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Di Lorenzo C. Faecal incontinence: Retentive, non-retentive and when to suspect organic pathology. Aliment Pharmacol Ther 2024; 60 Suppl 1:S54-S65. [PMID: 38924573 DOI: 10.1111/apt.17832] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/30/2023] [Revised: 11/13/2023] [Accepted: 11/28/2023] [Indexed: 06/28/2024]
Abstract
BACKGROUND Faecal incontinence is a highly prevalent and very distressing condition that occurs throughout the entire paediatric age. AIM To summarise advances in the understanding of the epidemiology, pathophysiology, evaluation and treatment of children with faecal incontinence due to either disorders of gut-brain interaction or organic diseases. METHODS Literature review on prevalence, impact, diagnosis and treatment options for children with faecal incontinence, interspersed with observations from the author's lifelong career focused on evaluation of children with motility disorders. RESULTS Faecal incontinence in children is most commonly due to unrecognised or insufficiently treated functional constipation with overflow incontinence. Non-retentive faecal incontinence (NRFI) is probably more common than previously thought and is particularly challenging to treat. Organic diseases such as anorectal malformations (ARMs), Hirschsprung disease and spinal defects are often associated with faecal incontinence; in these conditions, faecal incontinence has a profound impact on quality of life. Recognition of the different pathophysiologic mechanisms causing the incontinence is essential for a successful treatment plan. A thorough physical examination and history is all that is needed in the diagnosis of the causes of faecal incontinence related to disorders of gut-brain interaction. Colonic transit studies or x-rays may help to differentiate retentive from NRFI. Manometry tests are helpful in determining the mechanisms underlying the incontinence in children operated on for ARMs or Hirschsprung diseases. Multiple behavioural, medical and surgical interventions are available to lessen the severity of faecal incontinence and its impact on the daily life of affected individuals. CONCLUSIONS Recent advances offer hope for children with faecal incontinence.
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Affiliation(s)
- Carlo Di Lorenzo
- Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Nationwide Children's Hospital, Columbus, Ohio, USA
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Keshtgar AS, Selim IM. Role of 3D high resolution anorectal manometry compared to conventional technique in management of constipation and fecal incontinence in children. J Pediatr Surg 2023; 58:251-257. [PMID: 36414477 DOI: 10.1016/j.jpedsurg.2022.10.018] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/04/2022] [Accepted: 10/11/2022] [Indexed: 11/21/2022]
Abstract
BACKGROUND Anorectal manometry is a valuable tool for objective assessment of motility motor and sensory function of the anorectum. The aim of this study was to investigate role of water-perfused (WP) three dimensional high-resolution anorectal manometry (3D-HRARM) compared to WP conventional manometry (CM) in the management of chronic idiopathic constipation (CIC) and faecal incontinence (FI) in children. METHODS This was a retrospective review of 122 consecutive children, who had WP 8-channel CM or 24 channel 3D-HRARM and endosonography under ketamine anaesthesia from September 2012 to February 2019. All patients had a validated symptom severity score questionnaire ranging from 0 (best) to 65 (worst). Mann-Whitney-U test and Spearman rank test were used and p<0.05 was considered significant. RESULTS Subjects were divided according to investigation: CM group (n = 75) and 3D-HRAM (n = 47), who were otherwise comparable. Median anal resting pressure was 50 mmHg and rectoanal inhibitory reflex (RAIR) threshold volume was 10mls across the entire cohort. There were no significant differences in resting pressure or RAIR threshold when using conventional or 3D-HRARM. Rectal capacity was significantly higher in the CM group (p = 0.002). Rectal capacity and internal anal sphincter (IAS) thickness positively correlated with symptom severity, duration and patient age. 3D-HRARM provided a more detailed depiction of the anorectal pressure profile. Botulinum toxin was injected into the external anal sphincter (EAS) muscle (n = 75, 61%) and an anterograde colonic enema (ACE) stoma was needed subsequently in 19 (16%) to treat recurrent constipation and soiling symptoms. Rectal biopsy was done in 43(35%) patients, if RAIR was absent or inconclusive. CONCLUSION CM and 3D-HRARM are comparable in terms of resting pressure and RAIR threshold. 3D-HRAM is safe and provides detailed functional morphology of anal sphincters and it is useful to understand underlying pathophysiology of constipation and faecal incontinence in children and plan further treatment. LEVEL OF EVIDENCE Level I.
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Affiliation(s)
- Alireza S Keshtgar
- Evelina London Children's Hospital, Guy's and St. Thomas' NHS Foundation Trust, London, United Kingdom; Kings College London, Guy's and St. Thomas' NHS Foundation Trust, London, United Kingdom.
| | - Iman M Selim
- Kings College London, Guy's and St. Thomas' NHS Foundation Trust, London, United Kingdom
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Athanasakos E, Cleeve S. Anorectal Manometry, Conventional and High Resolution in Paediatrics. ANO-RECTAL ENDOSONOGRAPHY AND MANOMETRY IN PAEDIATRICS 2022:55-73. [DOI: 10.1007/978-3-030-97668-2_6] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/03/2025]
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Yates G, Friedmacher F, Cleeve S, Athanasakos E. Anorectal manometry in pediatric settings: A systematic review of 227 studies. Neurogastroenterol Motil 2021; 33:e14006. [PMID: 33118295 DOI: 10.1111/nmo.14006] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/26/2020] [Revised: 09/03/2020] [Accepted: 09/14/2020] [Indexed: 12/20/2022]
Abstract
BACKGROUND There is wide variation in the clinical use of diagnostic tools for children with chronic constipation and functional/structural fecal incontinence (CCFSFI). Anorectal manometry (ARM) is a well-recognized technique to assess the function of the anorectum. PURPOSE Our aim was to perform an up-to-date review on ARM in pediatric patients with CCFSFI, with specific focus on the indication of use and protocol. Variation of its use in pediatrics will be explored. METHODS A systematic search was conducted for empirical studies utilizing ARM with a pediatric sample. A keyword search of literature published in English before July 2018 was conducted and updated to October 2019. Data on demographics, clinical information, study aims, ARM parameters and use of sedation/anesthesia were collected. KEY RESULTS A total of 227 studies were included in this systematic review. The age of study participants at the time of ARM ranged from birth to 18 years. ARM was most commonly used in patients with organic conditions (65%) compared to functional constipation (41%). In almost half [108/227 (48%)] of the studies, ARM was performed awake. The ARM parameters most frequently assessed were the rectoanal inhibitory reflex, which was evaluated in 198/227 studies (87%) and the anal resting pressure [166/227 studies (73%)]. CONCLUSIONS AND INFERENCES This systematic review has highlighted the vast variation of ARM use within pediatrics and the need to strive toward standardization and use of consensus guidelines. We anticipate this will further advance our understanding of the pathophysiological mechanisms involved in children with defecation disorders.
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Affiliation(s)
- Gregory Yates
- Department of Paediatric Surgery, The Royal London Hospital, London, UK.,Barts and The London, School of Medicine and Dentistry, London, UK
| | - Florian Friedmacher
- Department of Paediatric Surgery, The Royal London Hospital, London, UK.,Barts and The London, School of Medicine and Dentistry, London, UK.,Department of Pediatric Surgery, Goethe University Frankfurt, University Hospital Frankfurt, Frankfurt am Main, UK
| | - Stewart Cleeve
- Department of Paediatric Surgery, The Royal London Hospital, London, UK.,Barts and The London, School of Medicine and Dentistry, London, UK
| | - Eleni Athanasakos
- Department of Paediatric Surgery, The Royal London Hospital, London, UK.,Barts and The London, School of Medicine and Dentistry, London, UK
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Alessandrella A, Turco R, Russo M, Poziello A, Miele E, Staiano A. High-resolution anorectal manometry in children with functional constipation with or without fecal incontinence. Neurogastroenterol Motil 2020; 32:e13882. [PMID: 32476213 DOI: 10.1111/nmo.13882] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/22/2019] [Revised: 03/29/2020] [Accepted: 04/21/2020] [Indexed: 12/20/2022]
Abstract
BACKGROUND High-resolution anorectal manometry (HR-ARM) is expected to be better than conventional manometry. Our aim was to characterize HR-ARM pressures in children with functional constipation (FC), with or without fecal incontinence (FI). METHODS Children with diagnosis of FC, with or without FI, according to Rome-IV criteria, were enrolled. All patients underwent HR-ARM using 24-channel water-perfused catheter. RESULTS Twenty-nine consecutive children (M/F: 21/8; mean age ± SD: 9.5 ± 3.1 years; range 4-15), of whom 21 affected by FC without FI (mean age ± SD: 9.3 ± 3.23 years) and 8 affected by FC with FI (mean age ± SD: 10.2 ± 3.08 years), were enrolled. No significant differences were found regard to gender and age. The analysis of HR-ARM 3D plots demonstrated asymmetry of the anal canal, with higher pressures in distal halves. Comparing pressures between the two groups, we found lower values in FC with FI than in FC without FI group, with a statistically significance for maximum and mean resting pressures (P = .032 and P = .008, respectively). When evaluating our study population respect to asymptomatic children, we found lower resting pressures, lower maximum squeeze pressure, and higher rectoanal inhibitory reflex (RAIR) values. CONCLUSIONS Our data demonstrate that HR-ARM pressures at rest and during squeezing in FC with FI children are lower than FC without FI subjects, particularly in anteroposterior quadrants. Compared to children without lower gastrointestinal symptoms, children with FC with or without FI show lower pressures and higher values of RAIR.
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Affiliation(s)
- Annalisa Alessandrella
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
| | - Rossella Turco
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
| | - Marina Russo
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
| | - Antonio Poziello
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
| | - Erasmo Miele
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
| | - Annamaria Staiano
- Section of Pediatrics, Department of Translational Medical Science, University of Naples "Federico II", Naples, Italy
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Scientific solution to a complex problem: physiology and multidisciplinary team improve understanding and outcome in chronic constipation and faecal incontinence. Pediatr Surg Int 2020; 36:295-303. [PMID: 31844977 DOI: 10.1007/s00383-019-04605-y] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 12/06/2019] [Indexed: 01/25/2023]
Abstract
PURPOSE There is a lack of diagnostic credibility to direct focused management for children with chronic constipation (CC) and faecal incontinence (FI). The aim is to assess the impact of an innovative Children's Anorectal Physiology Service (CAPS) focusing on improving outcomes in children with CC/FI. METHODS Prospective data: demographics, bowel and quality of life (QoL)/risk of distress questionnaires. Diagnostics: awake high-resolution anorectal manometry (AHRAM), endoanal ultrasound and transit marker studies (TMS). RESULTS Total patients: 112; 66 males (59%); median 9 years (17 months to 16 years). Patient groups included: 89 (79%) had functional CC/FI; 9 (8%), Hirschsprung's disease; 12 (11%), anorectal malformations and 2 (2%), trauma. St Marks Incontinence score (SMIS) abnormal in 91 (81%) and Cleveland Constipation Score (CCS) in 101 (90%) patients. Anorectal manometry: 94 (84%) awake and 18 (17%) under anaesthesia. Play specialist input 37 (33%) patients. AHRAM abnormal 65 (58%): sphincter dysfunction 36 (32%) and altered rectal sensation: hyposensitive 22% (20/91); 21% (19/91) hypersensitive. TMS normal in 64 (57%), 17 (15%) slow transit and 27 (24%) rectal evacuatory disorder. Risk of distress in 38% and poor QoL in 55% patients which correlated with abnormal SMIS (p = 0.02). Patient/parent satisfaction improved significantly (p < 0.05). CONCLUSIONS Scientific investigations combined with multidisciplinary team improve patient satisfaction and reduces patient self-report illness severity. A complex problem requires a scientific solution.
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Gfroerer S, Rolle U. Pediatric intestinal motility disorders. World J Gastroenterol 2015; 21:9683-9687. [PMID: 26361414 PMCID: PMC4562951 DOI: 10.3748/wjg.v21.i33.9683] [Citation(s) in RCA: 27] [Impact Index Per Article: 2.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/29/2015] [Revised: 05/07/2015] [Accepted: 07/03/2015] [Indexed: 02/06/2023] Open
Abstract
Pediatric intestinal motility disorders affect many children and thus not only impose a significant impact on pediatric health care in general but also on the quality of life of the affected patient. Furthermore, some of these conditions might also have implications for adulthood. Pediatric intestinal motility disorders frequently present as chronic constipation in toddler age children. Most of these conditions are functional, meaning that constipation does not have an organic etiology, but in 5% of the cases, an underlying, clearly organic disorder can be identified. Patients with organic causes for intestinal motility disorders usually present in early infancy or even right after birth. The most striking clinical feature of children with severe intestinal motility disorders is the delayed passage of meconium in the newborn period. This sign is highly indicative of the presence of Hirschsprung disease (HD), which is the most frequent congenital disorder of intestinal motility. HD is a rare but important congenital disease and the most significant entity of pediatric intestinal motility disorders. The etiology and pathogenesis of HD have been extensively studied over the last several decades. A defect in neural crest derived cell migration has been proven as an underlying cause of HD, leading to an aganglionic distal end of the gut. Numerous basic science and clinical research related studies have been conducted to better diagnose and treat HD. Resection of the aganglionic bowel remains the gold standard for treatment of HD. Most recent studies show, at least experimentally, the possibility of a stem cell based therapy for HD. This editorial also includes rare causes of pediatric intestinal motility disorders such as hypoganglionosis, dysganglionosis, chronic intestinal pseudo-obstruction and ganglioneuromatosis in multiple endocrine metaplasia. Underlying organic pathologies are rare in pediatric intestinal motility disorders but must be recognized as early as possible.
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Longitudinal and radial characteristics of intra-anal pressures in children using 3D high-definition anorectal manometry: new observations. Am J Gastroenterol 2013; 108:1918-28. [PMID: 24169274 DOI: 10.1038/ajg.2013.361] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/24/2013] [Accepted: 09/14/2013] [Indexed: 12/11/2022]
Abstract
OBJECTIVES The pathophysiology of fecal incontinence is not well understood. Standard or high-resolution anorectal manometry (ARM) provides simple two-dimensional (2D) intra-anal pressure measurements and do not identify radial asymmetry or localize abnormal sphincter function. 3D high-definition ARM (HDARM) has 256 pressure sensors distributed circumferentially and provides a detailed topographical and 3D pressure gradient representation of the anal canal. The objective of this study was to use HDARM to characterize intra-anal pressure profiles in children during rest and squeeze. METHODS HDARM manometric tracings of 30 children with constipation referred for ARM were reviewed. 2D pressure profiles using high-resolution manometry were used to measure the length of the high-pressure zone (HPZ). The HPZ was divided into four equal segments from the anal verge to adjust for the variable sphincter length. Longitudinal and radial measurements of the HPZ during rest and squeeze (anterior, left, posterior, right quadrants of the HPZ) were taken along each segment in 2D and 3D topographical views. A 3D reconstruction combining all patients was then constructed. RESULTS Mean age was 149.3±1.8 months and mean HPZ length was 3.0±0.1 cm. Using 2D manometry, the mean peak HPZ pressure at rest was 72.0±2.5 mm Hg, and was located in the second segment of the HPZ. The mean peak HPZ pressure at squeeze was 202.9±13.1, and was located in the second segment. 3D measurement demonstrated both longitudinal and radial asymmetry along the anterior, left, posterior, and right quadrants of the HPZ. Left and right quadrant pressures were higher than anterior and posterior pressures at the anal verge and segment 1 during rest and squeeze. Anterior pressures were lower than posterior pressures longitudinally and radially in segments 2, 3, and 4 both during rest and squeeze. Our findings also suggest that in pediatrics it may be necessary to adjust pressure measurement to the anal canal length to get a more accurate picture. CONCLUSIONS 3D HDARM allows for a detailed characterization of intra-anal pressures. 3D topographic pressure measurements demonstrate longitudinal and radial asymmetry of the anal canal at rest and during squeeze. This is the first time longitudinal and radial asymmetry of the anal canal has been described in children. 3D HDARM may allow for a better understanding of the mechanisms of fecal continence in children.
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Rajindrajith S, Devanarayana NM, Benninga MA. Review article: faecal incontinence in children: epidemiology, pathophysiology, clinical evaluation and management. Aliment Pharmacol Ther 2013; 37:37-48. [PMID: 23106105 DOI: 10.1111/apt.12103] [Citation(s) in RCA: 55] [Impact Index Per Article: 4.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/05/2012] [Revised: 05/23/2012] [Accepted: 10/02/2012] [Indexed: 12/12/2022]
Abstract
BACKGROUND Faecal incontinence (FI) in children is a significant gastrointestinal problem, with great personal and social impacts. It is characterised by recurrent loss of faecal matter into the underwear. Both functional and organic causes contribute to its aetiology with the former predominating. AIM To review the epidemiology, pathophysiology, clinical evaluation and management of functional faecal incontinence in children. METHODS A PubMed search was conducted using search terms f(a)ecal incontinence, and encopresis. Articles on epidemiology, pathophysiology, clinical evaluation, investigation and management of functional FI in children were retrieved and assessed. RESULTS Community prevalence of this distressing problem ranges from 0.8% to 7.8% globally. Male: female ratio varies from 3:1 to 6:1. The diagnosis of FI is often based on established clinical criteria. The majority (82%) have constipation associated functional FI. Biopsychosocial factors play a crucial role in the pathogenesis. Limited physiological testing of anorectal function is recommended in the diagnostic procedures, particularly in children with atypical symptoms and possible organic disorders. Management of FI needs a multidisciplinary approach which includes establishment of an effective doctor-patient partnership, understanding the underlying mechanisms, pharmacotherapy and behavioural treatment. Approximately 15% of children with functional nonretentive faecal incontinence (FNRFI) had the same symptoms at the age of 18 years. CONCLUSION Significant therapeutic advances have been made for retentive faecal incontinence, but treatment options for functional nonretentive faecal incontinence are limited. Limited long-term outcome data show that the majority outgrow faecal incontinence. A substantial proportion of children progress to adulthood with faecal incontinence.
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Affiliation(s)
- S Rajindrajith
- Department of Paediatrics, Faculty of Medicine, University of Kelaniya, Ragama, Sri Lanka.
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Balloon expulsion test as a screen for outlet obstruction in children with chronic constipation. J Pediatr Gastroenterol Nutr 2013; 56:23-6. [PMID: 22847462 DOI: 10.1097/mpg.0b013e31826a909f] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/10/2022]
Abstract
OBJECTIVE Chronic constipation (CC) is a common problem in pediatrics and is often the result of obstructed defecation. The aim of the present study was to study the feasibility and efficacy of the balloon expulsion test (BET) in the diagnosis and management of children with CC. METHODS Retrospective study comparing BET and high-resolution anorectal manometry (ARM). The BET was done together with ARM in 29 children, ages 8 to 19 years, with CC. For BET, a 60-mL balloon was used. Passage of balloon in 1 minute or less was considered normal. RESULTS Fifteen of the 29 children had a normal BET. Of these, 14 also had an ARM, all of which were normal (except for 2 cases with a hypertonic baseline anal sphincter). Thus 12 of 14 with BET and ARM were normal on both (correlation between the tests 86%). Of the 14 children that failed BET, 10 had distal abnormalities by ARM, contrast studies, EMG, or assessment by a pelvic physical therapist. All of the patients with a nonrelaxing sphincter or outlet obstruction were treated with laxatives, anal sphincter Botox, and/or pelvic physical therapy and biofeedback. In follow-up of at least 3 months, all of the patients with a failed BET were improved. CONCLUSIONS We found a high correlation between a normal ARM and BET. If the BET is abnormal and the ARM does not identify a cause for the distal obstruction, additional studies may be needed, including contrast enema, defecography, or electromyography. BET appears to be a safe, reliable, and useful test in the evaluation and management of CC in children.
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Abstract
Chronic pelvic pain syndrome is a poorly understood clinical entity associated with urinary symptoms, pelvic floor dysfunction, and multisystem disorders. Treatment of pelvic floor dysfunction is difficult and often frustrating for the patient as well as for the involved physician. The purpose of this review is to update clinicians on the latest research for the treatment of pelvic floor dysfunction in relation to chronic pelvic pain syndrome.
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