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BACKGROUND: For children with constipation and fecal incontinence treated with antegrade continence enemas (ACE), a fluoroscopic study with contrast administered via appendicostomy/cecostomy can define the anatomy of the colon and simulate the flush to investigate associated symptoms or inadequate response. These studies can at times show retrograde flow into the small intestine. Our objective was to investigate the significance of this finding. METHODS: We reviewed studies at our institution with contrast administered via appendicostomy/cecostomy in children treated with ACE, identifying those demonstrating retrograde flow of contrast. We recorded demographics, medical history, interventions, and outcomes. RESULTS: We identified 162 studies (52% male, median age 10.7 years) with contrast via appendicostomy (76%) or cecostomy (24%). Diagnoses included anorectal malformation (38%), spinal cord anomaly (26%), functional constipation (24%), colonic dysmotility (18%), and Hirschsprung disease (12%). Fifty-nine (36%) studies showed retrograde flow: 28/59 children (48%) were not responding adequately and 21/59 (36%) had symptoms with ACE. Children with retrograde flow were more likely to have symptoms with ACE than those without (36% vs. 15%, p < 0.01). Fourteen children underwent interventions for this finding, including administering flushes more distally (4/8 responded), changing positioning of the child during flush administration, (1/2 responded), and slowing administration (1/1 responded). Retrograde flow was associated with younger age (p < 0.01), not sex or underlying diagnosis. CONCLUSION: Identifying retrograde flow during studies with contrast administered via appendicostomy/cecostomy can be useful for children with a poor response or symptoms associated with ACE, as adjustments to the mechanics of the flush can alleviate those symptoms. LEVEL OF EVIDENCE: Prognostic study, Level III.
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OBJECTIVES: Rumination syndrome (RS) beginning in early childhood or infancy is understudied and challenging to treat. Our objective is to compare the characteristics and outcomes of early-onset (EO) and adolescent-onset (AO) patients with RS. METHODS: We conducted an ambidirectional cohort study of children diagnosed with RS at our institution. Patients were included in two groups: EO (RS symptom onset ≤5 years and diagnosis ≤12 years) and AO (onset >12 years). Patient characteristics, severity, and outcomes were compared between the groups. RESULTS: We included 49 EO and 52 AO RS patients. The median ages of symptom onset and diagnosis in EO were 3.5 and 6 years, respectively; AO, 14.5 and 15 years. EO RS had a slight male predominance while AO was predominantly female (p = 0.016). EO patients were more likely to have developmental delay (24% vs. 8%, p = 0.029) and less likely to have depression (0% vs. 23%, p < 0.001) or anxiety (14% vs. 40%, p = 0.004). At baseline, EO RS was less severe than AO RS: EO RS had greater regurgitation frequency (p < 0.001) but lower vomiting frequency (p = 0.001), resulting in less meal skipping (p < 0.001), reliance on tube feeding or parenteral nutrition (p < 0.001), and weight loss (p = 0.035). EO RS symptoms improved over time: at follow-up, patients had lower regurgitation (p < 0.001) and vomiting frequency (p < 0.001) compared to baseline. CONCLUSION: EO RS is clinically distinct from AO RS, with differences in sex distribution, comorbid conditions, and severity of initial presentation. The pathogenesis and natural history of EO RS may be distinct from that of AO RS.
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Síndrome da Ruminação , Criança , Humanos , Masculino , Pré-Escolar , Feminino , Adolescente , Estudos de Coortes , Idade de Início , Redução de Peso , Vômito/etiologiaRESUMO
OBJECTIVES: Rumination syndrome (RS) is challenging to diagnose, which can lead to diagnostic delays. Our objective was to evaluate the length of time from RS symptom onset to diagnosis in patients referred to our institution and to examine whether this duration predicts treatment outcomes. METHODS: We conducted a review of patients with RS evaluated at our institution. Data were collected from chart review and patient/family reported questionnaires. We evaluated the time from symptom onset to diagnosis over time and whether it was associated with symptom resolution. RESULTS: We included 247 patients with RS (60% female, median age of 14 years, interquartile range [IQR]: 9-16 years). The median age at symptom onset was 11 years (IQR: 5-14 years) and median age at diagnosis was 13 years (IQR: 9-15 years) for a median duration of 1 year (IQR: 0-3 years) between symptom onset and diagnosis. Length of time between symptom onset and diagnosis did not change significantly at our institution from 2016 to 2022. Among the 164 children with outcome data, 47 (29%) met criteria for symptom resolution after treatment. A longer time to diagnosis was associated with a lower likelihood of symptom resolution after treatment (p = 0.01). CONCLUSION: In our experience, the time to RS diagnosis after symptom onset is shorter than previously described. A longer delay in diagnosis is associated with lower likelihood of symptom resolution after treatment, emphasizing the importance of a prompt recognition of rumination symptoms and a timely diagnosis.
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OBJECTIVES: For children with intractable functional constipation (FC), there are no evidence-based guidelines for subsequent evaluation and treatment. Our objective was to assess the practice patterns of a large, international cohort of pediatric gastroenterologists. METHODS: We administered a survey to physicians who attended the 2nd World Congress of Pediatric Neurogastroenterology and Motility held in Columbus, Ohio (USA) in September 2023. The survey included 29 questions on diagnostic testing, nonpharmacological and pharmacological treatment, and surgical options for children with intractable FC. RESULTS: Ninety physicians from 18 countries completed the survey. For children with intractable FC, anorectal manometry was the most commonly used diagnostic test. North American responders were more likely than Europeans to use stimulant laxatives (97% vs. 77%, p = 0.032), prosecretory medications (69% vs. 8%, p < 0.001), and antegrade continence enemas (ACE; 83% vs. 46%, p = 0.009) for management. Europeans were more likely than North Americans to require colonic transit testing before surgery (85% vs. 30%, p < 0.001). We found major differences in management practices between Americans and the rest of the world, including use of prosecretory drugs (73% vs. 7%, p < 0.001), anal botulinum toxin injections (81% vs. 58%, p = 0.018), ACE (81% vs. 58% p = 0.018), diverting ileostomies (56% vs. 26%, p = 0.006), and colonic resections (42% vs. 16%, p = 0.012). No differences were found when respondents were compared by years of experience. CONCLUSIONS: Practice patterns in the evaluation and treatment of children with intractable FC differ widely among pediatric gastroenterologists from around the world. A clinical guideline regarding diagnostic testing and surgical decision-making is needed.
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Constipação Intestinal , Padrões de Prática Médica , Humanos , Constipação Intestinal/terapia , Constipação Intestinal/diagnóstico , Padrões de Prática Médica/estatística & dados numéricos , Criança , Laxantes/uso terapêutico , Inquéritos e Questionários , Manometria/estatística & dados numéricos , Feminino , Masculino , Gastroenterologia/estatística & dados numéricosRESUMO
OBJECTIVES: We have had success treating children with severe rumination syndrome using a multidisciplinary intensive outpatient program (IOP) involving multiple treatment sessions daily. During the coronavirus disease 2019 (COVID-19) pandemic, we temporarily transitioned care to telemedicine. The objective of this study is to compare outcomes of patients with rumination syndrome who completed IOP treatment in person versus by telemedicine. METHODS: We performed a retrospective review of patients diagnosed with rumination syndrome who participated in IOP treatment from 2018 to 2020. Similar treatment sessions were performed involving medical and behavioral techniques provided by a multidisciplinary team during telemedicine visits. Families/patients were asked to complete a survey outlining their child's current rumination symptom severity and review the IOP. RESULTS: We included 34 patients (79% F, median age 15 years, range 7-19 years) who completed IOP treatment. Twenty-six patients (76%) were treated in person and 8 patients (24%) by telemedicine. For patients treated in person, 76% (19/25) had improvement in symptoms while 16% (4/25) had complete resolution of symptoms. For patients treated by telemedicine, 88% (7/8) had improvement in their symptoms. There were no significant differences between groups in likelihood of improvement. Overall, 78% (18/23) preferred in person therapy while 17% (4/23) did not have a preference. All 18 of the in-person cohort who completed follow-up surveys preferred in-person management. CONCLUSIONS: Multidisciplinary intensive outpatient treatment for children and adolescents with severe rumination syndrome is effective. Although telemedicine may be an alternative to in person therapy, majority of families prefer in person visits.
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COVID-19 , Síndrome da Ruminação , Telemedicina , Adolescente , Criança , Humanos , Adulto Jovem , Adulto , COVID-19/terapia , Pacientes Ambulatoriais , Assistência Ambulatorial , Telemedicina/métodosRESUMO
OBJECTIVES: Rumination syndrome (RS) can be challenging to treat and data on treatment outcomes in children are limited. The objective of this study was to evaluate outcomes of children with RS treated with tailored outpatient and inpatient strategies. METHODS: We performed a retrospective cohort study of children <18 years old with RS evaluated at our institution from 2018 to 2020. At our institution, we use a multidisciplinary, tiered approach to treatment based on presentation severity. Children with RS either undergo outpatient treatment program (OP) or participate in an intensive outpatient program (IOP) or an intensive inpatient program (IP). We reviewed baseline characteristics and assessed severity (including frequency of regurgitation/vomiting, route of nutrition, and weight loss) at baseline, at completion of treatment, and at a follow-up time point. RESULTS: We included 171 children with RS (64% female, median age 13 years, interquartile range (IQR) 10-15), 123 of whom had post-treatment data after completing OP, IOP, or IP. At baseline, 66% of patients were vomiting daily and 40% were losing weight. After treatment, 72% of OP, 95% of IOP, and 96% of IP patients reported that symptoms were better or fully resolved compared to baseline. In all 3 treatment groups, patients were vomiting, losing weight, and skipping meals significantly less after treatment compared to baseline. At follow-up (median 5.3 months), 86% of IOP and 66% of IP patients had symptoms that remained better or resolved. CONCLUSIONS: RS can cause severe symptoms, impacting nutritional status and school participation. However, multidisciplinary care in a tiered approach leads to significant symptomatic improvement.
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Síndrome da Ruminação , Adolescente , Criança , Feminino , Humanos , Masculino , Assistência Ambulatorial , Estudos Retrospectivos , Resultado do Tratamento , Vômito/etiologia , Vômito/terapiaRESUMO
OBJECTIVES: Our objective was to investigate if there is a difference in the detection of the rectoanal inhibitory reflex (RAIR) when an anorectal manometry (ARM) is performed awake or under general anesthesia. METHODS: A retrospective review of ARM studies was performed to identify children who had undergone ARMs both while awake and under general anesthesia. We compared ARM outcomes including the detection of the RAIR and anal canal resting pressure. RESULTS: Thirty-four children had received ARMs both while awake and under general anesthesia (53% female, median age at first ARM 7.5 years [range 3-18 years]). In 9 of 34 (26%) children the RAIR was solely identified during ARM under general anesthesia and not during ARM while awake. In 6 of 9 (66%) this was unrelated to the balloon volumes used during balloon inflations. In 4 of 34 (12%) children, assessment of the RAIR was inconclusive during ARM under general anesthesia due to too low, or loss of anal canal pressure. In 2 of those children, ARMs while awake showed presence of a RAIR. Anal canal resting pressures were higher during ARM while awake versus ARM under general anesthesia (median 70 [interquartile range, IQR 59-85] vs 46 mmHg [IQR 36-65] respectively, P < 0.001). CONCLUSIONS: General anesthesia may affect the detection of a RAIR in 2 ways. On the one hand, it may facilitate better visualization in children in whom a RAIR could not be visualized while awake. On the other hand, it may cause a loss of anal canal pressure resulting in an inconclusive test result.
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Reto , Vigília , Humanos , Criança , Feminino , Pré-Escolar , Adolescente , Masculino , Manometria/métodos , Canal Anal , Reflexo , Anestesia GeralRESUMO
OBJECTIVES: To evaluate whether providing a constipation action plan (CAP) to families of children with constipation at outpatient gastroenterology (GI) visits affects health care utilization of the GI department. METHODS: We created a CAP to be included in the after visit summary of children seen in our GI Clinic. We compared the number of patient telephone calls, electronic messages, and urgent care (UC) visits, emergency department (ED) visits, and hospitalizations within 3 months after the visit of patients who received the CAP compared to those who did not using inverse probability treatment weighting (IPTW) analysis. For families who received the CAP at a follow-up visit, we compared these variables in the 3 months before and after the CAP was provided using paired t test and McNemar's test as appropriate. RESULTS: We included 336 patients who received the CAP and 2812 who did not. After IPTW adjustment, there were fewer patient telephone calls for patients who received the CAP (P = 0.0006). The difference in patient electronic messages was not statistically significant (P = 0.09). For the 45 patients who received the CAP at a follow-up visit, there were on average 1.8 more patient telephone calls made prior to receiving the CAP than after (95% confidence interval (CI) = 0.8-2.8; P = 0.0007) and 2.3 more patient electronic messages received (95% CI = 0.1-4.5; P = 0.04). There were no differences in UC/emergency department visits or hospitalizations. CONCLUSIONS: We found that providing a CAP to families of children with constipation decreases health care utilization. Further studies are needed to determine whether this impacts patient outcomes.
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Serviço Hospitalar de Emergência , Aceitação pelo Paciente de Cuidados de Saúde , Criança , Humanos , Assistência Ambulatorial , Hospitalização , Constipação Intestinal/terapiaRESUMO
OBJECTIVES: To identify predictors of hospitalization in pediatric patients presenting to an emergency department (ED) for a cyclic vomiting syndrome (CVS) attack. STUDY DESIGN: We retrospectively reviewed patients with CVS seen at our institution between 2015 and 2018 and included those who met the Rome IV criteria for CVS. We identified all CVS-related ED visits and subsequently performed a case-control analysis, utilizing multivariate logistic regression, to identify clinical and demographic factors that may predict hospitalization. RESULTS: In total, 219 patients with CVS (using International Statistical Classification of Diseases and Related Health Problems, 10th Revision) were identified, of which 65% met the inclusion criteria (median age 11 years). We identified 152 CVS-related ED visits, of which 62% resulted in hospitalization. Factors found to predict hospitalization using multivariate analyses included male sex (P = .04), younger age (P = .027), delayed presentation (>24 hours) to the ED (P < .001), and longer wait time prior treatment with antiemetics (P = .029). CONCLUSION: One-quarter of all patients with CVS had presented to the ED and nearly two-thirds of these ED visits resulted in hospitalization. A delayed presentation to the ED following the onset of symptoms was the strongest independent predictor of hospital admission, alongside male sex, younger age, and longer ED wait times before treatment with antiemetics. These findings suggest that early intervention may be key to successfully mitigating the risk of hospitalization for a CVS attack.
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Regras de Decisão Clínica , Hospitalização/estatística & dados numéricos , Índice de Gravidade de Doença , Vômito/diagnóstico , Vômito/terapia , Adolescente , Estudos de Casos e Controles , Criança , Pré-Escolar , Serviço Hospitalar de Emergência/estatística & dados numéricos , Utilização de Instalações e Serviços/estatística & dados numéricos , Feminino , Humanos , Modelos Logísticos , Masculino , Estudos Retrospectivos , Tempo para o Tratamento/estatística & dados numéricos , Adulto JovemRESUMO
A few studies have shown that esophageal air events (EAEs), such as air-swallows, may be associated with symptoms that have historically been associated with gastroesophageal reflux disease (GERD). To objectively test a hypothesis that all EAE types (air-swallows, supragastric belches and gastric belches) can be associated with GERD-like symptoms, we removed the impedance "tags" from the GER episodes (placed during autoscan) and instead tagged either air-swallows, supragastric belches or gastric belches in each of 3 copies of the 24-hour impedance tracing for 2 infant patients who presented with symptoms suggestive of GER as an etiology. Impedance system software (MMS) analyses revealed that, in both infants, all EAE types were significantly associated (SAP >95%) with 1 or more of the GERD-like symptom types (cough, pain/crying, back-arching, and gagging). These data underscore the importance of considering other diagnoses when developing management strategies for treating GERD-like symptoms in infants.
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Aerofagia/diagnóstico , Impedância Elétrica , Eructação/diagnóstico , Refluxo Gastroesofágico/diagnóstico , Diagnóstico Diferencial , Monitoramento do pH Esofágico , Esôfago/fisiopatologia , Feminino , Humanos , Lactente , MasculinoRESUMO
OBJECTIVES: Motility and functional disorders are common in children and often debilitating, yet these disorders remain challenging to treat effectively. At the 2018 Annual North American Society for Pediatric Gastroenterology, Hepatology and Nutrition meeting, the Neurogastroenterology and Motility Committee held a full day symposium entitled, 2018 Advances In Motility and In NeuroGastroenterology - AIMING for the future. The symposium aimed to explore clinical paradigms in pediatric gastrointestinal motility disorders and provided a foundation for advancing new scientific and therapeutic research strategies. METHODS: The symposium brought together leading experts throughout North America to review the state of the art in the diagnosis and management of motility and functional disorders in children. Presentations were divided into esophageal, antral duodenal, and colorectal modules. Each module included oral presentations by experts in the respective fields, leading to thought-provoking discussions. There were 2 breakout sessions with small group discussions on select topics, focusing on defining scientific insights into the diagnosis and management of pediatric functional gastrointestinal and motility disorders in a systematic, segment-based approach. CONCLUSIONS: The field of neurogastroenterology has made remarkable progress in the last decade. The current report summarizes the major learning points from the symposium highlighting the diagnosis and promising therapies on the horizon for pediatric neurogastrointestinal and motility disorders.
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Gastroenterologia , Gastroenteropatias , Criança , Esôfago , Gastroenteropatias/diagnóstico , Gastroenteropatias/terapia , Motilidade Gastrointestinal , Humanos , América do NorteRESUMO
OBJECTIVES: Among adolescents with inflammatory bowel disease (IBD), nonadherence rates are 50 to 88% across medications. Improving education in adults with IBD has been shown to improve coping and adherence to treatment in adults with IBD. Therapeutic patient education (TPE) has been used in patients with chronic diseases to train patients in skills to support treatment adaptation and condition management. This study tested the feasibility and preliminary efficacy of a novel TPE intervention in adolescents with IBD. METHODS: In this pilot, mixed-methods study, we evaluated the feasibility and preliminary efficacy of TPE with the IBD Pocket Guide on medication adherence, IBD knowledge, and transition readiness in adolescents ages 11 to 18 years. Medication adherence was monitored using a MedMinder Pill Dispensing system. Participants who were <90% adherent during a 4-week pre-intervention monitoring period were randomized to either a usual care group or an educational intervention (EI) group. Participants were followed for an additional 4 weeks after intervention. RESULTS: Trends were found in the EI group indicating improved medication adherence and IBD knowledge compared with the usual care group, though differences between groups did not reach statistical significance. Qualitative data showed that participants perceived that they had improved knowledge after the educational intervention. CONCLUSIONS: Therapeutic patient education may be beneficial for improving patient medication adherence and IBD knowledge. Future directions include testing the effects of the intervention with a larger sample.
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Adaptação Psicológica , Doenças Inflamatórias Intestinais/psicologia , Adesão à Medicação , Educação de Pacientes como Assunto , Adolescente , Serviços de Saúde do Adolescente , Criança , Estudos de Viabilidade , Feminino , Humanos , Masculino , Ohio , Projetos Piloto , Inquéritos e QuestionáriosRESUMO
BACKGROUND: Our objective was to evaluate long-term outcomes of sacral nerve stimulation (SNS) for children with functional and organic defecation disorders. METHODS: We performed a prospective study of children <21 years of age who started SNS treatment between 2012 and 2018. We recorded demographics, medical history, and diagnostic testing. We obtained measures of symptom severity and quality of life at baseline and follow up at 1, 6, 12, 24, 36, 48, and ≥60 months. Successful response was defined as bowel movements >2 times/week and fecal incontinence (FI) <1 time/week. Families were contacted to administer the Glasgow Children's Benefit Inventory and to evaluate patient satisfaction. KEY RESULTS: We included 65 patients (59% female, median age at SNS 14 years, range 9-21) with median follow-up of 32 months. Thirty patients had functional constipation (FC), 15 had non-retentive FI (NRFI), and 16 had an anorectal malformation (ARM). The percentage with FI <1 time/week improved from 30% at baseline to 64% at 1 year (p < 0.001) and 77% at most recent follow-up (p < 0.001). Patients with FC, NRFI, and ARM had sustained improvement in FI (p = 0.02, p < 0.001, p = 0.02). Patients also reported fewer hard stools (p = 0.001). Bowel movement frequency did not improve after SNS. At most recent follow-up, 77% of patients with a functional disorder and 50% with an organic disorder had responded (p = 0.03). Nearly all families reported benefit. CONCLUSIONS AND INFERENCES: SNS led to sustained improvement in FI regardless of underlying etiology, but children with functional disorders were more likely to respond than those with organic disorders.
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Constipação Intestinal , Terapia por Estimulação Elétrica , Incontinência Fecal , Qualidade de Vida , Humanos , Incontinência Fecal/terapia , Incontinência Fecal/fisiopatologia , Feminino , Criança , Adolescente , Masculino , Terapia por Estimulação Elétrica/métodos , Constipação Intestinal/terapia , Constipação Intestinal/fisiopatologia , Estudos Prospectivos , Adulto Jovem , Resultado do Tratamento , Plexo Lombossacral , Defecação/fisiologia , SeguimentosRESUMO
The objective was to estimate the growth curves and genetic parameters using random regression methodology for Nellore cattle raised in Pantanal, MS, Brazil (6974 calves; n = 53,233 weights), with at least four weighings per individual. The model considered direct and maternal genetic additives and maternal permanent environmental effects at random. Orthogonal Legendre polynomials of cubic order were used to fit the growth curve. Analyses of variance were performed using the GLM procedure. The model used contained the fixed effects of sex, year of birth, farm, and the covariates calf birth month (linear and quadratic) and cow age at calving (linear and quadratic). The adjusted mean weight at 120 days of age was 93.43 ± 19.78 kg, and for 205 days of age, it was 180.42 ± 26.58 kg. Animals born in the dry season had a higher average weight [kg] (219.57 vs. 211.78, 3.7% higher) and, consequently, had higher weights at 646 days of age. Estimates of direct heritabilities (h2a) ranged from 0.35 to 0.75 (high magnitudes), and maternal heritabilities (h2m) along the trajectory of low magnitudes ranged from 0.03 to 0.08, respectively. The use of random regression to evaluate beef animals allows for adjusting the growth curve and selecting the best animals to be the parents of future generations.
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BACKGROUND: Baclofen can decrease rumination frequency in adults with rumination syndrome. Outcomes of baclofen treatment in children with rumination syndrome have not been described. The aim of this study was to examine the safety and efficacy of baclofen in children with rumination syndrome in combination with behavioral therapy at a single center. METHODS: We performed a retrospective review of children aged 0-18 years with rumination syndrome based on Rome criteria and prescribed baclofen by a pediatric gastroenterologist at the Nationwide Children's Hospital, Columbus, Ohio, USA, between 2012 and 2019. Children without follow-up data or who were prescribed baclofen for other symptoms were excluded. RESULTS: We identified 44 children with rumination syndrome who were prescribed baclofen by a pediatric gastroenterologist. Seventeen either did not have follow-up data or never started the medication. We included 27 patients in the study: 22 (81.5%) female, median age 14.5 years (range 10-18 years) and 100% Caucasian. Twenty patients (74%) received baclofen 5 mg and seven patients (26%) received baclofen 10 mg three times daily. Most patients received behavioral therapy and baclofen simultaneously. Thirteen patients (48%) reported improvement in symptoms, primarily a decrease in rumination frequency, at their first follow-up visit. Regurgitation frequency per week decreased after starting baclofen (p < 0.05). One patient experienced dizziness. No other side effects were reported. CONCLUSION: Nearly half of our patients with rumination syndrome improved after baclofen. It was well tolerated with minimal side effects. This suggests that baclofen in addition to behavioral therapy can improve symptoms of rumination syndrome. Prospective, controlled studies in a larger cohort of children with rumination syndrome are needed to confirm these findings.
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Baclofeno , Síndrome da Ruminação , Adulto , Humanos , Criança , Feminino , Adolescente , Masculino , Baclofeno/efeitos adversos , Estudos Retrospectivos , Síndrome da Ruminação/terapia , Estudos ProspectivosRESUMO
INTRODUCTION: Anal sphincter botulinum toxin injections (BTIs) are used in the treatment of children with severe defecation disorders, including Hirschsprung disease (HD) and functional constipation (FC). Our objective was to evaluate the outcomes of BTI in these children. MATERIALS AND METHODS: We performed a prospective cohort study of children undergoing BTI from July 2018 to December 2018. We recorded perceived effect of the BTI, including effectiveness ranging from 0 (not at all effective) to 4 (extremely effective). In addition, we recorded symptoms and the Cleveland Clinic Constipation Score (CCCS). Data were collected at baseline and at 2 weeks, 2 months, and 4 months post-injection. RESULTS: Forty-two children (HD = 25, FC = 17) were included in the study (median age 4.3 years, IQR 2.4-7.2, 52% male). Twenty-two (88%) children with HD and eight (47%) children with FC had previously undergone a BTI. BTIs were perceived effective in 16 (76%) and 12 (71%) children with HD and eight (47%) and seven (47%) children with FC at 2-week and 2-month follow-up, respectively. Effectiveness was not rated differently between groups except at the 2-month follow-up, when patients with HD rated the BTI more effective compared to those with FC (median 2 [HD] vs. median 1 [FC], p = 0.022). Over the course of the study, 17/39 (44%) children reported self-limiting adverse effects such as fecal incontinence and pain at the injection site. CONCLUSION: Anal sphincter BTIs can be effective in the treatment of constipation in both HD and FC patients.
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Toxinas Botulínicas , Doença de Hirschsprung , Criança , Humanos , Masculino , Pré-Escolar , Feminino , Canal Anal , Doença de Hirschsprung/complicações , Doença de Hirschsprung/tratamento farmacológico , Estudos Prospectivos , Resultado do Tratamento , Constipação Intestinal/tratamento farmacológico , Constipação Intestinal/etiologiaRESUMO
Anal sphincter defects can lead to fecal incontinence. The relationship between the extent of defect and continence is controversial. Magnetic resonance imaging (MRI) of the pelvis can assess anal sphincter defects. Transrectal ultrasonography (TRUS) is used to assess sphincter integrity in adults. We present a 17-year-old male with history of sexual abuse, rectal prolapse, and fecal incontinence. MRI showed a small defect that did not explain his clinical presentation. TRUS identified more extensive defects which were not picked up by MRI. The patient had rectopexy, and his rectal prolapse and fecal incontinence resolved. TRUS was superior in identifying sphincter defects compared with MRI. Our case also highlights that continence is possible despite large sphincter defects in pediatric patients. This may reflect the compensatory mechanism of residual sphincter in the absence of the aggravating factors like rectal prolapse.
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IMPORTANCE: Although it is widely believed that concealment or deception is required to elicit a placebo response, recent studies with adults suggest that open-label placebo (OLP) (ie, honestly prescribed placebos) can yield significant benefits. No studies of OLP have been performed with children. OBJECTIVE: To evaluate the efficacy of OLP for the treatment of children and adolescents with functional abdominal pain or irritable bowel syndrome. DESIGN, SETTING, AND PARTICIPANTS: This multicenter crossover randomized clinical trial was conducted from July 1, 2015, to June 15, 2018, at 3 US centers among children and adolescents aged 8 to 18 years with functional abdominal pain or irritable bowel syndrome defined per Rome III criteria. Statistical analysis was performed from March 1, 2019, to September 30, 2020, on an intention-to-treat basis. INTERVENTIONS: Patients completed 1 week of observation prior to randomization to 1 of 2 counterbalanced groups: OLP for 3 weeks followed by a 3-week control period or control period for 3 weeks followed by OLP for 3 weeks. During the OLP period, participants took 1.5 mL of an inert liquid placebo twice a day. A standardized method for explaining the OLP was used, and the interaction with clinicians had the same duration and style for both time periods. Hyoscyamine was allowed as a rescue medication. MAIN OUTCOMES AND MEASURES: The primary outcome was the mean daily pain score during each of the interventions, measured on a 0- to 100-mm visual analog scale, where higher scores indicated greater pain. The number of rescue medications taken during each intervention served as an objective secondary measure. RESULTS: Thirty patients (mean [SD] age, 14.1 [3.4] years; 24 female participants [80.0%]; 16 [53.3%] with functional abdominal pain and 14 [46.7%] with irritable bowel syndrome) completed the study. The mean (SD) pain scores were significantly lower during OLP treatment compared with the control period (39.9 [18.9] vs 45.0 [14.7]; difference, 5.2; 95% CI, 0.2-10.1; P = .03). Patients took nearly twice as many hyoscyamine pills during the control period compared with during the OLP period (mean [SD] number, 3.8 [5.1] pills vs 2.0 [3.0] pills; difference, 1.8 pills; 95% CI, 0.5-3.1 pills). CONCLUSIONS AND RELEVANCE: During OLP, patients with functional abdominal pain or irritable bowel syndrome reported significantly less pain and took significantly fewer pain medications. Open-label placebo may be an effective treatment for children and adolescents with functional abdominal pain or irritable bowel syndrome. TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT02389998.
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Síndrome do Intestino Irritável , Dor Abdominal/tratamento farmacológico , Adolescente , Adulto , Criança , Método Duplo-Cego , Feminino , Humanos , Síndrome do Intestino Irritável/tratamento farmacológico , Medição da Dor , Projetos de Pesquisa , Resultado do TratamentoRESUMO
BACKGROUND: Recently, a new high-definition (or three-dimensional "3D") high-resolution anorectal manometry (3D-ARM) catheter has been introduced. This catheter allows for a more detailed visualization of the anal canal. However, its clinical utility and tolerability in children with constipation are unknown. Our primary objective was to evaluate the agreement between findings from solid-state high-resolution anorectal manometry (HR-ARM) and 3D-ARM. Secondary objectives were to investigate if 3D-ARM has additional value over HR-ARM and to evaluate patient and provider experience. METHODS: Prospective pilot study including children (8-18 years of age) with functional constipation scheduled for anorectal manometry. Children underwent HR-ARM and 3D-ARM consecutively. We compared manometry results of both procedures and collected data on patient and provider experience via self-developed questionnaires. KEY RESULTS: Data of ten patients were analyzed (60% female, median age 14.9 years). In the majority of patients, ARMs were performed awake (n = 8, 80%). In two patients, the recto-anal inhibitory reflex (RAIR) was visualized during HR-ARM but not during 3D-ARM. Anal canal resting pressures were significantly higher during 3D-ARM compared to HR-ARM (median 77 mmHg [IQR 59-94] vs. 69 mmHg [IQR 51-91], respectively, p = 0.037). No significant anatomical or muscular abnormalities were visualized during the 3D-ARM. The majority of children identified the 3D-ARM as the more unpleasant (5/7 [71%]) and more painful procedure (6/7 [86%]) and therefore preferred the HR-ARM (4/7 [57%]). CONCLUSIONS & INFERENCES: In our patient sample, 3D-ARM was associated with more discomfort without providing more useful information and even resulted in an inconsistent visualization of the RAIR.