|Year : 2018 | Volume
| Issue : 3 | Page : 148-152
Revision surgery in the management of anorectal malformations: Experience from a tertiary center of India
Basant Kumar1, Vijai Dutta Upadhyaya1, Manish Kumar Gupta1, Srinivasa Kishore1, JB Nijagal Mutt1, Rajanikant Yadav2, Sheo Kumar2
1 Department of Paediatric Surgical Superspeciality, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Raibareli Road, Lucknow, Uttar Pradesh, India
2 Department of Radiodiagnosis, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Raibareli Road, Lucknow, Uttar Pradesh, India
|Date of Web Publication||4-Jul-2018|
Dr. Basant Kumar
Department of Paediatric Surgical Superspeciality, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Raibareli Road, Lucknow - 226 014, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Aim and Objectives: Despite the significant advancements in the management of anorectal malformations (ARMs), there are various surgical and functional complications reported. Complications are closely related with the surgical techniques adopted and the types of malformations. In this article, we present our experiences with ARM patients who required reoperation after unsuccessful previous surgeries or who had developed complications related to the previous surgical techniques.
Materials and Methods: We retrospectively reviewed clinical and electronic records of all the patients with ARM who were operated for ARMs in our institute from June 2010 to May 2016. All ARM patients who needed reoperation were included in the study. These patients were previously operated outside our institute and referred to us with ongoing problems of constipation, stool impaction with overflow incontinence, perineal soiling, and difficult urination.
Results: There were 31 patients (M:F = 2.1:1) of ARM, reoperated for 38 indications during the above-mentioned period. Five patients had more than one problem. Presentation included neoanal stenosis (11), complete obliteration of neoanus (2), malpositioned neoanus (2), persistent/recurrent rectourethral fistula (3), iatrogenic rectovaginal fistula (4), rectal prolapse (5), large widened neoanus with soiling (2), and urethral stricture (2), which required revision interventions. Six patients had megarectum. All patients showed improvement in their symptoms after revision surgery, but 10 (41.7%) patients required further regular bowel management program (BMP) to avoid the soiling and constipation. Fourteen (58.3%) patients stayed clean without regular BMP.
Conclusion: All these complications had clear explanations and are well described in the literature. Revision surgery in such patients had fair outcome, but some sort of BMP was required. Both posterior sagittal anorectoplasty and anterior sagittal anorectoplasty are excellent techniques for revision surgery with few simple modifications.
Keywords: Anorectal malformation, anterior sagittal anorectoplasty, complication, posterior sagittal anorectoplasty, revision surgery
|How to cite this article:|
Kumar B, Upadhyaya VD, Gupta MK, Kishore S, Nijagal Mutt J B, Yadav R, Kumar S. Revision surgery in the management of anorectal malformations: Experience from a tertiary center of India. J Indian Assoc Pediatr Surg 2018;23:148-52
|How to cite this URL:|
Kumar B, Upadhyaya VD, Gupta MK, Kishore S, Nijagal Mutt J B, Yadav R, Kumar S. Revision surgery in the management of anorectal malformations: Experience from a tertiary center of India. J Indian Assoc Pediatr Surg [serial online] 2018 [cited 2020 Nov 25];23:148-52. Available from: https://www.jiaps.com/text.asp?2018/23/3/148/235889
| Introduction|| |
Despite the significant advancements in the management of anorectal malformations (ARMs), various surgical and functional complications are known, which are closely related with surgical techniques and types of malformation., These complications are relatively more frequent because of high incidence of ARMs and its wide spectrum of presentations.,, Unfortunately, most of these complications are preventable. Consequences of complications are significant and can lead to prolonged morbidity, which may require revision surgery and result in suboptimal outcome. Revision surgery is needed for various reasons but mostly attempted because of inadequate results related with bowel or urinary control after first surgery or because of catastrophic complications related with surgery to alleviate pain, discomfort, or other sequelae.,,, Various indications for redo surgeries have been described in the literature.,,
Here, we retrospectively reviewed our patients with ARMs who required reoperation/interventions after unsuccessful/inadequate previous surgeries or who had developed complications related to surgical techniques. We also tried to analyze the associated problems and pitfalls of revision surgery contributing further to suboptimal results.
| Materials and Methods|| |
We retrospectively reviewed clinical and electronic data, operation theater, and follow-up records of all patients operated for ARMs from July 2010 to May 2016. Data collected were pertaining to the age, sex, clinical presentations, complications, diagnostic investigations, associated anomalies, treatment, outcomes, and follow-up.
All patients needing revision surgery for previous suboptimal results or who had developed complications after previous surgery for ARMs and were followed up for >3 months were included in this study.
Patients whose records were deficient or who could not be followed up for >3 months were excluded from the study.
All patients included in the study were from a single unit of our department, a tertiary level pediatric surgical referral center. All patients were operated outside initially, and almost none had proper records of previous surgery. Clinical presentations at the time of referral were ongoing constipation, stool impaction with overflow incontinence, fecal and urinary soiling, perineal excoriation, difficult defecation or urination, and mucosal prolapse. After initial management, all patients were evaluated to delineate the anatomy and associated anomalies. Patients were reoperated and followed up according to the method established by Krickenbeck in 2005 for the assessment of outcome. We routinely used some sort of bowel management program (BMP) in the first month of follow-up and anal dilatation for 2–3 months or till stoma closure. We maintained follow-up cards of each patient by asking leading questions and recorded the complaints/satisfaction of the parents and patients.
| Results|| |
From July 2010 to June 2016, 31 patients with ARM were included in the study. Ten patients were female (M:F = 2.1:1). The age of patients varied from 1.5 months to 16 years (median age 8 years). Revision surgery/intervention was performed in all 31 patients for 38 indications. Five patients had >1 complaint. Individual patients were evaluated and investigated according to the presentations/complications. Magnetic resonance imaging (MRI) pelvis was performed in two patients, while two others already had computed tomography (CT) pelvis at the time of presentation. Six patients had dilated distal colon (megacolon) and three male patients had persistent rectourethral fistula on barium enema and distal loopogram. Indications for revision surgery/intervention were neoanal stenosis (11), complete closure of neoanus (2), malpositioned neoanus (2), persistent fistulae (3), iatrogenic rectovaginal fistulae (4), rectal mucosal prolapse (5), large wide neoanus with soiling (2), and urethral stricture with urinary retention (2) [Figure 1] and [Figure 2]. Associated anomalies were left renal agenesis (1), Vesico ureteric reflux (2), hypospadias (1), Undescended testes (1), and hemivertebrae (1). All patients were operated outside our institute for intermediate-to-high ARMs and referred to us with above-mentioned problems [Table 1] and [Table 2]. Redo surgery/interventions performed were V-Y anoplasty, posterior sagittal anorectoplasty (PSARP), anterior sagittal anorectoplasty (ASARP), abdominoperineal anorectoplasty, mucosectomy, minimum PSARP and cystoscopy, and urethral dilatation followed by clean intermittent catheterization [Table 2]. All patients showed improvement in their symptoms after redo surgery. Constipation and soiling were resolved/reduced with decreased need of laxatives and rectal washouts (manageable).
|Figure 1: Complete obliteration of neoanus (a); malpositioned neoanus with stenosis (b); and large wide neoanus created by dilated pulled colon (c) after posterior sagittal anorectoplasty during the first surgery|
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|Figure 2: Magnetic resonance imaging (a) showing right side deviation of pulled bowel (arrowhead) and thinning of right upper part of external sphincter (arrow); and contrast study showing rectourethral fistula (b)|
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|Table 1: Detail of patients regarding primary pathology and surgery performed outside|
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|Table 2: Indications, redo procedures performed, complications, and outcome of patients (n=31)|
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Postoperative problems (<1 months) were superficial wound infection in 8 (25.8%) out of 31 patients, perineal excoriation in 4 patients (12.9%), and neoanal stenosis in 1 patient (3.2%). Child with neoanal stenosis needed further surgery later. Follow-up ranged from 3 months to 5 years (median follow-up 26 months). Four out of 31 patients were <3 years, while 3 patients were lost in follow-up. The rest 24 patients (100%) had voluntary bowel movements with feeling of urge and the capacity to verbalize and hold the bowel movement. Constipation (Grades I and II) was observed in 9 (37.5%) patients and fecal soiling (Grades I and II) in 4 (16.7%) patients (no social problem). Almost all patients needed some sort of BMP in the form of dietary change, laxatives, and rectal washouts in first 3 months after surgery, while 10 (41.7%) patients still required regular BMP to avoid the soiling and constipation. Fourteen (58.3%) patients stayed clean without regular BMP, but almost all these patients had soiling incidents during stress (diarrhea/constipations) and approximately 50% of them needed laxatives/washouts, on on-off basis.
| Discussion|| |
Bowel control is the main concern in ARM surgery, but urinary control and sexual function should also be taken into consideration. Understanding the spectrum of anomaly and anatomy is necessary to prevent damage during surgery., Delineation of anatomy before redo surgery is quite essential to reduce postoperative morbidity. CT scan and MRI are indicated for this purpose in complicated cases.,,,, We prefer MRI pelvis over CT scan in patients with mislocated neoanus to see the lie of whole pulled bowel in relation to the sphincter-muscle complex. Placing of mislocated bowel within the sphincter–muscle complex provides better bowel control. MRI gives better visualization of soft tissues and relation of pulled bowel with perineal musculature including sphincter–muscle complex. Simultaneously, we can detect vertebral and spinal cord anomalies better.
All these complications presented in this series have clear explanations and are well described in the literature.,,,, Pena et al. described largest series of 334 patients for reoperation in ARMs and divided the patients into various groups. Follow-up results are described in each group separately. Our series has lower enrolled patients and cannot be compared to Pena's series. It has limited representation of spectrum of ARMs. Most of the patients in our series belong to Pena's pseudoincontinent group. This group is thought to provide better functional results after revision surgery, but our experiences do not measure up to our self-satisfaction. We performed standard ASARP/PSARP with some simple modifications.,,,
We experienced difficulty in positioning the patients for surgery in adolescents. Creation of neoanus during revision surgery should be little pouting after adequate mobilization. Application of side sutures over pulled bowel with adjacent muscles should also be avoided. Only mucocutaneous sutures should be placed during anoplasty. Terminal pulled bowel attains its position by itself after closure of sagittal incision because of significant peristalsis Placing fixation suture over it makes it tube-like rigid, nonperistaltic bowel. We agree that terminal 2–3 cm of pulled bowel always behaves like a tube after surgery, regardless of fixation sutures, but we experienced better functional results in patients without fixation sutures. This finding further needs validation by a larger, comparative study and probably by manometric/electromyographic studies.
We also experienced a patient with neoanal stenosis after revision surgery. We diagnosed it in the 1st month of surgery and tried to dilate it repeatedly even under general anesthesia but failed. The patient required minimal PSARP later. It occurred because of vascular injury during redo surgery.,,, Perineal excoriation was found in patients who lost their rectosigmoid after surgery but managed by perineal care and early institution of BMP., Although all patients in this series had voluntary bowel movements, almost all of them needed some sort of BMP for variable periods of time. Approximately 10 (41.7%) patients still required it to avoid soiling and constipation in the form of on and off rectal washouts and laxatives.
Anal dilatation after revision surgery should be monitored seriously, especially in children above 5 years. We found it difficult because of developed gluteal folds and noncooperation of children, probably because of comparatively more pain during anal dilatation. Liberal use of diverting stoma is recommended in complex/complicated cases to prevent postoperative complications. It should not be considered as a social stigma. We had low thresholds for stoma creation and created a stoma when there was suspicion of iatrogenic injury, compromised vascularity of pulled bowel or when we repaired a rectourethral fistula.
This study has various limitations that include a small series, retrospective nature, and selection bias. There was limitation in follow-up too. We do not have fixed questionnaires or scoring system for follow-up. Results were based on subjective opinion of parents and patients and on their satisfactions. Almost all patients of this series were from remote areas and belonged to lower and lower middle-class families. Mostly mother of variable intelligence took the care of patient and there was frequent change of care givers during follow-up that made final assessment difficult. Hence, we had to resort to leading questions about voluntary bowel movements, soiling, constipation (as per Krickenbeck recommendations), stool and feeding habits, frequency and quantity of use of laxatives and rectal washouts and enemas, etc. We modified treatment according to their satisfaction.
| Conclusion|| |
Adequate definitive repair at first attempt gives the best result. Revision surgery has promising functional results in group of patients with ARMs with few simple modifications. Management of every patient should be individualized according to the anatomy and complications. BMP should be instituted early in the patients with suboptimal results.
We would like to thank all the patients, their parents, technicians, and paramedical staff for their support.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Pena A, Levitt MA. Anorectal malformations. In: Grosfield JL, O'Neil JA, Fonkalsrud EW, Coran AG, editors. Pediatric Surgery. 6th
ed. Philadelphia, PA, USA: Mosby Elsevier; 2006. p. 1566-89.
Peña A, Grasshoff S, Levitt M. Reoperations in anorectal malformations. J Pediatr Surg 2007;42:318-25.
Rosen NG, Hong AR, Soffer SZ, Rodriguez G, Peña A. Rectovaginal fistula: A common diagnostic error with significant consequences in girls with anorectal malformations. J Pediatr Surg 2002;37:961-5.
Peña A, Hong AR, Midulla P, Levitt M. Reoperative surgery for anorectal anomalies. Semin Pediatr Surg 2003;12:118-23.
Davies MC, Creighton SM, Wilcox DT. Long-term outcomes of anorectal malformations. Pediatr Surg Int 2004;20:567-72.
Peña A, Hong A. Advances in the management of anorectal malformations. Am J Surg 2000;180:370-6.
Nakayama DK, Templeton JM Jr., Ziegler MM, O'Neill JA, Walker AB. Complications of posterior sagittal anorectoplasty. J Pediatr Surg 1986;21:488-92.
Holschneider A, Hutson J, Peña A, Beket E, Chatterjee S, Coran A, et al.
Preliminary report on the international conference for the development of standards for the treatment of anorectal malformations. J Pediatr Surg 2005;40:1521-6.
Peña A, Devries PA. Posterior sagittal anorectoplasty: Important technical considerations and new applications. J Pediatr Surg 1982;17:796-811.
Pena A. Anorectal malformations. In: Ziegler M, Azizkhan RG, editors. Operative Pediatric Surgery. New York, USA: Gauderer & Weber, McGraw-Hill; 2002. p. 739-62.
Okada A, Kamata S, Imura K, Fukuzawa M, Kubota A, Yagi M, et al.
Anterior sagittal anorectoplasty for rectovestibular and anovestibular fistula. J Pediatr Surg 1992;27:85-8.
Wakhlu A, Pandey A, Prasad A, Kureel SN, Tandon RK, Wakhlu AK, et al.
Anterior sagittal anorectoplasty for anorectal malformations and perineal trauma in the female child. J Pediatr Surg 1996;31:1236-40.
Boemers TM, Ludwikowski B, Forstner R, Schimke C, Ardelean MA. Dynamic magnetic resonance imaging of the pelvic floor in children and adolescents with vesical and anorectal malformations. J Pediatr Surg 2006;41:1267-71.
Peña A, Guardino K, Tovilla JM, Levitt MA, Rodriguez G, Torres R, et al.
Bowel management for fecal incontinence in patients with anorectal malformations. J Pediatr Surg 1998;33:133-7.
Peña A, el Behery M. Megasigmoid: A source of pseudoincontinence in children with repaired anorectal malformations. J Pediatr Surg 1993;28:199-203.
Bhat NA, Grover VP, Bhatnagar V. Manometric evaluation of postoperative patients with anorectal anomalies. Indian J Gastroenterol 2004;23:206-8.
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[Table 1], [Table 2]