|Year : 2019 | Volume
| Issue : 1 | Page : 45-51
Experience with the redo pull-through for hirschsprung's disease
Devendra K Gupta, Kashish Khanna, Shilpa Sharma
Department of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi, India
|Date of Web Publication||19-Dec-2018|
Prof. Devendra K Gupta
R No: 4002, Department of Pediatric Surgery, All India Institute of Medical Sciences, New Delhi
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Aim: This study aims to evaluate the need of Redo pull-through (Re PT) procedures for Hirschsprung's disease (HD) and suggest preventive strategies.
Materials and Methods: Patients who underwent redo procedures for HD from 1980 to 2016 by a single surgeon were retrospectively reviewed.
Results: Of 167 patients operated for HD, 32 underwent Re PT; 7 were previously operated by the same surgeon, while 25 were referred from outside. Indication for Re PT included residual disease including the rectal pouch following-Duhamel (12), false-negative biopsy (3), retraction of bowel (5), anorectal stricture (2), bowel twist (1), cuff inversion (2), postmyectomy continued symptoms after primary PT (1), fecal fistula (1), Re PT after surgery for adhesive intestinal obstruction (3), bleeding (1), and combination of causes, including scarred perineum (1). Age at follow-up ranged from 2.5 to 26 years. Proximal diversion was performed in 19 and 14 underwent open Scott Boley's/Soave PT and 5 ileoanal anastomosis. Of the remaining, nondiverted 13 patients, 5 underwent transanal endorectal PT and 8 underwent PT of colostomy.
Conclusions: Most patients of Re PT came after an initial Duhamel's procedure. Retraction of bowel, inversion of cuff, twist, distal bowel stricture, and perianal fibrosis were found after Scott Boley procedure. Proper planning with an initial diversion, nutritional buildup, barium study evaluation, frozen section facility, experienced pathologist, and an expert surgeon are prerequisites for a successful outcome after an initial as well as Re PT.
Keywords: Duhamel procedure, Hirschsprung's disease, redo pull-through, Scott Boley procedure, Soave's procedure, Swenson's procedure
|How to cite this article:|
Gupta DK, Khanna K, Sharma S. Experience with the redo pull-through for hirschsprung's disease. J Indian Assoc Pediatr Surg 2019;24:45-51
|How to cite this URL:|
Gupta DK, Khanna K, Sharma S. Experience with the redo pull-through for hirschsprung's disease. J Indian Assoc Pediatr Surg [serial online] 2019 [cited 2020 Apr 5];24:45-51. Available from: http://www.jiaps.com/text.asp?2019/24/1/45/247907
| Introduction|| |
The basic principle of treatment of Hirschsprung's disease (HD) is resection of the aganglionic bowel, followed by the pull-through (PT) of the ganglionic segment. The PT procedures can be either of the modifications of the originally described Soave, Swenson, and the Duhamel procedure.,,, Most patients do well in the long term, however, some may complain of persistent postoperative symptoms such as obstruction, constipation, incontinence, and recurrent Hirschsprung's-associated enterocolitis (HAEC).,,, Most persistent symptoms resolve with nonoperative procedures such as bowel management and behavioral modification. Redo PT (Re PT) is required in certain situations, namely, persistent aganglionosis most commonly due to incomplete resection of the transition zone (TZ), long spur and pouch, retraction of the bowel, anastomotic stricture, and rigidity of the anorectal cuff and fistulas.,,,,,
The aim of this study was to determine the need for redo surgery in 32 patients, who required Re PT for HD over a period of 37 years; to assess the type of Re PT procedure performed; to comment upon the best technique for Re PT; and finally to share the experience gained, while dealing with cases of Re PT.
| Materials and Methods|| |
A retrospective review of patients, who underwent Re PT for HD by a single surgeon from 1980 to 2016 was performed. A total of 167 cases were operated for HD during this period, out of which 32 cases were of Re PT. The causes for Re PT, histology after initial surgery, the type of the initial procedure, the type of Re PT, the postoperative complications, and follow-up of the 32 patients were recorded by reviewing clinical case records.
| Results|| |
Out of 32 patients, who underwent Re PT procedures, 25 (78.1%) patients were initially operated elsewhere and were referred to us from outside. These included four patients, who were operated multiple,,,,,, times previously. The rest of the 7 (21.9%) patients were previously operated at our institute by the same surgeon. There were 26 (81.2%) boys and 6 (18.8%) girls with a male: female ratio was 4.3:1. The mean age at follow-up was 3.6 years (range 2.5–26 years) and the mean age at redo surgery was 5.2 years.
Twenty (62.5%) patients complained of persistent obstructive symptoms, 4 (12.5%) of persistent constipation not managed conservatively, 5 (15.6%) patients were diagnosed to have bowel retraction, 2 (6.25%) had recurrent episodes of acute HAEC, and 1 (3.12%) had fecal fistula.
Nineteen (59.4%) patients required a stoma in the form of ileostomy in 6 and colostomy in 13, before reoperation. Eight out of 19 patients already had a stoma fashioned elsewhere before being referred to us for further management. In the remaining 11, an enterostomy was performed for following reasons. Large, dilated bowel with multiple or hard fecalomas which hindered the decompression of the normal bowel segment in 7 patients [Figure 1]. In two cases with recurrent HAEC not responding to antibiotics and flatus tube decompression, multiple admissions were required within a short span leading to failure to thrive; a diverting stoma was performed. Third, in the other two patients with the leakage of the anastomosis and the pelvic floor fibrosis, stoma was made to tide over the period of acute crisis before a redo surgery could be undertaken.
|Figure 1: (a) Barium study showing hugely dilated distal colon post completion of all stages of surgery for Hirschsprung's disease and (b) clinical picture showing grossly distended abdomen of the same child, thus requiring colostomy|
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Eleven of 19 (57.9%) patients with stoma had complications which included prolapse, retraction, skin excoriation, and stomal stenosis. A waiting period of at least 6 weeks was observed before the definitive radical surgery was planned. Five patients with dilated bowel could be managed well with repeated bowel washes and could be taken up for a Re PT after about 2 weeks, avoiding the need for a diverting stoma.
Type of Hirschsprung's disease
Of the 32 cases for Re PT, 28 had a previous classical rectosigmoid type of HD, 2 had a long-segment HD, and 2 had total colonic aganglionosis (TCA).
Previous failed surgery
Of the 32 cases requiring redo procedures, 24 (75%) had undergone Duhamel procedure previously, 6 (18.8%) had undergone an initial Scott Boley procedure, and 2 (6.2%) were postprimary Swenson PT. Four patients had been operated multiple times (3–9 times), previously before being referred.
Indications for redo pull-through
The indications for Re PT procedures were residual disease in 12 (37.5%) which also included two patients with large residual rectal pouch post Duhamel, false-negative biopsy for HD in 3 (9.4%), retraction of bowel in 5 (15.6%), adhesive intestinal obstruction in 3 (9.4%), anorectal stricture in 2 (6.3%), rectal stricture due to cuff inversion in 2 (6.3%), bowel twist 1 (3.1%), postmyectomy persistence of symptoms after PT 1 (3.1%), bleeding in 1 (3.1%), fecal fistula in 1 (3.1%), and a combination of causes in 1 (3.1%).
Surgical technique for redo pull-through
The type of procedure for Re PT was tailored to the problem as per case to case basis depending on the type of presentation (emergency/elective), the duration of symptoms, nutritional status of the child, presence of fecalomas, bowel dilatation, available length of the residual colon, site of stoma, the cause of persistent obstructive symptoms, and most importantly, the surgeon's experience in a particular technique. Re PT procedures included an open ERPT Soave's/Scott Boley type of PT in 22 (68.8%), an ileoanal type PT in 5 (15.6%), and a transanal endorectal PT in 5 (15.6%) [Figure 2].
|Figure 2: Chart showing the type of redo pull-through procedures performed in cases (n = 32)|
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The most commonly performed redo surgeries were an open modified Scott Boley's procedure due to the surgeon's previous experience and the wide access it gave to assess the problem. In this procedure, after a thorough bowel preparation, a digital dilatation of the anal canal was performed. Thereafter, the anal mucosa was meticulously scored at least 1.5 cm proximal to the dentate line. A mucosal tube was scored out, creating a long cuff of about 6–8 cm. The previous inguinal skin crease incision was utilized in most if only the distal bowel were to be handled. However, a midline abdominal incision was preferred for the Re PT in complicated cases with suspected long HD requiring more proximal bowel mobilization. The frozen section biopsy was taken at multiple desired sites, the aganglionic, narrow, or the strictured segment of the bowel was resected and the remaining normal proximal bowel segment was adequately mobilized. Adhesions with bowel loops were usually found and released with patience. Colostomy, if required, was mobilized to gain the length to have a tension-free anastomosis of the pulled-down ganglionic bowel to the perineum. Ureters were identified and carefully protected. Retraction of the bladder was gentle. Dissection remained intraluminal within the muscular cuff. Care was taken to avoid ischemia and/or rotation of the pulled-down bowel segment. A frozen section was again performed to confirm the presence of ganglion cells in the most distal segment of the PT segment. The muscular cuff was slit open posteriorly in midline to avoid constricting effect on the pulled bowel. Excess length of the cuff, if any, was removed. Three or four fixation sutures were taken to fix the edges of the cuff with the pulled bowel. The mesentery was tucked in the retroperitoneum to prevent internal herniation. An end-to-end single-layer anastomosis was performed at the anal verge 1–1.5 cm above the dentate line.
In patients with total colon aganglionosis requiring ileoanal PT, the muscular cuff was intentionally kept a little longer (8–10 cm) and the anastomosis was performed about 2 cm above the dentate line (with ileal segment), to create a little more resistance so as to decrease the frequency of stools after surgery.
In three cases, an original Soave was performed due to the absence of mucosal lining and the presence of extensive pelvic fibrosis, leaving the healthy ganglionic bowel hanging outside the anal verge for about 15 days. This allowed the adhesions to form between the pulled bowel and the cuff and also retract the bowel without the risk of peritonitis. Trimming of the excess bowel was done after 2–3 weeks after ensuring healing.
Aganglionosis was confirmed intraoperatively in 17 (53.1%) by a frozen section biopsy, in cases primarily reoperated due to residual or acquired aganglionosis. In the remaining ones, other causes for the redo surgery were identified. A few of these cases are detailed below.
A child presented with repeated constipation after all stages of HD being completed. There was a history that the saline instilled for rectal washes went in easily but did not come out. The per-rectal examination was normal. A barium study revealed an obstruction with a cutoff at a lower down pelvic level. A bowel rotation was suspected. Intraoperatively, there was rotation of the pulled bowel. The gut was derotated and redo anastomosis was performed keeping all the vessels intact. Postoperative outcome was excellent.
Another child presented with constipation, not managed by conservative treatment post-Soave's PT surgery. Barium study revealed a constricting ring at the muscular cuff level [Figure 3]. At laparotomy, the constriction ring was divided vertically at three places and the edges of the rim were excised. Postoperative dilation was done. The child was relieved of symptoms postprocedure. Barium study showed resolution of the dilated bowel segment [Figure 3].
|Figure 3: (a) Barium study of a previously performed Scott Boley's procedure, showing obstruction like a constricting ring in the region of seromuscular cuff and (b) resolution of the obstruction postdivision of the ring at multiple levels and excision of the cuff from the abdominal route|
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A child with colostomy was referred from elsewhere with retracted PT bowel visible on clinical examination and on barium study. In this case, the short retracted segment of bowel distal to the colostomy was discarded, the proximal bowel adequately mobilized and a redo anastomosis was performed without a covering stoma. In eight patients, the colostomy itself was pulled-down to achieve coloanal anastomosis without a covering stoma.
In two cases, only the ascending colon was left, and hence, the residual colon was very short and not found sufficient enough to reach the anal verge; thus a modified approach using an isolated ileal segment on its vascular pedicle was used to bridge the gap. This preserved the segment of the residual ascending colon and the ileocecal junction for absorption of water and essential nutrients such as Vitamin B12, bile salts, bile acids, and cholesterol. Care was taken to prevent ischemia of the interposed ileal segment during and after surgery.
The child with fecal fistula presented at 12 years of age after completing all stages of surgery for HD by Duhamel technique, with a dilated, fibrotic, patulous anal opening without any mucosal lining. There was complete loss of bowel and external sphincter [Figure 4]. She underwent a proximal diversion followed by an open Soave's type PT.
|Figure 4: (a) Barium study showing stricture in the region of anorectum and (b and c) clinical pictures of the patient (12-year-old girl) presenting with fecal fistula, patulous anal opening, and loss of anal sphincter control|
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A child had undergone transanal PT for a suspected rectosigmoid variety of HD at 3 months of age. The frozen biopsy had confirmed the presence of ganglion cells at the lower end of the PT bowel. However, after an initial asymptomatic period for several months, the child again developed symptoms of intestinal obstruction. Barium study was suggestive of TCA which was confirmed on laparotomy. A Martin's modification with Scott Boley PT using the left colon patch (15 cm) was performed at 9 months of age, with success. Postoperatively, the child had extensive perineal excoriation with frequency of stools, painful holding and fecal incontinence, requiring intensive management for several years needing repeated hospitalizations for anemia and pain relief. He had developed UTI with Grade III (left) and Grade I vesicoureteric reflux for which deflux injections had been given with a subsequent complete resolution of reflux bilaterally. A recent follow-up at 13-year post-redo surgery, he had fecal incontinence with dry period relief for only about 5–6 h after the bowel cleaning therapy. He is now in 9th grade and manages well with diapers. His height and weight (23.5 kg) though remained poor.
A 4-month-old boy was diagnosed with rectosigmoid type of HD on barium and rectal biopsy. Bowel was too dilated, thus required a proximal diversion with transverse colostomy. A transanal PT was performed at 1 year of age, though the descending colon was found to be too narrow and pencil like (postenterocolitis). Postoperatively, this narrow bowel did not work and the PT had to be revised. The narrow descending colon was sacrificed. The remaining bowel distal to the colostomy site was not found sufficient for a tension-free anastomosis. Thus, the transverse colostomy was dismantled and closed. After this maneuver, there was sufficient bowel length to bring it down to the perineum for a satisfactory anastomosis. A proximal diversion with an ileostomy was added, to protect the colocolic and coloanal anastomosis.
Complications in redo pull-through
Eight of 32 (25%) Re PT patients experienced postoperative complications, namely, wound infection in 5, adhesive intestinal obstruction in 1, anastomotic stricture 1, and enterocolitis in 1. In a case of TCA requiring repeated resection of the dilated bowel, the ureter got entrapped between the dilated colonic patch and the ileal loop. The left ureter got injured during the dissection for the third redo ileoanal PT. The ureteric injury was identified and repaired on table, with renal function fully preserved.
Outcomes postredo pull-through
Postoperative assessment included calibration per rectal examination (not the dilatation) at 3 weeks. Patients on proximal stoma required graded dilation for the next 6–8 weeks till the closure of stoma. Others with no stoma did not undergo dilatation program. A 5–6 times stool frequency was common in the immediate postoperative period. However, the frequency was reduced gradually over the months and years. Out of 32 patients, 4 were still on the stoma. Of the remaining 28, 5 (17.9%) had a normal stooling pattern, 12 (42.9%) complained of occasional soiling, 2 (7.1%) complained of daily soiling, and 4 (14.3%) complained of constipation.
| Discussion|| |
Indication for redo pull-through
The most common indication for Re PT has been a retained aganglionosis or TZ pathology. Previous studies well elucidate that there are mainly two pathomechanisms which are responsible for persistent unresponsive obstructive symptoms: first is the histopathological problems such as insufficient resection of the TZ or acquired aganglionosis;, second is due to the anatomical reasons such as anastomotic stricture, rigid cuff, fecal fistulae, PT bowel retraction, bowel ischemia, pelvic abscess, fibrosis, and bowel twist. These are often seen in patients, where the surgery had been performed by a young member in a noninstitutional setup.
Several authors have reported the disappearance of the ganglion cells after the PT procedure leading to the secondary aganglionosis.,,,,, This could possibly be due to either infection or overstretching of the bowel to make it ischemic and the consequent loss of ganglion cells in the PT segment.
The frozen section biopsy by a trained pathologist is pivotal in the era of increasing number of primary single-staged PT procedures being performed worldwide. In these patients, the intraoperative frozen section showed the presence of ganglion cells in the biopsied specimen, but the postoperated report showed the absence of ganglion cells in the proximal-most part of the resected bowel. In this series, there were three well-documented cases with disparity on frozen section and formal biopsy reports. Frozen section diagnosis and the final pathologic diagnosis may show a discrepancy in up to 3% of the patients. The other problem faced by a developing nation like ours is the lack of frozen section facility and experienced pathologists in most centers, even in the presence of a trained pediatric surgeon. In such situations, clinical acumen of the surgeons helps to determine the TZ. Most surgeons do a PT at approximately 10 cm proximal to the proposed TZ and later review the biopsy of the tip of PT. However, in redo cases, the importance of frozen section biopsy and of a trained pathologist cannot be overlooked and hence such cases should be referred to centers where all facilities are available. Lawal et al. believe that many interpretations of intestinal neuronal dysplasia are actually TZ pathology in HD and would advocate a reanalysis of the specimens.
Best technique for redo pull-through
In most redo cases, a proximal diversion in the form of colostomy/ileostomy before attempting a Re PT is useful. Proximal diversion was required in about 60% of our cases to protect the distal anastomosis. An ileostomy and not the colostomy should be fashioned where the residual length of distal colon was short so as to first reduce the dilatation of the colon and then make use of the same for a PT. Similar to the series by Langer where 5 (55.6%) of the 9 patients who underwent repeat PT had a defunctioning stoma. A stoma provided an adequate rest and protection to the pelvic area, where Re PT had been performed; ensuring proper healing.
The type of failed procedure is important, while planning a Re PT. Nonetheless, the choice of procedure for Re PT would depend on the indication for redo surgery, the nutritional status of the patients, the type of bowel preparation, the available length of the bowel, the scarring in the perineum, and most importantly, the experience of the surgeon with a particular surgical technique. All our Re PT were done by the modified Scott Boley technique due to the experience and ease of performing this procedure by the senior operating surgeon since 1980.
Schweizer et al. performed Duhamel PT in all 17 patients post initial failed surgery for HD (Soave in 3, Rehbein in 13, and Duhamel in 1), with good outcome.
Most of our redo HD PT cases were post-Duhamel procedure complications. Duhamel is considered and promoted as the simplest of all surgical techniques and good for newcomers in the field. Second, Duhamel surgery is associated with common complications such as residual spur, dilated residual pouch, a long suture line vulnerable to leak, pseudocavity and abscess formation, and subsequent fibrosis and stricture development. In such cases, Re PT required both excision and trimming of the residual pouch to shorten the length and reduce the width of the muscular cuff. Furthermore, post-Duhamel surgery, though possible, it becomes difficult to make a mucosal tube for Scott Boley's type PT procedure from a big and dilated residual pouch. However, in redo cases, where the bowel is normal and healthy, with no bleeding or fibrosis in the retrorectal presacral space, a redo Duhamel procedure can be performed. If a Soave's approach was previously performed, the virgin presacral plane permitted space and called for an easier pelvic dissection in redo cases.
Although others have promoted, the authors do not prefer transanal PT approach for the major redo cases requiring a proper evaluation of the bowel that is possible only after the abdomen has been opened and the bowel status examined properly. For those children who develop a stricture after initial surgery, a transabdominal redo PT using any of the three techniques is feasible. The redo procedure can be chosen depending on the site of a stricture (high/low) and its length (cuff stricture/anastomotic site stricture). Pini–Prato et al. used a classical Soave's/Boley-Scott approach to deal with three such cases of anastomotic site strictures in his series of 70 cases.
In the authors' experience, open Scott Boley's procedure was repeatedly practiced and proved reliable in all Re PT cases. A similar series of follow-up of 36 cases requiring Re PT for HD, reported successful outcomes of 22 patients managed by a repeat classical Soave's or its modification (Soave–Boley's).
Recognize redo cases early for a better outcome
Patients of HD with long-standing constipation remain undiagnosed for months to years. In India, about 82% children with HD may present late beyond infancy. The major concern in these patients is to differentiate them from other causes of habitual constipation. An early diagnostic workup consisting of clinical history, including family history; complete clinical examination including a digital rectal examination; a radiological contrast study; and histology by an experienced pathologist is necessary to establish the diagnosis. However, the difficulty is experienced with newborns presenting with constipation. A suction biopsy used liberally in the West must include the presence of submucosa for even the specially trained pathologist to make a diagnosis of HD with certainty.
Many patients present with persistent obstructive symptoms not responding to conservative measures like bowel management, laxatives, and behavior training, even after surgery for HD. Some of them may outgrow these obstructive symptoms with time; however, others may require surgical intervention.
Redo surgeries for HD should preferably be performed at tertiary care institutes with the trained pediatric surgeon and experienced pathologists, who are available at the time when the frozen section biopsy is performed. The discrepancy in the results is a preventable cause for Re PT.
In most cases, a series of Re PT, the surgical complications, cosmetic results, stooling scores, enterocolitis scores, and soiling frequency are worse when compared to those who undergo a single effective primary PT procedure., Hence, a Re PT should best be prevented as far as possible.
| Conclusions|| |
Most patients requiring Re PT for HD belonged to the subgroup who got complicated after a Duhamel's procedure resulting in a residual rectal pouch, fecaloma formation, long residual spur, anastomotic leak, and pelvic abscess including fecal fistula. Retraction of bowel, inversion of cuff, twist, distal bowel stricture, and perianal fibrosis were the reasons for redo PT after the Scott Boley's procedure. Proper planning with an initial diversion, nutritional buildup, barium study evaluation, frozen section facility, experienced pathologist, and most importantly the surgeon's experience are prerequisites for a successful initial surgery, and especially for Re PT whenever the need be.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]