Babette Peeters

Sacral Neuromodulation Therapy: A Promising Treatment for Adolescents With Refractory Functional Constipation

BACKGROUND: Sacral neuromodulation therapy has been successfully applied in adult patients with urinary and fecal incontinence and in adults with constipation not responding to intensive conservative treatment. No data, however, are available on sacral neuromodulation therapy as a treatment option in adolescents with refractory functional constipation. OBJECTIVES: This study aimed to describe the short-term results of sacral neuromodulation in adolescents with chronic functional constipation refractory to intensive conservative treatment. DESIGN: This is a retrospective review. SETTING: This study took place at the Department of Surgery, Maastricht University Medical Centre, The Netherlands. PATIENTS: Thirteen patients (all girls, age 10–18 years) with functional constipation according to the ROME III criteria not responding to intensive oral and rectal laxative treatment were assigned for sacral neuromodulation. MAIN OUTCOME MEASURES: When improvement of symptoms was observed during the testing phase, a permanent stimulator was implanted. Patients were prospectively followed up to at least 6 months after implantation of the permanent stimulator by interviews, bowel diaries, and Cleveland Clinic constipation score. Improvement was defined as spontaneous defecation ≥2 times a week. RESULTS: At presentation, none of the patients had spontaneous defecation or felt the urge to defecate. All patients had severe abdominal pain. Regular school absenteeism was present in 10 patients. After the testing phase, all but 2 patients had spontaneous defecation ≥2 times a week with a reduction in abdominal pain. After implantation, 11 (of 12) had a normal spontaneous defecation pattern of ≥2 times a week without medication, felt the urge to defecate, and perceived less abdominal pain without relapse of symptoms until 6 months after implantation. The average Cleveland Clinic constipation score decreased from 20.9 to 8.4. One lead revision and 2 pacemaker relocations were necessary. LIMITATIONS: This study is limited by its small sample size, single-institution bias, and retrospective nature. CONCLUSION: Sacral neuromodulation appears to be a promising new treatment option in adolescents with refractory functional constipation not responding to intensive conservative therapy. Larger randomized studies with long-term follow-up are required.

Infantile hypertrophic pyloric stenosis—genetics and syndromes

Infantile hypertrophic pyloric stenosis (IHPS) is a common condition in neonates that is characterized by an acquired narrowing of the pylorus. The aetiology of isolated IHPS is still largely unknown. Classic genetic studies have demonstrated an increased risk in families of affected infants. Several genetic studies in groups of individuals with isolated IHPS have identified chromosomal regions linked to the condition; however, these associations could usually not be confirmed in subsequent cohorts, suggesting considerable genetic heterogeneity. IHPS is associated with many clinical syndromes that have known causative mutations. Patients with syndromes associated with IHPS can be considered as having an extreme phenotype of IHPS and studying these patients will be instrumental in finding causes of isolated IHPS. Possible pathways in syndromic IHPS include: (neuro)muscular disorders; connective tissue disorders; metabolic disorders; intracellular signalling pathway disturbances; intercellular communication disturbances; ciliopathies; DNA-repair disturbances; transcription regulation disorders; MAPK-pathway disturbances; lymphatic abnormalities; and environmental factors. Future research should focus on linkage analysis and next-generation molecular techniques in well-defined families with multiple affected members. Studies will have an increased chance of success if detailed phenotyping is applied and if knowledge about the various possible causative pathways is used in evaluating results.

Stressful Life Events in Children With Functional Defecation Disorders.

Objectives: The aim of the study was to determine the prevalence of stressful life events including (sexual) abuse in children with functional defecation disorders by performing a systematic review. Methods: We searched MEDLINE, EMBASE, and PsycINFO for cohort, case-control and cross-sectional studies investigating the prevalence of stressful life events, including (sexual) abuse in children with functional defecation disorders. Results: The search yielded 946 articles, of which 8 were included with data from 654 children with functional constipation and 1931 children with (constipation-associated) fecal incontinence (FI). Overall, children with functional defecation disorders had been significantly more exposed to stressful life events than healthy children, with prevalence rates ranging from 1.6% to 90.9%. Being bullied, being a relational victim, interruption of toilet training, punishment by parents during toilet training, and hospitalization were significantly related to FI, whereas separation from the best friend, failure in an examination, severe illness in a close family member, loss of job by a parent, frequent punishment, and living in a war-affected area were significantly related to constipation. Only 1 study measured the prevalence of child abuse, which reported a significantly higher prevalence of child (sexual) abuse in children with FI compared with controls. Conclusions: The prevalence of stressful life events, including (sexual) abuse is significantly higher in children with functional defecation disorders compared with healthy children. To gain more insight into the true prevalence of child (sexual) abuse in children with functional defecation disorders, more studies are clearly needed.

Parental knowledge of fecal incontinence in children.

Objectives: Fecal incontinence (FI) is a common and stressful symptom of constipation in children. Recurrent FI causes psychological and physiological changes, complicating treatment as symptom duration progresses; however, parental misconceptions about the causes of FI may delay seeking medical care. The aim of the present study was to assess parental knowledge about FI and determine how this relates to the care and treatment of FI. Methods: A questionnaire was developed from qualitative interviews and clinician input. The questionnaire was administered to 251 parents and tested for reliability and validity. Subscales were compared between parents who did and did not consult a clinician. In addition, 30 parents completed the questionnaire before and after consultation with a pediatric gastroenterologist and after 2 months of treatment. Results: Two subscales were identified with good psychometric properties: “Blame and Punish” and “Worry and Help.” Families who consulted a physician for their childs FI acknowledged the role of constipation and scored higher on Worry and Help (mean 36.4 vs mean 46.9; P < 0.0001). Trends were found for Blame and Punish to decrease after consultation with a pediatric gastroenterologist (mean 1.7 vs 1.5; P = 0.08) and after 2 months of treatment (mean 1.5; P = 0.08). Conclusions: Parental knowledge about FI changes with physician consultation. These findings can help in developing educational materials for parents to encourage early diagnosis and treatment and prevent chronic problems that are difficult to manage.

Mo1216 Personality, Psychological Distress Physical Health and Child Rearing Practices of Parents of Children With Functional Constipation

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Su2071 Unraveling the Pathophysiology of Infantile Hypertrophic Pyloric Stenosis

Introduction: The aging, regarded as dynamic and progressive phenomenon, have been increasingly observed in the world population (Martins et al., 2009). It is essential for any health care, which is identified indications of obesity and visceral adiposity levels (France et al., 2008), providing guidelines to help prevent and alleviate this situation, therefore preventing future illnesses that can lead to death. Objective: Investigate the effect of physical exercise program on height (H), weight (W), fat mass (FM), visceral fat area (VFA), and skeletal muscle mass index (SMMI) of post-menopausal women. Methods: One hundred sixty-nine women (56.80±6.47 years,) were randomized into an exercise group (EG, n=91) and the control group (CG, n=78). The EG performed 60 minutes of exercise, 3 times a week (step, weight training and flexibility).Were evaluated, by using octopolar bioimpedance, before and after a 12-month period heigth (H), weight (W), fat mass (FM), fat-free mass (FFM), visceral fat area (VFA) and skeletal muscle mass index (SMI). Skeletal muscle mass index was calculated (SMI= SM/W ×100) and the food record method was used. The variable averages (absolute values and rates of change) were compared by using t-tests and the degree of statistical significance considered was 1%. Results: The EG revealed a significant increase on height, evolving the average 155.61 cm in the pre-test to 156.20 in post-test. The percentage of obese women on EG, in the post-test, was reduced by 1.1% in fat mass, being 80 women (87.9%) remained with values of AAV≥ 100 cm2. The CG increased (p <0.01) total fat (1.86% FM) and central (3.92 cm2), raising the percentage of obese women in the post-test at 6.4%. The reduction in CG (SMMI) was -1.06 (p <0.01), with the number of women with sarcopenia almost doubled (from 10.3% in the pre-test to 19.2% in the posttest). Conclusion: The exercise of strength, endurance and flexibility suggests an improvement in posture, reduces global and central obesity, preventing sarcopenia increased, reducing the risk of falls, implying possible benefits for the quality to the life of this population. Mean and standart deviation of variables in both groups and differences between them

Mo1993 The Prevalence of Stressful Life Events Including Sexual Child Abuse in Children With Functional Defecation Disorders: A Systematic Review

Background: Early life exposure to antibiotics might alter the development of the host microbiome and influence Crohns disease (CD) risk. Although this association has been suggested by a few recent studies, larger studies with longer duration of follow-up and more rigorous control of confounding are needed. We sought to further evaluate the association between antibiotic use during the first year of life and subsequent CD by performing a nationwide birth cohort study in Denmark.Methods: All children born in Denmark between 1995 and 2007 were identified in the Danish Medical Birth Registry, and followed until death, emigration, or end of study (January 1, 2011). Outpatient antibiotic use was identified using the National Prescription Registry. Cases of CD were identified using the Danish National Patient Registry; a subset was confirmed by linkage to the National Pathology Registry. As a negative control, we analyzed exposure to systemic adrenergic agents, the second most commonly prescribed class of medications in the first year of life. Relative risk was determined by Cox proportional hazards models, after adjusting for route of delivery, birth order, gender, birth year, and family history of CD.Results: The birth cohort consisted of 850,962 children followed for a mean of 8.5 years (7,206,533 person-years of follow up). Of these, 51.3% were male, 17.5% were born by c-section, 1.0% had a family history of CD and 41.1% had ≥ 1 filled antibiotic prescription in the first year of life. 291 cases of CD were observed during follow-up. A positive association between antibiotic use and CD did not reach statistical significance (adjusted HR 1.2, 95% CI 0.9-1.5). Results of a subanalysis of confirmed cases were similar (adjusted HR 1.4, 95% CI 0.9-2.2). As expected, there was a positive association with family history of CD (adjusted HR 16.4, 95% CI 8.631.1). There was no association between use of systemic adrenergic drugs and CD (adjusted HR 1.0, 95% CI 0.5-1.8).Discussion: We found a non-significant trend for the association between early-life antibiotic exposure and risk of CD. No association was observed in our negative control group. Our findings are consistent with previously observed associations between antibiotic use and CD.

269 Sacral Nerve Neuromodulation Therapy; a Promising New Treatment for Children with Refractory Functional Constipation

Background: Sacral nerve neuromodulation (SNM) therapy has been successfully applied in adult patients with urinary and fecal incontinence. Recently, SNM therapy showed good results in adults with refractory functional constipation. No data, however, are available of SNM therapy as a treatment option in children with refractory functional constipation. Aim: To describe the preliminary results of SNM therapy in children with chronic functional constipation refractory to intensive conservative treatment. Material and methods: Children with functional constipation according to the ROME III criteria not responding to intensive conservative treatment (laxatives, enemas and colonic lavage) were assigned for SNM therapy. When improvement of symptoms was seen during the percutaneous nerve evaluation (PNE) with a permanent electrode in the 3 weeks testing phase, a permanent sacral nerve neuromodulator was implanted. Patients were followed up for at least six months after implantation by interviews and bowel diaries. Improvement was defined as spontaneous defecation ≥ 3 times a week. Results: 5 patients (all females) 14 - 18 years old with functional constipation were included. None of them had spontaneous defecation at presentation. Duration of symptoms varied from 6 - 18 years and the duration of treatment from 1 -17 years. In all patients the testing phase was successful. After implantation of the permanent neuromodulator, all children had spontaneous defecation ≥ 3 times a week without medication. Conclusion: SNM is a promising new treatment option in children with refractory functional constipation not responding to intensive conservative therapy. However, larger prospective studies with long-term follow up are required.