Constipation is one of the conditions most commonly referred to pediatric gastroenterologists, accounting for up to 25% of all visits (1). The aim of treating childhood constipation is to make defecation as easy and painless as possible. Long-term follow-up studies of children with constipation who are younger than 5 years have shown 50% recovered within 1 year and 65% to 70% recovered within 2 years (2). Those who do not improve require laxatives to have daily bowel movements or they may continue to soil for years.
Children with neuromuscular handicaps, such as cerebral palsy and spinal dysraphism, often have persistent defecation problems, including recurrent fecal impaction and overflow soiling. To manage fecal incontinence and constipation in this subset of constipated children, various techniques have been tried, which recently include the use of antegrade enemas through a cecostomy (3,4).
In 1990, Malone et al. (5) described forming a continent appendicocecostomy through which the cecum could be intermittently catheterized for administration of an antegrade continence enema. A catheterizable stoma was placed in the right iliac fossa or at the level of the umbilicus. A tubularized enteric conduit from the ileum or cecum was created in patients without an appendix (6). Using the antegrade continence enema, the large bowel could be cleaned out at regular intervals, avoiding accumulation of stools and subsequent soiling (7). To avoid the complications of stoma stenosis and prolapse associated with appendicocecostomy, newer techniques for antegrade enema administration have been developed (8). A surgeon can perform the cecostomy, or an interventional radiologist or a gastroenterologist can perform the procedure using local anesthesia, similar to placing a percutaneous gastrostomy (9,10).
In this study, we report the use of antegrade enemas through cecostomy catheters in a group of children who are neurologically normal and who had failed conventional medical treatments for constipation.
Using a retrospective review of medical records from between 1997 and 1999 at Children's Hospital of Pittsburgh, we identified all patients who had undergone cecostomy placement for administration of antegrade enemas and who had no evidence of neurologic handicap. These children had been referred to a tertiary motility center for further evaluation of intractable constipation. All patients had undergone extensive evaluations to rule out organic causes for their constipation. Evaluation included rectal biopsy, magnetic resonance imaging of the spine, and contrast studies to rule out anatomic abnormalities. The medical records were reviewed for evidence of neurologic or developmental deficit. The physician to whom the child was referred for further evaluation also performed neurologic and developmental assessment at the time of initial evaluation. No child was being treated for attention deficit disorder or had a history of premature birth.
A questionnaire was used to interview caregivers 13.1 ± 8.5 months after cecostomy placement. No caregiver refused to participate in the interview. Outcome measurements included number of weekly bowel movements, number of weekly soiling episodes, number of medications used for constipation, weekly episodes of abdominal pain (0 = none, 1 = once or twice, 2 = a few times, 3 = fairly often, 4 = very often, 5 = everyday), number of missed school days each month, and number of physician office visits each year because of constipation. Primary caretakers also rated their children's overall health and emotional state on a 1-to-5 scale (1 = poor, 2 = fair, 3 = good, 4 = very good, 5 = excellent) before cecostomy catheter and at follow-up.
The Wilcoxon test and the Student t test were used for statistical analyses. All P values were two sided with P less than 0.05 considered statistically significant.
The Human Rights Committee of Children's Hospital of Pittsburgh approved the chart review and telephone interview.
We identified 12 children and adolescents who were developmentally and neurologically normal (9 male, aged 8.7 ± 4.4 years) and who underwent cecostomy placement for administration of antegrade enemas. Duration of symptoms at the time of referral ranged from 1.5 years to 9.5 years, with a mean of 5.3 ± 2.5 years. All patients were receiving at least two medications for constipation treatment and had fecal soiling at the time of referral. Eight patients had been previously hospitalized at least once and had required nasogastric infusion of lavage solutions to clear fecal impactions before cecostomy. Four patients had received anorectal biofeedback therapy without success.
Ten patients had undergone motility testing with colonic and anorectal manometry (11). Nine of these patients had abnormal motility of the dilated distal segment of colon with simultaneous low-amplitude contractions and normal motility in the more proximal colon. Ninety percent of patients had an increased rectal sensory threshold (60–360 mL; mean, 169 ± 110 mL; normal threshold for sensation, 20–40 mL of air). All ten patients had normal resting anal sphincter pressure. Results of motility testing did not predict treatment success or which patients would be able to discontinue cecostomy.
The cecostomy was performed surgically in 11 patients and by interventional radiology in 1 patient. After cecostomy, nursing personnel educated the family in administering antegrade enemas and in care of the cecostomy site. Parents were instructed to check the site for drainage and redness. The choice of irrigation solution used after cecostomy varied, based on the preference of the treating physician. Most patients began with low-volume infusions of solution, which were increased according to the therapeutic response. Sixty-seven percent of patients used 200 mL to 1,000 mL (mean 478 mL ± 262 mL) polyethylene glycol irrigation solution, daily to every other day. Twenty-five percent of patients used a combination of saline and glycerin, mixing 60 mL to 75 mL of glycerin in 240 mL to 300 mL of saline. One patient received 90 mL phosphate soda solution followed by 300 mL of saline. Evacuation occurred within 1 hour of enema administration in seven children and occurred within 3 hours in the other five children.
Cecostomy with the use of antegrade enemas led to significant improvement in all outcomes measures (Table 1). This included increased number of bowel movements each week, fewer soiling episodes each week, lower abdominal pain scores, increased emotional health scores, increased overall health score, fewer medications given for constipation, fewer missed school days each month, and fewer physician office visits each year.
No acute adverse events occurred related to cecostomy. Postoperative adverse events included skin breakdown and development of granulation tissue in one patient, leakage of irrigation solution in one patient, and accidental removal of the catheter in two patients with subsequent easy catheter replacement by the interventional radiologist. No adverse event led to discontinuation of antegrade enema use.
No child has required admission to a hospital because of fecal impaction since starting antegrade enemas. Five patients discontinued antegrade enemas with removal of the cecostomy at a mean of 14.6 ± 9.1 months after beginning treatment. None of the five children has redeveloped problems with constipation or fecal soiling.
Constipation that fails to respond to routine medical treatment can be a challenging clinical problem in children (12). Although most childhood constipation responds to a combination of behavioral and medical treatment, a sizable subset of nonresponders exists (13). Often extensive evaluation of these patients fails to find organic causes. Chronic constipation is associated with progressive fecal retention, distension of the rectum, and loss of rectal sensory and motor function. Fecal incontinence as a result of retained stools has tremendously negative impacts on the emotional and social development of affected children (14).
In this study, administering antegrade enemas through a cecostomy in children who were neurologically normal was safe and effective. The patients studied had very challenging cases of constipation, as shown by repeated hospitalizations, number of medications used, duration of symptoms, and visits to health care providers.
Currently, the most widely used technique for administering antegrade continence enemas is through an appendicocecostomy, first described in 1990 by Malone et al. (5). The original procedure combined the principle of an antegrade enema for colonic washout with the Mitrofanoff technique of forming a continent, nonrefluxing conduit that could be catheterized easily through the abdominal wall (6). Modifications to Malone technique include orthotopic appendicostomy with or without an antireflux imbrication, which can be performed laparoscopically (10). The procedure is well tolerated, and most families report significant improvement in their children's constipation (15). However, problems with stenosis of the cutaneous opening and prolapse of the stoma have occurred. Griffiths and Malone (16), in a recently published report on 21 patients, noted a high proportion of problems related to the stoma, requiring laparotomy on four occasions. One literature review of surgically performed appendicocecostomy reported complications such as stoma stenosis (up to 25%), stoma leak (5–10%), difficulty catheterizing the stoma (5%), pain with enema administration (3%), wound infection (3–5%), adhesive bowel obstruction (1–2%), granulation tissue (1%), cecal volvulus (0.5%), and hyperphosphatemia (0.5%) (17).
Percutaneous cecostomy using interventional radiology is emerging as a simple, nonsurgical technique that seems to have success rates similar to more invasive methods (7,8). It has been used to treat colonic pseudoobstruction, true colonic obstruction, and enterocolitis (18,19). It also has been used in selected cases of chronic constipation with fecal incontinence in children with neurologic defects (3,4,7,8,10).
Percutaneous colonoscopic cecostomy has recently been described for managing chronic defecation disorders in children with known medical causes for their constipation (9). The standard pull technique for percutaneous gastrostomy is used. The most important advantage of this technique is that the cecum is directly visualized, avoiding inadvertently performing the cecostomy in the terminal ileum. In the series that Rivera et al. (9) reported, most patients underwent the procedure under general anesthesia. Complications included pain, fever, abdominal distension, and one episode of enterocolitis in a patient with Hirschsprung disease.
Our patients benefited from antegrade enemas with improvement in overall health and emotional scores and in fewer days out of school. Visits to health care providers significantly decreased. No child reported discomfort at the cecostomy sufficient to interfere with daily activities.
The main advantage of antegrade enemas in treating chronic fecal incontinence is in achieving a predictable free-of-soiling interval. Cleaning the entire colon reliably avoids “accidents.” Enemas administered through the rectum allow only a partial clean out of the left side of the colon. A cecostomy button is easily covered with a shirt, and children can administer the antegrade enemas independently as they get older. Satisfaction with antegrade enemas has been reported as high as 93% in children (10,14,20).
Preoperative motility testing suggested that rectal dilation was secondary to chronically retained stools. This dilation also may have contributed to the very high threshold for the urge to defecate noted in most patients. These findings are consistent with a history of long-standing functional constipation and are likely the consequence of fecal retention rather than the cause of constipation. The most likely mechanism by which antegrade enemas are beneficial is in freeing the colon of retained stools, which allows a decrease in the size of the rectum and subsequent normalization of rectal sensory and motor function.
In summary, this study describes the use of cecostomy placement and antegrade enemas to treat the most severe cases of intractable nonorganic constipation. We stress that this study included a carefully evaluated and highly selected group of healthy children. This intervention should be considered before proceeding with partial or total colectomy, a more invasive option occasionally used in children with severe constipation (21). Candidates for antegrade enemas may include patients with severe constipation that leads to hospitalization, those in whom multiple medical regimens have failed, those who refuse either oral medications or retrograde enemas, and those without generalized colonic dysmotility (22).
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