multiple↗
PubMed1995

Mucosal proctectomy and colo-anal anastomosis as treatment of rectal atresia.

Rectal atresia is a rare condition in which the anus and sphincter muscles are normally developed. Therefore it seems logical to preserve those structures unaltered during surgical therapy. Mucosal proctectomy and colo-anal anastomosis is a safe, technically well known technique that achieves this goal.

Anal Canal↗
PubMed1995

Anal agenesis associated with rectal atresia.

The authors report an unusual case of anal agenesis associated with rectal atresia. Such an anomaly has been reported only once before. Coincidentally, this patient also had a rarely encountered skeletal anomaly--unilateral cleft foot.

Abnormalities, Multiple↗