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Antegrade enemas for defecation disorders: do they improve the colonic motility? Ann Aspirot a,b, , Sergio Fernandez b , Carlo Di Lorenzo b , Beth Skaggs b , Hayat Mousa b a Service de Chirurgie Générale Pédiatrique, CHU Sainte-Justine, Université de Montréal, Montréal, Québec, Canada H3T 1C5 b Division of Pediatric Gastroenterology, Nationwide Children's Hospital, Ohio State University Columbus, OH 43205, USA Received 30 July 2008; revised 25 November 2008; accepted 28 November 2008 Key words: Antegrade enema; Cecostomy; Appendicostomy; Colonic motility; Colonic manometry; Constipation; Fecal incontinence Abstract Purpose: The aim of the study was to describe the changes in colonic motility occurring after chronic antegrade enema use in children and young adults. Methods: Colonic manometry tracings of patients who had used antegrade enemas for at least 6 months and were being evaluated for possible discontinuation of this treatment were retrospective reviewed. Results: Seven patients (median age of 12 years, range 3-15 years) met our inclusion criteria. Four patients had idiopathic constipation, 2 had tethered cord, and 1 had Hirschsprung disease. Colonic manometry before the use of antegrade enemas showed dysmotility in 6 (86%) children, mostly in the distal colon. None of the patients underwent colonic resection between the 2 studies. All the patients had colonic manometry repeated between 14 and 46 months after the creation of the cecostomy. All patients with abnormal colonic manometry improved with the use of antegrade enema with a complete normalization of colonic motility in 5 (83%) patients. Conclusion: Use of antegrade enema alone, without diversion or resection, may improve colonic motility. © 2009 Elsevier Inc. All rights reserved. Chronic constipation is a common problem worldwide in the pediatric population with a prevalence ranging from 0.7% to 29.6% [1]. Even if the most common cause of childhood constipation is functional fecal retention with no organic disease [2], constipation when severe can have significant adverse psychosocial consequences and great impact on quality of life [3,4]. Some patients need intensive medical treatment and represent a real challenge for the physician and for the family [5]. The antegrade continence enema (ACE) was originally described for treatment of fecal incontinence [6] and is now more widely applied to different forms of defecation disorders [7,8]. It consists of antegrade infusion of cleansing solutions in the proximal colon to provide a complete evacuation of feces at regular intervals. Youssef et al [8] reported that antegrade enemas can eventually be discontinued in selected patients after a mean of 14.6 months. However, it is not known whether the clinical improvement is associated with colonic motility normalization. The aim of the study was to review and compare colonic motility before and after antegrade enema use for at least 6 months in children and young adults. Corresponding author. Service de Chirurgie Pédiatrique, CHU Sainte- Justine, Université de Montréal, Montréal, Québec, Canada. E-mail address: [email protected] (A. Aspirot). www.elsevier.com/locate/jpedsurg 0022-3468/$ see front matter © 2009 Elsevier Inc. All rights reserved. doi:10.1016/j.jpedsurg.2008.11.061 Journal of Pediatric Surgery (2009) 44, 15751580

Antegrade enemas for defecation disorders: do they improve the colonic motility?

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Page 1: Antegrade enemas for defecation disorders: do they improve the colonic motility?

www.elsevier.com/locate/jpedsurg

Journal of Pediatric Surgery (2009) 44, 1575–1580

Antegrade enemas for defecation disorders: do theyimprove the colonic motility?Ann Aspirot a,b,⁎, Sergio Fernandez b, Carlo Di Lorenzo b, Beth Skaggs b, Hayat Mousa b

aService de Chirurgie Générale Pédiatrique, CHU Sainte-Justine, Université de Montréal, Montréal, Québec, Canada H3T 1C5bDivision of Pediatric Gastroenterology, Nationwide Children's Hospital, Ohio State University Columbus, OH 43205, USA

Received 30 July 2008; revised 25 November 2008; accepted 28 November 2008

Ju

0d

Key words:Antegrade enema;Cecostomy;Appendicostomy;Colonic motility;Colonic manometry;Constipation;Fecal incontinence

AbstractPurpose: The aim of the study was to describe the changes in colonic motility occurring after chronicantegrade enema use in children and young adults.Methods: Colonic manometry tracings of patients who had used antegrade enemas for at least 6 monthsand were being evaluated for possible discontinuation of this treatment were retrospective reviewed.Results: Seven patients (median age of 12 years, range 3-15 years) met our inclusion criteria. Fourpatients had idiopathic constipation, 2 had tethered cord, and 1 had Hirschsprung disease. Colonicmanometry before the use of antegrade enemas showed dysmotility in 6 (86%) children, mostly in thedistal colon. None of the patients underwent colonic resection between the 2 studies. All the patients hadcolonic manometry repeated between 14 and 46 months after the creation of the cecostomy. All patientswith abnormal colonic manometry improved with the use of antegrade enema with a completenormalization of colonic motility in 5 (83%) patients.Conclusion: Use of antegrade enema alone, without diversion or resection, may improve colonic motility.© 2009 Elsevier Inc. All rights reserved.

Chronic constipation is a common problem worldwide inthe pediatric population with a prevalence ranging from0.7% to 29.6% [1]. Even if the most common cause ofchildhood constipation is functional fecal retention with noorganic disease [2], constipation when severe can havesignificant adverse psychosocial consequences and greatimpact on quality of life [3,4]. Some patients need intensivemedical treatment and represent a real challenge for the

⁎ Corresponding author. Service de Chirurgie Pédiatrique, CHU Sainte-stine, Université de Montréal, Montréal, Québec, Canada.E-mail address: [email protected] (A. Aspirot).

022-3468/$ – see front matter © 2009 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2008.11.061

physician and for the family [5]. The antegrade continenceenema (ACE) was originally described for treatment of fecalincontinence [6] and is now more widely applied to differentforms of defecation disorders [7,8]. It consists of antegradeinfusion of cleansing solutions in the proximal colon toprovide a complete evacuation of feces at regular intervals.Youssef et al [8] reported that antegrade enemas caneventually be discontinued in selected patients after a meanof 14.6 months. However, it is not known whether theclinical improvement is associated with colonic motilitynormalization. The aim of the study was to review andcompare colonic motility before and after antegrade enemause for at least 6 months in children and young adults.

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1576 A. Aspirot et al.

1. Materials and methods

1.1. Study population

We retrospectively reviewed the colonic manometry tracingsof all the patients who were evaluated at the NationwideChildren's Hospital between 1999 and 2007 for defecationdisorders and who met the following inclusion criteria:

- Age birth to 21 years- Colonic manometry performed prior to placement ofcecostomy for treatment of severe constipation with orwithout fecal incontinence

- Use of antegrade enemas on a regular basis- Colonic manometry performed after at least 6 months oftreatment.

We identified 7 patients who met the criteria. All thepatients had failed maximal medical treatment including highdoses of osmotic laxatives (usually polyethylene glycol) andstimulants (senna and bisacodyl). They had received acecostomy placement to administer antegrade enemas.Medical records and colonic manometry studies werereviewed. This study was approved by the InstitutionalReview Board of Nationwide Children's Hospital, Colum-bus, Ohio.

Table 1 Patients characteristics

Patient Sex Age (y) Past medical history

1 Male 8 Tethered cord2 Male 11 None3 Male 5 None4 Female 4 Tethered cord5 Male 3 Hirschsprung disease6 Male 15 None7 Male 11 Attention deficit

hyperactivity disorder

1.2. Colonic manometry

All colonic manometries were performed according topreviously published protocols [9]. After a colonic cleanout, the manometry catheters were inserted into the colonand their position confirmed radiologically. The colonicmanometry catheters used had 8 recording sites spaced 10 to15 cm apart. The catheters were perfused using apneumohydraulic infusion system (Dentsleeve, Mississauga,Ontario, Canada), connected to a polygraph and computersoftware (Medtronics, Minneapolis, Minn). Colonic motilitywas recorded during fasting and 1 hour after ingestion of ahigh-caloric meal. Then, bisacodyl (0.2 mg/kg, max 10 mg)was administered through the central lumen of the catheteras a stimulant of colonic motility, and recordings werecontinued for another hour or until the catheter wasevacuated. High-amplitude propagating contractions(HAPC) were defined by an amplitude of at least 60 mmHg, a duration of 10 seconds or more, and an antegradepropagation over at least 30 cm. The motility patterns wereclassified as normal motility when HAPC occurred fromproximal to distal colon; distal dysmotility when HAPC didnot propagated to the distal colon (from splenic flexure tosigmoid); colonic inertia when no contractions occurred;and pancolonic dysmotility when only low-amplitude orsimultaneous contractions occurred (not fulfilling thecriteria for HAPC).

1.3. Cecostomy

The cecostomy was performed either percutaneously[10,11] or surgically with an open technique [12]. Acomplete description of the 2 techniques is beyond thescope of this article. The device was irrigated twice dailywith 10 mL of water for the first 2 weeks after the insertion,then regular antegrade irrigations were started. Irrigationsolutions used varied among different patients and weremodified over time according to the clinical response.Phosphate solutions followed by normal saline were used atthe beginning of our experience. Because of the risk ofhyperphosphatemia, they were subsequently replaced bypolyethylene glycol (starting with 10 mL/kg with the volumebeing increased until optimal result was achieved) mixedwith 10 to 20 mL bisacodyl enema. Antegrade continenceenemas were performed daily. Before considering theremoval of the cecostomy tube, the enema frequency wasdecreased progressively with close monitoring of thefrequency of bowel movement and consistency of the stools.

2. Results

Seven patients met our inclusion criteria, 6 males and 1female, with ages from 3 to 15 years (median age of 12 yearsold). All the patients had chronic constipation present forseveral years and had failed aggressive medical laxative andstimulant treatment and behavioral interventions. Threepatients presented with associated fecal incontinence. Comor-bid conditions included tethered cord in 2, Hirschsprungdisease in 1, and attention deficit hyperactivity disorder in 1.The patient with Hirschsprung disease had a short segmentform that had been resected at 6 weeks old (Soave pull-through). Three patients had no significant medical historybeside the problems related to defecation (Table 1).

Of the 7 patients, 6 had a barium enema, whichdemonstrated mild colonic dilatation in only one of thechildren. All the others had normal caliber colon. Patient 2 didnot have a barium enema but had a computed tomography(CT) scan performed showing rectosigmoid impaction with-out proximal dilatation. This patient had distal colonicdysmotility at the prececostomy manometry that improved

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Table 2 Result of manometry studies and clinical evolution

Patient Type ofcecostomy

Prececostomymanometry

Postcecostomy manometry Time betweenmanometries (mo)

Clinicalimprovement

1 Percutaneous Distal dysmotility Normal 14 Yes2 Percutaneous Distal dysmotility Distal dysmotility but higher amplitude 20 Yes3 Percutaneous Distal dysmotility Normal 14 Yes4 Surgical, open Distal dysmotility Normal 17 Yes5 Percutaneous Normal Simultaneous contractions 46 No6 Percutaneous Colonic inertia Normal 46 Yes7 Percutaneous Pan-colonic dysmotility Normal 52 Yes

1577Antegrade enemas for defecation disorders

but did not normalized at the postcecostomy manometryperformed 20 months after beginning the use of antegradeenemas. None of the patients had a radiologic evaluation ofthe entire colon after the creation of the cecostomy.

As shown in Table 2, prececostomy colonic manometrywas performed on all the patients and showed distaldysmotility in 4 of them (Fig. 1). One patient had pan-colonic dysmotility demonstrated by the presence of low-amplitude propagated contractions with the distal colonbeing more severely abnormal than the proximal colon. Onepatient had colonic inertia. Only 1 patient had a completelynormal colonic manometry before the insertion of thececostomy, meaning that 86% of the study patients hadcolonic dysmotility before the use of antegrade enemas,mostly involving the distal colon.

All the patients underwent creation of a cecostomy in ourCenter. Six were inserted percutaneously, and 1 wasperformed surgically.

Postcecostomy colonic manometry was performedbetween 14 and 52 months after beginning the use ofantegrade enemas (median time, 30 months). None of thepatients underwent colonic resection between the 2 studies.All patients with abnormal colonic manometry improvedwith the use of antegrade enema with a complete normal-ization of colonic motility in 5 (83%) of them (Fig. 2).Somewhat surprisingly, the patient who had a normalprececostomy colonic manometry had an abnormal post-cecostomy study that showed simultaneous contractions inthe descending colon. All patients except the one who hadabnormal motility clinically improved and had completeresolution of constipation and fecal incontinence after usingthe antegrade enemas. Two patients had the cecostomy tuberemoved, and 3 patients are progressively being weanedfrom the antegrade enemas. The other 2 patients continue touse daily antegrade enemas because of persistent dysmotility(1 deteriorated, 1 improved but not normalized).

3. Discussion

In this study, we report for the first time the improvementof colonic motility after using antegrade enemas. We foundthat using antegrade enemas on a regular basis in childrenand young adults with defecation disorders not only is

effective in improving the constipation and resolving thefecal incontinence but may also lead to normalization of apreviously abnormal colonic motility.

Cecostomy placement has been found to be beneficial forthe treatment of children with idiopathic constipation, spinalabnormalities, and imperforate anus [12]. It was found thatregular administration of antegrade enema increases fre-quency of defecation, decreases episodes of fecal incon-tinence, and improves quality of life. A recent study fromEurope confirmed the favorable outcome associated with theuse of ACEs with achievement of continence in 92% of thecases in their series (n = 48) and in 93% in their literaturereview (n = 676) [13]. The use of antegrade enemas can alsofacilitate behavioral interventions allowing for scheduledtoilet sitting and predictable passage of soft non painfulstools, avoiding the psychological trauma of anally invasiveinterventions (suppositories, enemas).

Colonic manometry is deemed to be a useful diagnostictool for the investigation of severe chronic constipation inchildren [9,14]. Our group reported earlier that results fromcolonic manometry studies are reliable predictors of cecost-omy success [15]. In that study, patients with generalizedcolonic dysmotility were less likely to benefit from the use ofantegrade enemas. Guided by these findings, it has been ourpractice to recommend use of antegrade enemas only topatients who do not have colonic inertia and who demonstratean identifiable colonic motor response to bisacodyl.

In selected patients, the use of antegrade enemas may bereduced over time and discontinued. In a small series ofpatients, 41.7% discontinued antegrade enemas at a mean of14.6 months after beginning treatment [8]. However, there isno published study using postcecostomy manometry toconfirm that the clinical improvement is associated withimproved colonic motility. A similar study has beenpublished by Villarreal et al [16] in patients with colonicdiversion who were evaluated with colonic manometrybefore closure of their ostomy. Of the 12 patients, 4recovered normal colonic motility after diversion and werereanastomosed without resection.

In our study, we demonstrated that the colonic motility canimprove after using antegrade enemas for a variable period oftime of at least 1 year. The reason why the motility improvedcan not be elucidated by our study but does not seem to resultsolely from resolution of chronic colonic dilatation. Colonic

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Fig. 1 Distal colonic dysmotility.

1578 A. Aspirot et al.

dilatation is a subjective observation usually based onradiologic studies. When assessed during simple abdominalradiograph or CT scan, the dilation can be because of thepresence of impacted stools and resolve after a clean out. Onthe other hand, barium enemas provide more information onthe compliance of the colonic wall and show variable degreeof dilatation depending on the amount of contrast infused andthe pressure generated intraluminally. Whether the improve-ment of the motility is simply the result of resolution of thedilatation remains a possibility. In most of our cases, it isunlikely that colonic dilatation was responsible for the

prececostomy dysmotility because the single patient withdemonstrated mild colonic dilatation on a barium enema hada normal prececostomy manometry. For the same reason, it isalso unlikely that the size of the colon had an impact on thepressure recorded during the manometry study. In the patientwith rectosigmoid impaction at the CT scan, the prececost-omy manometry showed distal dysmotility and the post-cecostomy improved but did not normalize. This patientnever had a barium enema to assess the colonic caliber.

Other possible explanation of the postcecostomy improve-ment of the motility could be related to resolution of chronic

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Fig. 2 Normal colonic motility.

1579Antegrade enemas for defecation disorders

stools stasis in the colon. Recently, methane production hasbeen correlated with the severity of constipation [17].Although hypothetic, it is possible that other substancesassociated with stasis have an effect on the colonic motility.Another possible explanation could be maturation of orresolution of injury to the enteric nervous system over time.Our series is too small to try to correlate the degree andrapidity of improvement with the age of the patient, durationof symptoms before placement of the cecostomy, or theduration of treatment with antegrade enemas, but thesefactors could be evaluated with a larger cohort.

It should also be emphasized that we are reporting ahighly selected group of patients. Most of the studied patientswere doing well clinically, and the manometry was repeatedto corroborate the decision of discontinuing the use ofantegrade enemas. Comorbid conditions in our patientspotentially contributed to but were probably not the mainetiology for the constipation. Obviously, ACE cannotprovide a cure for attention deficit disorder or tetheredcord. However, we objectively observed by colonicmanometry an improvement in colonic motility in thosepatients. The indication for repeating the manometry for

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patient 5, who had deteriorated, was because he was stillhaving problems despite the use of the antegrade enemas.This patient had been born with Hirschsprung disease.Although his first colonic manometry was normal, hisclinical deterioration correlated with the deterioration of hiscolonic motility as assessed by manometry. In that case, wehypothesize that this patient had a progressive motilitydisorder. A prospective study that would restudy system-atically all the patients after a standard period of antegradeenema use would be best suited to confirm our encouragingresults. In conclusion, our case series suggests that the use ofantegrade enema alone, without diversion or resection, mayimprove colonic motility in children and young adults.

References

[1] van den Berg MM, Benninga MA, Di Lorenzo C. Epidemiology ofchildhood constipation: a systematic review. Am J Gastroenterol 2006;101:2401-9.

[2] Rasquin-Weber A, Hyman PE, Cucchiara S, et al. Childhoodfunctional gastrointestinal disorders. Gut 1999;45(Suppl 2):II60-8.

[3] Youssef NN, Langseder AL, Verga BJ, et al. Chronic childhoodconstipation is associated with impaired quality of life: a case-controlled study. J Pediatr Gastroenterol Nutr 2005;41:56-60.

[4] Clarke MC, Chow CS, Chase JW, et al. Quality of life in children withslow transit constipation. J Pediatr Surg 2008;43:320-4.

[5] LiemO,BenningaMA,MousaHM, et al. Novel and alternative therapiesfor childhood constipation. Curr Gastroenterol Rep 2007;9:214-8.

[6] Malone PS, Ransley PG, Kiely EM. Preliminary report: the antegradecontinence enema. Lancet 1990;336:1217-8.

[7] Griffiths DM, Malone PS. The Malone antegrade continence enema.J Pediatr Surg 1995;30:68-71.

[8] Youssef NN, Barksdale Jr E, Griffiths JM, et al. Management ofintractable constipation with antegrade enemas in neurologically intactchildren. J Pediatr Gastroenterol Nutr 2002;34:402-5.

[9] Di Lorenzo C, Hillemeier C, Hyman P, et al. Manometry studies inchildren: minimum standards for procedures. NeurogastroenterolMotil 2002;14:411-20.

[10] Chait PG, Shandling B, Richards HM, et al. Fecal incontinence inchildren: treatment with percutaneous cecostomy tube placement—aprospective study. Radiology 1997;203:621-4.

[11] Chait PG, Shlomovitz E, Connolly BL, et al. Percutaneous cecostomy:updates in technique and patient care. Radiology 2003;227:246-50.

[12] Mousa HM, van den Berg MM, Caniano DA, et al. Cecostomy inchildren with defecation disorders. Dig Dis Sci 2006;51:154-60.

[13] Sinha CK, Grewal A, Ward HC. Antegrade continence enema (ACE):current practice. Pediatr Surg Int 2008;24:685-8.

[14] Baker SS, Liptak GS, Colletti RB, et al. Constipation in infants andchildren: evaluation and treatment. A medical position statement of theNorth American Society for Pediatric Gastroenterology and Nutrition.J Pediatr Gastroenterol Nutr 1999;29:612-26.

[15] van den Berg MM, Hogan M, Caniano DA, et al. Colonic manometryas predictor of cecostomy success in children with defecationdisorders. J Pediatr Surg 2006;41:730-6 [discussion 730-736].

[16] Villarreal J, Sood M, Zangen T, et al. Colonic diversion for intractableconstipation in children: colonic manometry helps guide clinicaldecisions. J Pediatr Gastroenterol Nutr 2001;33:588-91.

[17] Chatterjee S, Park S, Low K, et al. The degree of breath methaneproduction in IBS correlates with the severity of constipation. Am JGastroenterol 2007;102:837-41.