JOURNAL OF LAPAROENDOSCOPIC & ADVANCED SURGICAL TECHNIQUES Volume 24, Number 4, 2014 ª Mary Ann Liebert, Inc. DOI: 10.1089/lap.2013.0292
2013 IPEG Papers
Laparoscopic-Assisted Percutaneous Cecostomy for Antegrade Continence Enema Lori DeFreest, MD, PhD, Janeen Smith, PA-C, and Christine Whyte, MD
Abstract
Purpose: The antegrade continence enema (ACE) is an option in the management of fecal incontinence and chronic constipation. We report our experience with a simple laparoscopic technique. Subjects and Methods: Data were collected on 16 children (8 boys) who underwent laparoscopic cecostomy for ACE. Success was defined as cessation of fecal soiling with no need for diapers. Results: Mean age at laparoscopic cecostomy was 11 years (range, 6–16 years). Mean follow-up after initial cecostomy was 22 months (range, 6–51 months). Diagnoses in 16 patients were functional constipation with soiling (n = 14), incontinence after surgery for Hirschsprung’s disease (n = 1), and constipation secondary to mitochondrial disease (n = 1). Seven had significant developmental or psychiatric problems. Three patients had primary placement of a trapdoor device (Chait); 13 had placement of a long tube, with later replacement by a skin-level device. We have evolved a laparoscopic-assisted percutaneous technique, using metallic anchor sutures on the cecum, and a dilator and peel-away sheath for introduction of the catheter. Complications occurred in 5 patients; 3 returned to the operating room: 1 for tube occlusion, 1 for suture granuloma, and 1 for a dislodged tube at 7 months postoperatively. One patient received intravenous antibiotics because of suspected peritonitis on the first postoperative day. One was re-admitted with abdominal pain. Five of 16 patients have failed therapy (four tubes removed and one tube in situ). Three have had only minor improvement. Eight have had successful ACE management, of whom 1 patient has had his tube removed after resolution of symptoms. Of 8 patients with no or minimal improvement with ACE, 5 have significant psychiatric problems. Conclusions: Laparoscopic-assisted percutaneous cecostomy has an excellent safety profile and patient comfort. The procedure is simple, secure, and reversible. Results were excellent in half of the patients. Associated psychiatric or behavioral problems may predict poor response to ACE.
Introduction
T
he antegrade continence enema (ACE) is an option in the management of fecal incontinence and chronic constipation. First described by Malone et al.1 in 1990, it was a major advance in the care of patients with fecal incontinence from several causes, including intractable constipation. This technique involves surgical construction of a stoma into the cecum to allow for daily, self-administered, antegrade enemas, resulting in predictable colonic emptying and social cleanliness. It was originally described for patients with incontinence related to myelomeningocele and anorectal malformations. The success rate of the procedure during its early years was reported to be as high as 80%.2 Later, the technique was applied to more patients with chronic functional constipation, and more recent data on long-term outcomes sug-
gest that successful results can be obtained in about twothirds of patients.3 The ACE may be administered via a catheterizable stoma fashioned from an appendicostomy or tubularized bowel or via a tube cecostomy. Appendicostomy is common, but drawbacks include patient reticence about stomal catheterization and high rates of stomal stenosis requiring surgical revision.3 An acceptable alternative is to use an implanted tube cecostomy, with the option of an unobtrusive skin-level device. We have developed a safe and effective means of creation of a tube cecostomy that has few stomal complications. Initially this procedure used laparoscopically placed transparietal fixation sutures and placement of the tube under direct laparoscopic visualization, but it has evolved to utilize a modified percutaneous technique for placement of a tube as the initial conduit for the ACE. This tube is changed to a skin-
Division of Pediatric Surgery, Albany Medical College, Albany, New York.
261
262
level device at approximately 6 weeks. Here we report excellent safety and low complication rates with a laparoscopicassisted percutaneous tube cecostomy. Subjects and Methods
Records were obtained for all patients who had tube cecostomy for ACE from April 2009 to June 2013, in the Division of Pediatric Surgery at our institution. The procedures were all performed by a single surgeon (C.W.). Patients were managed closely by one of the authors ( J.S.), a dedicated physician’s assistant, who was involved in patient and family education, routine follow-up, adjustment of the washout program, and trouble shooting of difficulties postoperatively. Information collected included age at surgery, gender, diagnoses, results of investigations, details of the procedure, type of tube initially placed, current device, duration of follow-up, outcome, and complications. Institutional Review Board approval was obtained for the study (Albany Medical College Institutional Review Board protocol number 3492). Success is defined as cessation of soiling with no need for diapers. Before cecostomy was decided upon, all patients were examined under anesthesia, to rule out anatomical problems, and except for patients with known Hirschsprung’s disease, all had rectal biopsy. Patients had bowel preparation before surgery. Severely impacted patients had manual disimpaction before bowel preparation. The technique of laparoscopic-assisted percutaneous cecostomy has evolved over the time period studied. A dose of intravenous antibiotic was administered at induction of anesthesia. Initially, a three-port technique was used, with hand placement of transparietal sutures for cecal fixation. Later, we evolved a two-port technique, using metallic T sutures (gastrointestinal anchor set with Saf T Pexy; Kimberly-Clark, Roswell, GA) for cecal fixation. Three sutures are placed around the cecostomy site. The tube is then introduced into the cecum using the dilators and sheath from the KimberlyClark MIC-Key G introducer set, which also contains the T sutures. The T sutures were cut after 10 days, or they separated spontaneously. The long tube used for initial insertion was the MIC* gastrostomy feeding tube (Kimberly-Clark). Skin-level devices were the Chait trapdoor (Chait trapdoor cecostomy; Cook Medical, Bloomington, IN) or the MIC-KEY* low profile gastrostomy feeding tube (Kimberly-Clark), depending on family preference. If a long tube was used for initial insertion, the skin-level device was placed 6 weeks later by the interventional radiology service. Prior to surgery, at the initial visit, families were given detailed information, including written materials, about the ACE. Patients were admitted to the hospital overnight after surgery. The parent was taught to flush the tube with 10 mL of tap water daily for the first 10 days and received a prescription for program supplies. At 10 days, patients returned to our office to receive training, with the physician’s assistant, to do the first washout in a dedicated examination room with en suite bathroom. The usual first washout was 400 mL of normal saline. Adjustments were made at subsequent visits. Results
During the study period, 21 patients had creation of a tube cecostomy for fecal soiling. Five had open surgery because of
DEFREEST ET AL.
concomitant urological procedures or abdominal adhesions. Sixteen had laparoscopic-assisted percutaneous cecostomy. These 16 patients are the subject of this report. Eight of the 16 patients were male. Mean age at laparoscopic cecostomy was 11 years (range, 6–16 years). Mean follow-up after cecostomy was 22 months (range, 6–51 months). Success is defined as cessation of fecal soiling with no need for diapers. Diagnoses in 16 patients were functional constipation with soiling (n = 14), incontinence after surgery for Hirschsprung’s disease (n = 1), and constipation secondary to mitochondrial disease (n = 1). The patients with functional constipation had failed medical management. Developmental, behavioral, and psychiatric comorbidities were documented in 7 patients, with multiple diagnoses in 5. They included developmental delay secondary to Down syndrome or prematurity (n = 2), attention deficit disorder (n = 3), anxiety (n = 2), oppositional defiance disorder (n = 1), bipolar disorder (n = 2), autism spectrum disorders (n = 2), behavioral disturbance (n = 2), and mood dysregulation (n = 1). Three patients had primary placement of a Chait trapdoor cecostomy. The remaining 13 patients, who had laparoscopic cecostomy, had placement of a long tube, a MIC* gastrostomy feeding tube size 12 French. All of these were subsequently changed to a skin-level device, either a Chait trapdoor cecostomy or a MIC-KEY* low profile feeding tube, at 6 weeks postoperatively. In the first 7 patients, transparietal sutures were placed by hand for cecopexy, and metallic T sutures were used for the last 9. There were no adverse events associated with the use of metallic sutures in the cecum. Mean length of stay was 3 days (range, 1–7 days). This included preoperative stay for bowel preparation. Mean stay after surgery was 1.5 days (range, 1–4 days). One child had an outpatient procedure but was re-admitted for pain control 2 days later. In the patients who had primary placement of a Chait trapdoor skin-level device, there was significant discomfort associated with manipulation of the tube in the early postoperative period. We found that comfort of patients is excellent if they learn the washout technique with a long tube that they can manipulate at a distance from the operative site and then are converted to a skin-level device after 6 weeks. Five patients had failed outcome from ACE. Four have had the tube removed for failure of therapy. These 4 patients all had intolerance and dislike of the tube and could not allow washouts and compliance with the program. One has the tube in place but uses it only occasionally. Three of these patients had significant psychiatric or behavioral problems. Three patients have only partial improvement in symptoms: 1 has problems with attention deficit disorder, anxiety, and bipolar disorder and cannot comply well, a second has attention deficit disorder and depression and has persistent problems with constipation and abdominal pain, and 1 has mitochondrial disease with colonic dysmotility and severe colonic distension. Despite an increase in her washout volume to 1000 mL, she continues to soil. Eight patients have done well with ACE and are clean. One has had his tube removed after 20 months of therapy because his symptoms completely resolved. In the 8 patients with treatment success, 2 had developmental or psychiatric diagnoses.
CECOSTOMY FOR ANTEGRADE CONTINENCE ENEMA
The first washout for patients is 400 mL of saline. It is administered, as a bolus, in our office suite by the parent under the supervision of one of the authors ( J.S.). Volumes are subsequently adjusted. In 6 patients, the fluid was changed from saline to polyethylene glycol 3350 and electrolyte solution (GoLYTELY; Braintree Laboratories, Braintree, MA) solution. Current data on washout volume are available on 12 patients. Mean washout volume is 600 mL (range, 400– 1000 mL). Four are adding glycerin (30 mL) or bisacodyl (Dulcolax; Boehringer Ingelheim Pharmaceuticals, Ridgefield, CT) (20 mL) to the washout solution. Two are using the ACE alternate daily, 1 only uses the tube occasionally, and the rest use the ACE daily. Complications occurred in 5 patients; 3 returned to the operating room: 1 for suture granuloma, 1 for tube occlusion by a suture (technical error), and 1 for tube dislodgment 7 months postinsertion. One patient required intravenous antibiotics for suspected peritonitis on the first postoperative day. She had high fevers and pain, but a contrast study showed no leakage. Her symptoms resolved by the second postoperative day. One was re-admitted for abdominal pain on the second postoperative day. She had no evidence of infection. Leakage around the tube was reported by 4 patients. These were the same 4 patients who required tube removal for failure of therapy. This problem coincided with the development of distal fecal impaction and obstruction in the patients. The tube was removed from these patients. Discussion
The introduction of the ACE was a major advance for children with fecal incontinence. Although the underlying condition is not cured, ACE allows the patient to empty the colon at a time of their choosing and, in most cases, to remain clean in between. Although similar results can be achieved with rectal washouts, the cecostomy permits antegrade cleaning while the patient sits on the toilet in privacy and comfort. Our results in terms of clinical response may be compared and contrasted with the large series of ACE cases reported by Siddiqui et al.,3 a majority of whom had appendicostomy and many of whom had functional constipation. They reported success in 69%, whereas we have had excellent results in 50% of our cases. Siddiqui et al.3 reported that, although their patients began with a program of saline washouts, a majority transitioned to using GoLYTELY. Of our 12 patients currently using their cecostomies, 6 are using GoLYTELY, having begun the program with saline. Most case series report ACE with appendicostomy, but we have pursued a different approach, preferring laparoscopicassisted percutaneous tube cecostomy. The tube cecostomy is technically simple and readily reversible. Most of our patients have functional constipation, a condition that can improve over time, and many of the patients may not require the ACE for life. Many of our patients and families expressed dislike of the idea of daily catheterization of a stoma and instead preferred to access a skin-level device. The devices, either the Chait trapdoor or the MIC-KEY* button, are unobtrusive, generally do not leak, and are compatible with swimming and sports (Fig. 1). Appendicostomies develop stomal stenosis in 14%–
263
FIG. 1. An 11-year-old girl demonstrates an acceptable cosmetic result with the Chait trapdoor device.
50%2–6 and leakage in 7%–35%.2–4 Up to 50% of patients require surgical revision of the stoma.3,5,6 More recent articles from a single center have reported improvement in these outcomes with technical refinements7,8 such as V-V plasty of the appendicostomy suture line and routine cecal plication. We have not experienced leakage in a majority of our patients. Four of our patients (25%) reported leakage; all were patients with failed outcomes. Appendicostomy is certainly a good alternative for many patients with fecal incontinence, but it is not as easily reversible as tube cecostomy, is a more complex procedure, and requires more skill on the part of the parent. Our patients are discharged quickly after surgery, whereas patients with appendicostomy may develop ileus after manipulation and suturing of the cecum, and a substantial fascial incision is required for an umbilical appendicostomy.7,8 In 3 patients, we placed a Chait trapdoor at the first procedure. These patients experienced discomfort and fear with connecting and disconnecting the trapdoor with a fresh operative site but adapted well to the trapdoor subsequently. We now prefer to place a long tube, usually a MIC* gastrostomy, size 12, at the first operation. The balloon on the tube allows for a good seal between the cecum and abdominal wall, reducing the risk of internal leakage and peritonitis. Also, our patient comfort is excellent when the family learn the washout procedure while interacting with the long tube, remote from the fresh operative site. After 6 weeks, the patients have placement of a skin-level device. For Chait trapdoors we typically have the procedure done by the vascular interventional radiology service. For the first seven laparoscopic cecostomies, we placed transparietal sutures for cecopexy and removed them on Day 10. However, these sutures were tedious to place and caused discomfort to the patients. The gastrointestinal anchor sutures (T sutures), which we have used for the last 9 cases, have been quick and easy to place, and they separate spontaneously or are removed by Day 10. They are associated with much less discomfort. We have had no adverse events related to the metallic sutures in the cecum. The procedure as we have evolved it, with T sutures for
264
cecopexy and a Seldinger approach for the tube insertion, is now safe, quick, and simple to perform. Other authors have reported a percutaneous endoscopic cecostomy procedure using a colonoscope. However, Siddiqui et al.3 reported that 3 of their 10 percutaneous endoscopic cecostomy patients had suspected peritonitis after the procedure. We have had only 1 such patient at the beginning of our series and none recently. A contrast study showed no leakage, and she responded quickly to intravenous antibiotics. Most of our patients have been admitted preoperatively for bowel preparation, but recently we have begun to do more preparation at home, without problems. This is a troubled group of patients. The indignities of dealing with fecal incontinence during the school years, combined with a high rate of behavioral, developmental, and psychiatric problems, severely impact the quality of life for these children and their families. Our method of tube placement has allowed us to institute ACE management while minimizing discomfort and psychological trauma for the children. The washouts are easy to learn without complicated maneuvering of stomas or tubing connections. The ultimate cosmetic result is acceptable, and the cecostomy devices are unobtrusive. Eight of our patients had failure of the ACE or poor outcome. In this series, failure was primarily caused by failure to comply with the treatment regimen. Five children or parents could not comply with the washouts, and ultimately 4 patients had their tube removed. In the remaining 3 patients with failure or poor outcome, 2 have documented dilatation of the colon on contrast studies, as well as documented abnormal motility of the colon, but despite increasing the washout volume to 1000 mL, they still remained constipated. Five of these 8 with poor outcome have documented psychiatric diagnoses. In this small series, we have noted a correlation between psychiatric diagnosis and failure of the ACE. However, two of our eight treatment successes had significant developmental and psychiatric diagnoses; therefore we would not exclude a patient from having an ACE solely on the grounds of developmental delay or psychiatric problems. Our treatment goals are to attain social cleanliness, allowing the child to attend school, wearing normal underwear and no diapers. No one procedure is going to provide a perfect solution. Laparoscopic cecostomy with subsequent placement of a skin-level device for ACE management is a reasonable approach in cooperative patients. Children who have success with the ACE are very satisfied with their outcomes.
DEFREEST ET AL. Acknowledgments
We would like to thank our colleagues in interventional radiology for their help with changing the initial cecostomy tube to a more comfortable and cosmetically appealing skinlevel device. Disclosure Statement
No competing financial interests exist. References
1. Malone PS, Ransley PG, Kiely EM. Preliminary report: The antegrade continence enema. Lancet 1990;336:1217–1218. 2. Curry JI, Osborne A, Malone PS. The MACE procedure: Experience in the United Kingdom. J Pediatr Surg 1999; 34:338–340. 3. Siddiqui AA, Fishman SJ, Bauer SB, Nurko S. Long-term follow-up of patients after antegrade continence enema procedure. JPGN 2011;52:574–580. 4. Bani-Hani AH, Cain MP, Kaefer M, et al. The Malone antegrade continence enema: Single institutional review. J Urol 2008;180:1106–1110. 5. Mattix KD, Novotny NM, Shelley AA, Rescorla FJ. Malone antegrade continence enema (MACE) for fecal incontinence in imperforate anus improves quality of life. Pediatr Surg Int 2007;23:1175–1177. 6. Cascio S, Flett ME, De la Hunt M, et al. MACE or caecostomy button for idiopathic constipation in children: A comparison of complications and outcomes. Pediatr Surg Int 2004;20:484–487. 7. Rangel SJ, Lawal TA, Bischoff A, Chatoorgoon K, Louden E, Pena A, Levitt MA. The appendix as conduit for antegrade continence enemas in patients with anorectal malformations: Lessons learned from 163 cases treated over 18 years. J Pediatr Surg 2011;46:1236–1242. 8. Lawal TA, Rangel SJ, Bischoff A, Pen˜a A, Levitt, MA. Laparoscopic-assisted Malone appendicostomy in the management of fecal incontinence in children. J Laparoendosc Adv Surg Tech A 2011;21:455–459.
Address correspondence to: Christine Whyte, MD Division of Pediatric Surgery Albany Medical College 47 New Scotland Avenue Albany, NY 12208 E-mail: [email protected]
2013 IPEG Papers
Laparoscopic-Assisted Percutaneous Cecostomy for Antegrade Continence Enema Lori DeFreest, MD, PhD, Janeen Smith, PA-C, and Christine Whyte, MD
Abstract
Purpose: The antegrade continence enema (ACE) is an option in the management of fecal incontinence and chronic constipation. We report our experience with a simple laparoscopic technique. Subjects and Methods: Data were collected on 16 children (8 boys) who underwent laparoscopic cecostomy for ACE. Success was defined as cessation of fecal soiling with no need for diapers. Results: Mean age at laparoscopic cecostomy was 11 years (range, 6–16 years). Mean follow-up after initial cecostomy was 22 months (range, 6–51 months). Diagnoses in 16 patients were functional constipation with soiling (n = 14), incontinence after surgery for Hirschsprung’s disease (n = 1), and constipation secondary to mitochondrial disease (n = 1). Seven had significant developmental or psychiatric problems. Three patients had primary placement of a trapdoor device (Chait); 13 had placement of a long tube, with later replacement by a skin-level device. We have evolved a laparoscopic-assisted percutaneous technique, using metallic anchor sutures on the cecum, and a dilator and peel-away sheath for introduction of the catheter. Complications occurred in 5 patients; 3 returned to the operating room: 1 for tube occlusion, 1 for suture granuloma, and 1 for a dislodged tube at 7 months postoperatively. One patient received intravenous antibiotics because of suspected peritonitis on the first postoperative day. One was re-admitted with abdominal pain. Five of 16 patients have failed therapy (four tubes removed and one tube in situ). Three have had only minor improvement. Eight have had successful ACE management, of whom 1 patient has had his tube removed after resolution of symptoms. Of 8 patients with no or minimal improvement with ACE, 5 have significant psychiatric problems. Conclusions: Laparoscopic-assisted percutaneous cecostomy has an excellent safety profile and patient comfort. The procedure is simple, secure, and reversible. Results were excellent in half of the patients. Associated psychiatric or behavioral problems may predict poor response to ACE.
Introduction
T
he antegrade continence enema (ACE) is an option in the management of fecal incontinence and chronic constipation. First described by Malone et al.1 in 1990, it was a major advance in the care of patients with fecal incontinence from several causes, including intractable constipation. This technique involves surgical construction of a stoma into the cecum to allow for daily, self-administered, antegrade enemas, resulting in predictable colonic emptying and social cleanliness. It was originally described for patients with incontinence related to myelomeningocele and anorectal malformations. The success rate of the procedure during its early years was reported to be as high as 80%.2 Later, the technique was applied to more patients with chronic functional constipation, and more recent data on long-term outcomes sug-
gest that successful results can be obtained in about twothirds of patients.3 The ACE may be administered via a catheterizable stoma fashioned from an appendicostomy or tubularized bowel or via a tube cecostomy. Appendicostomy is common, but drawbacks include patient reticence about stomal catheterization and high rates of stomal stenosis requiring surgical revision.3 An acceptable alternative is to use an implanted tube cecostomy, with the option of an unobtrusive skin-level device. We have developed a safe and effective means of creation of a tube cecostomy that has few stomal complications. Initially this procedure used laparoscopically placed transparietal fixation sutures and placement of the tube under direct laparoscopic visualization, but it has evolved to utilize a modified percutaneous technique for placement of a tube as the initial conduit for the ACE. This tube is changed to a skin-
Division of Pediatric Surgery, Albany Medical College, Albany, New York.
261
262
level device at approximately 6 weeks. Here we report excellent safety and low complication rates with a laparoscopicassisted percutaneous tube cecostomy. Subjects and Methods
Records were obtained for all patients who had tube cecostomy for ACE from April 2009 to June 2013, in the Division of Pediatric Surgery at our institution. The procedures were all performed by a single surgeon (C.W.). Patients were managed closely by one of the authors ( J.S.), a dedicated physician’s assistant, who was involved in patient and family education, routine follow-up, adjustment of the washout program, and trouble shooting of difficulties postoperatively. Information collected included age at surgery, gender, diagnoses, results of investigations, details of the procedure, type of tube initially placed, current device, duration of follow-up, outcome, and complications. Institutional Review Board approval was obtained for the study (Albany Medical College Institutional Review Board protocol number 3492). Success is defined as cessation of soiling with no need for diapers. Before cecostomy was decided upon, all patients were examined under anesthesia, to rule out anatomical problems, and except for patients with known Hirschsprung’s disease, all had rectal biopsy. Patients had bowel preparation before surgery. Severely impacted patients had manual disimpaction before bowel preparation. The technique of laparoscopic-assisted percutaneous cecostomy has evolved over the time period studied. A dose of intravenous antibiotic was administered at induction of anesthesia. Initially, a three-port technique was used, with hand placement of transparietal sutures for cecal fixation. Later, we evolved a two-port technique, using metallic T sutures (gastrointestinal anchor set with Saf T Pexy; Kimberly-Clark, Roswell, GA) for cecal fixation. Three sutures are placed around the cecostomy site. The tube is then introduced into the cecum using the dilators and sheath from the KimberlyClark MIC-Key G introducer set, which also contains the T sutures. The T sutures were cut after 10 days, or they separated spontaneously. The long tube used for initial insertion was the MIC* gastrostomy feeding tube (Kimberly-Clark). Skin-level devices were the Chait trapdoor (Chait trapdoor cecostomy; Cook Medical, Bloomington, IN) or the MIC-KEY* low profile gastrostomy feeding tube (Kimberly-Clark), depending on family preference. If a long tube was used for initial insertion, the skin-level device was placed 6 weeks later by the interventional radiology service. Prior to surgery, at the initial visit, families were given detailed information, including written materials, about the ACE. Patients were admitted to the hospital overnight after surgery. The parent was taught to flush the tube with 10 mL of tap water daily for the first 10 days and received a prescription for program supplies. At 10 days, patients returned to our office to receive training, with the physician’s assistant, to do the first washout in a dedicated examination room with en suite bathroom. The usual first washout was 400 mL of normal saline. Adjustments were made at subsequent visits. Results
During the study period, 21 patients had creation of a tube cecostomy for fecal soiling. Five had open surgery because of
DEFREEST ET AL.
concomitant urological procedures or abdominal adhesions. Sixteen had laparoscopic-assisted percutaneous cecostomy. These 16 patients are the subject of this report. Eight of the 16 patients were male. Mean age at laparoscopic cecostomy was 11 years (range, 6–16 years). Mean follow-up after cecostomy was 22 months (range, 6–51 months). Success is defined as cessation of fecal soiling with no need for diapers. Diagnoses in 16 patients were functional constipation with soiling (n = 14), incontinence after surgery for Hirschsprung’s disease (n = 1), and constipation secondary to mitochondrial disease (n = 1). The patients with functional constipation had failed medical management. Developmental, behavioral, and psychiatric comorbidities were documented in 7 patients, with multiple diagnoses in 5. They included developmental delay secondary to Down syndrome or prematurity (n = 2), attention deficit disorder (n = 3), anxiety (n = 2), oppositional defiance disorder (n = 1), bipolar disorder (n = 2), autism spectrum disorders (n = 2), behavioral disturbance (n = 2), and mood dysregulation (n = 1). Three patients had primary placement of a Chait trapdoor cecostomy. The remaining 13 patients, who had laparoscopic cecostomy, had placement of a long tube, a MIC* gastrostomy feeding tube size 12 French. All of these were subsequently changed to a skin-level device, either a Chait trapdoor cecostomy or a MIC-KEY* low profile feeding tube, at 6 weeks postoperatively. In the first 7 patients, transparietal sutures were placed by hand for cecopexy, and metallic T sutures were used for the last 9. There were no adverse events associated with the use of metallic sutures in the cecum. Mean length of stay was 3 days (range, 1–7 days). This included preoperative stay for bowel preparation. Mean stay after surgery was 1.5 days (range, 1–4 days). One child had an outpatient procedure but was re-admitted for pain control 2 days later. In the patients who had primary placement of a Chait trapdoor skin-level device, there was significant discomfort associated with manipulation of the tube in the early postoperative period. We found that comfort of patients is excellent if they learn the washout technique with a long tube that they can manipulate at a distance from the operative site and then are converted to a skin-level device after 6 weeks. Five patients had failed outcome from ACE. Four have had the tube removed for failure of therapy. These 4 patients all had intolerance and dislike of the tube and could not allow washouts and compliance with the program. One has the tube in place but uses it only occasionally. Three of these patients had significant psychiatric or behavioral problems. Three patients have only partial improvement in symptoms: 1 has problems with attention deficit disorder, anxiety, and bipolar disorder and cannot comply well, a second has attention deficit disorder and depression and has persistent problems with constipation and abdominal pain, and 1 has mitochondrial disease with colonic dysmotility and severe colonic distension. Despite an increase in her washout volume to 1000 mL, she continues to soil. Eight patients have done well with ACE and are clean. One has had his tube removed after 20 months of therapy because his symptoms completely resolved. In the 8 patients with treatment success, 2 had developmental or psychiatric diagnoses.
CECOSTOMY FOR ANTEGRADE CONTINENCE ENEMA
The first washout for patients is 400 mL of saline. It is administered, as a bolus, in our office suite by the parent under the supervision of one of the authors ( J.S.). Volumes are subsequently adjusted. In 6 patients, the fluid was changed from saline to polyethylene glycol 3350 and electrolyte solution (GoLYTELY; Braintree Laboratories, Braintree, MA) solution. Current data on washout volume are available on 12 patients. Mean washout volume is 600 mL (range, 400– 1000 mL). Four are adding glycerin (30 mL) or bisacodyl (Dulcolax; Boehringer Ingelheim Pharmaceuticals, Ridgefield, CT) (20 mL) to the washout solution. Two are using the ACE alternate daily, 1 only uses the tube occasionally, and the rest use the ACE daily. Complications occurred in 5 patients; 3 returned to the operating room: 1 for suture granuloma, 1 for tube occlusion by a suture (technical error), and 1 for tube dislodgment 7 months postinsertion. One patient required intravenous antibiotics for suspected peritonitis on the first postoperative day. She had high fevers and pain, but a contrast study showed no leakage. Her symptoms resolved by the second postoperative day. One was re-admitted for abdominal pain on the second postoperative day. She had no evidence of infection. Leakage around the tube was reported by 4 patients. These were the same 4 patients who required tube removal for failure of therapy. This problem coincided with the development of distal fecal impaction and obstruction in the patients. The tube was removed from these patients. Discussion
The introduction of the ACE was a major advance for children with fecal incontinence. Although the underlying condition is not cured, ACE allows the patient to empty the colon at a time of their choosing and, in most cases, to remain clean in between. Although similar results can be achieved with rectal washouts, the cecostomy permits antegrade cleaning while the patient sits on the toilet in privacy and comfort. Our results in terms of clinical response may be compared and contrasted with the large series of ACE cases reported by Siddiqui et al.,3 a majority of whom had appendicostomy and many of whom had functional constipation. They reported success in 69%, whereas we have had excellent results in 50% of our cases. Siddiqui et al.3 reported that, although their patients began with a program of saline washouts, a majority transitioned to using GoLYTELY. Of our 12 patients currently using their cecostomies, 6 are using GoLYTELY, having begun the program with saline. Most case series report ACE with appendicostomy, but we have pursued a different approach, preferring laparoscopicassisted percutaneous tube cecostomy. The tube cecostomy is technically simple and readily reversible. Most of our patients have functional constipation, a condition that can improve over time, and many of the patients may not require the ACE for life. Many of our patients and families expressed dislike of the idea of daily catheterization of a stoma and instead preferred to access a skin-level device. The devices, either the Chait trapdoor or the MIC-KEY* button, are unobtrusive, generally do not leak, and are compatible with swimming and sports (Fig. 1). Appendicostomies develop stomal stenosis in 14%–
263
FIG. 1. An 11-year-old girl demonstrates an acceptable cosmetic result with the Chait trapdoor device.
50%2–6 and leakage in 7%–35%.2–4 Up to 50% of patients require surgical revision of the stoma.3,5,6 More recent articles from a single center have reported improvement in these outcomes with technical refinements7,8 such as V-V plasty of the appendicostomy suture line and routine cecal plication. We have not experienced leakage in a majority of our patients. Four of our patients (25%) reported leakage; all were patients with failed outcomes. Appendicostomy is certainly a good alternative for many patients with fecal incontinence, but it is not as easily reversible as tube cecostomy, is a more complex procedure, and requires more skill on the part of the parent. Our patients are discharged quickly after surgery, whereas patients with appendicostomy may develop ileus after manipulation and suturing of the cecum, and a substantial fascial incision is required for an umbilical appendicostomy.7,8 In 3 patients, we placed a Chait trapdoor at the first procedure. These patients experienced discomfort and fear with connecting and disconnecting the trapdoor with a fresh operative site but adapted well to the trapdoor subsequently. We now prefer to place a long tube, usually a MIC* gastrostomy, size 12, at the first operation. The balloon on the tube allows for a good seal between the cecum and abdominal wall, reducing the risk of internal leakage and peritonitis. Also, our patient comfort is excellent when the family learn the washout procedure while interacting with the long tube, remote from the fresh operative site. After 6 weeks, the patients have placement of a skin-level device. For Chait trapdoors we typically have the procedure done by the vascular interventional radiology service. For the first seven laparoscopic cecostomies, we placed transparietal sutures for cecopexy and removed them on Day 10. However, these sutures were tedious to place and caused discomfort to the patients. The gastrointestinal anchor sutures (T sutures), which we have used for the last 9 cases, have been quick and easy to place, and they separate spontaneously or are removed by Day 10. They are associated with much less discomfort. We have had no adverse events related to the metallic sutures in the cecum. The procedure as we have evolved it, with T sutures for
264
cecopexy and a Seldinger approach for the tube insertion, is now safe, quick, and simple to perform. Other authors have reported a percutaneous endoscopic cecostomy procedure using a colonoscope. However, Siddiqui et al.3 reported that 3 of their 10 percutaneous endoscopic cecostomy patients had suspected peritonitis after the procedure. We have had only 1 such patient at the beginning of our series and none recently. A contrast study showed no leakage, and she responded quickly to intravenous antibiotics. Most of our patients have been admitted preoperatively for bowel preparation, but recently we have begun to do more preparation at home, without problems. This is a troubled group of patients. The indignities of dealing with fecal incontinence during the school years, combined with a high rate of behavioral, developmental, and psychiatric problems, severely impact the quality of life for these children and their families. Our method of tube placement has allowed us to institute ACE management while minimizing discomfort and psychological trauma for the children. The washouts are easy to learn without complicated maneuvering of stomas or tubing connections. The ultimate cosmetic result is acceptable, and the cecostomy devices are unobtrusive. Eight of our patients had failure of the ACE or poor outcome. In this series, failure was primarily caused by failure to comply with the treatment regimen. Five children or parents could not comply with the washouts, and ultimately 4 patients had their tube removed. In the remaining 3 patients with failure or poor outcome, 2 have documented dilatation of the colon on contrast studies, as well as documented abnormal motility of the colon, but despite increasing the washout volume to 1000 mL, they still remained constipated. Five of these 8 with poor outcome have documented psychiatric diagnoses. In this small series, we have noted a correlation between psychiatric diagnosis and failure of the ACE. However, two of our eight treatment successes had significant developmental and psychiatric diagnoses; therefore we would not exclude a patient from having an ACE solely on the grounds of developmental delay or psychiatric problems. Our treatment goals are to attain social cleanliness, allowing the child to attend school, wearing normal underwear and no diapers. No one procedure is going to provide a perfect solution. Laparoscopic cecostomy with subsequent placement of a skin-level device for ACE management is a reasonable approach in cooperative patients. Children who have success with the ACE are very satisfied with their outcomes.
DEFREEST ET AL. Acknowledgments
We would like to thank our colleagues in interventional radiology for their help with changing the initial cecostomy tube to a more comfortable and cosmetically appealing skinlevel device. Disclosure Statement
No competing financial interests exist. References
1. Malone PS, Ransley PG, Kiely EM. Preliminary report: The antegrade continence enema. Lancet 1990;336:1217–1218. 2. Curry JI, Osborne A, Malone PS. The MACE procedure: Experience in the United Kingdom. J Pediatr Surg 1999; 34:338–340. 3. Siddiqui AA, Fishman SJ, Bauer SB, Nurko S. Long-term follow-up of patients after antegrade continence enema procedure. JPGN 2011;52:574–580. 4. Bani-Hani AH, Cain MP, Kaefer M, et al. The Malone antegrade continence enema: Single institutional review. J Urol 2008;180:1106–1110. 5. Mattix KD, Novotny NM, Shelley AA, Rescorla FJ. Malone antegrade continence enema (MACE) for fecal incontinence in imperforate anus improves quality of life. Pediatr Surg Int 2007;23:1175–1177. 6. Cascio S, Flett ME, De la Hunt M, et al. MACE or caecostomy button for idiopathic constipation in children: A comparison of complications and outcomes. Pediatr Surg Int 2004;20:484–487. 7. Rangel SJ, Lawal TA, Bischoff A, Chatoorgoon K, Louden E, Pena A, Levitt MA. The appendix as conduit for antegrade continence enemas in patients with anorectal malformations: Lessons learned from 163 cases treated over 18 years. J Pediatr Surg 2011;46:1236–1242. 8. Lawal TA, Rangel SJ, Bischoff A, Pen˜a A, Levitt, MA. Laparoscopic-assisted Malone appendicostomy in the management of fecal incontinence in children. J Laparoendosc Adv Surg Tech A 2011;21:455–459.
Address correspondence to: Christine Whyte, MD Division of Pediatric Surgery Albany Medical College 47 New Scotland Avenue Albany, NY 12208 E-mail: [email protected]
