Publications by authors named "Angelika Kindermann"

50 Publications

Health-related quality of life and distress of parents of children with avoidant restrictive food intake disorder.

J Pediatr Gastroenterol Nutr 2021 Apr 14. Epub 2021 Apr 14.

Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Department of Pediatric Gastroenterology, Hepatology and Nutrition, Amsterdam, The Netherlands Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Child and Adolescent Psychiatry & Psychosocial Care, Amsterdam Reproduction and Development, Amsterdam Public Health, Amsterdam, The Netherlands Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Department of Dietetics, Amsterdam, The Netherlands Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Department of Rehabilitation, Amsterdam, The Netherlands.

Objectives: Health-related quality of life (HRQOL) of children with avoidant restrictive food intake disorder (ARFID) is impaired.Aim To measure HRQOL and distress of parents of children with ARFID.

Methods: Cross-sectional cohort study. Parents of children with ARFID, visiting our multidisciplinary feeding team, completed questionnaires on the online KLIK portal; the TNO-AZL QOL to assess parental HRQOL and the Distress Thermometer for Parents. Reference groups of parents of healthy (HC) and chronically ill children (CIC) were used.

Results: 85 mothers and 62 fathers of 89 children with ARFID (58% female, median age 1.9y) were included (response rate 68%). No differences were found regarding HRQOL in 11/12 domains between parents of children with ARFID and HC. Mothers of children with ARFID reported significantly higher HRQOL regarding pain and fathers a significantly lower HRQOL on depressive emotions compared to HC. No differences were found in overall and clinical distress scores between parents of children with ARFID and HC/CIC. Mothers of children with ARFID had significantly higher distress scores regarding cognitive problems compared to HC and parenting problems in children <2y compared to HC/CIC. Significantly higher distress scores on parenting problems in children <2y were found in fathers of children with ARFID compared to HC/CIC.

Conclusion: Most HRQOL and distress scores of parents of children with ARFID were comparable to reference groups. Since parents of children with ARFID perceive a lack of understanding and support from the environment, professionals should suggest peer support through patient's organizations. Furthermore, it is important to offer professional support since parents indicated that they would like to talk to a professional about their situation.
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http://dx.doi.org/10.1097/MPG.0000000000003150DOI Listing
April 2021

Exclusive enteral nutrition mediates gut microbial and metabolic changes that are associated with remission in children with Crohn's disease.

Sci Rep 2020 11 3;10(1):18879. Epub 2020 Nov 3.

Tytgat Institute for Liver and Intestinal Research, Amsterdam UMC, Location AMC, Meibergdreef 69, 1105BK, Amsterdam, The Netherlands.

A nutritional intervention, exclusive enteral nutrition (EEN) can induce remission in patients with pediatric Crohn's disease (CD). We characterized changes in the fecal microbiota and metabolome to identify the mechanism of EEN. Feces of 43 children were collected prior, during and after EEN. Microbiota and metabolites were analyzed by 16S rRNA gene amplicon sequencing and NMR. Selected metabolites were evaluated in relevant model systems. Microbiota and metabolome of patients with CD and controls were different at all time points. Amino acids, primary bile salts, trimethylamine and cadaverine were elevated in patients with CD. Microbiota and metabolome differed between responders and non-responders prior to EEN. EEN decreased microbiota diversity and reduced amino acids, trimethylamine and cadaverine towards control levels. Patients with CD had reduced microbial metabolism of bile acids that partially normalized during EEN. Trimethylamine and cadaverine inhibited intestinal cell growth. TMA and cadaverine inhibited LPS-stimulated TNF-alpha and IL-6 secretion by primary human monocytes. A diet rich in free amino acids worsened inflammation in the DSS model of intestinal inflammation. Trimethylamine, cadaverine, bile salts and amino acids could play a role in the mechanism by which EEN induces remission. Prior to EEN, microbiota and metabolome are different between responders and non-responders.
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http://dx.doi.org/10.1038/s41598-020-75306-zDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7609694PMC
November 2020

Can 2 Different Fecal Calprotectin Assays be Used Interchangeably in IBD Treatment?

J Clin Gastroenterol 2020 Oct 27. Epub 2020 Oct 27.

Departments of Pediatric Gastroenterology, Emma Children's Hospital.

Background: Fecal calprotectin (FC) is a biomarker for inflammation in inflammatory bowel disease (IBD). Interpretation of results can be complicated because of the use of different assays to determine FC.

Goals: To assess the agreement between 2 different assays for determining FC in patients with IBD.

Methods: Samples from adults and children with IBD were tested with 2 assays: (1) EliA 2 Calprotectin and (2) EK-Cal. Samples were uniformly tested on the same day. Interassay variability was displayed in a Bland-Altman plot. The difference in categorization of the FC result (1: 0 to 250 mg/kg, 2: 250 to 500 mg/kg, 3: >500 mg/kg) was assessed with the linear weighted κ for adults and children separately.

Results: A total of 171 patients [mean age: 33 (range: 7 to 81); 92 (54%) female; 117 (68%) Crohn's disease; 53 (31%) ulcerative colitis] were included. Median (interquartile ranges) FC levels were 281 mg/kg (70 to 971) (EK-Cal) and 159 mg/kg (31 to 778) (EliA 2), and the mean delta FC was 89 mg/kg. In the adult population, there was substantial agreement between the 2 assays (κ: 0.72; SE: 0.06; 95% confidence interval, 0.60-0.83) and for pediatric patients, the agreement was almost perfect (κ: 0.83; SE: 0.06; 95% confidence interval: 0.70-0.95). Five of 171 patients (all aged ≥17 y and all with colonic disease) had a difference of 2 categories (1 vs. 3) between assays. Interassay variability was the highest in category 3.

Conclusions: The agreement between the EliA 2 and EK-Cal assay in this cohort of IBD patients is substantial to almost perfect. Interassay variability is higher in the highest FC category.
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http://dx.doi.org/10.1097/MCG.0000000000001460DOI Listing
October 2020

Helicobacter pylori Infection in Pediatric Patients Living in Europe: Results of the EuroPedHP Registry 2013 to 2016.

J Pediatr Gastroenterol Nutr 2020 10;71(4):476-483

Pediatric Gastroenterology, Kaplan Medical Centre, Rehovot, Israel.

Objectives: The aim of the study was to assess clinical presentation, endoscopic findings, antibiotic susceptibility and treatment success of Helicobacter pylori (H. pylori) infected pediatric patients.

Methods: Between 2013 and 2016, 23 pediatric hospitals from 17 countries prospectively submitted data on consecutive H. pylori-infected (culture positive) patients to the EuroPedHP-Registry.

Results: Of 1333 patients recruited (55.1% girls, median age 12.6 years), 1168 (87.6%) were therapy naïve (group A) and 165 (12.4%) had failed treatment (group B). Patients resided in North/Western (29.6%), Southern (34.1%) and Eastern Europe (23.0%), or Israel/Turkey (13.4%). Main indications for endoscopy were abdominal pain or dyspepsia (81.2%, 1078/1328). Antral nodularity was reported in 77.8% (1031/1326) of patients, gastric or duodenal ulcers and erosions in 5.1% and 12.8%, respectively. Primary resistance to clarithromycin (CLA) and metronidazole (MET) occurred in 25% and 21%, respectively, and increased after failed therapy. Bacterial strains were fully susceptible in 60.5% of group A, but in only 27.4% of group B. Primary CLA resistance was higher in Southern and Eastern Europe (adjusted odds ratio [ORadj] = 3.44, 95% confidence interval [CI] 2.22-5.32, P < 0.001 and 2.62, 95% CI: 1.63-4.22, P < 0.001, respectively) compared with Northern/Western Europe. Children born outside Europe showed higher primary MET resistance (ORadj = 3.81, 95% CI: 2.25-6.45, P < 0.001). Treatment success in group A reached only 79.8% (568/712) with 7 to 14 days triple therapy tailored to antibiotic susceptibility.

Conclusions: Peptic ulcers are rare in dyspeptic H. pylori-infected children. Primary resistance to CLA and MET is markedly dependent on geographical regions of birth and residence. The ongoing survey will show whether implementation of the updated ESPGHAN/NASPGHAN guidelines will improve the eradication success.
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http://dx.doi.org/10.1097/MPG.0000000000002816DOI Listing
October 2020

Outcomes Following Pouch Formation in Paediatric Ulcerative Colitis: A Study From the Porto Group of ESPGHAN.

J Pediatr Gastroenterol Nutr 2020 09;71(3):346-353

Shaare Zedek Medical Center, The Hebrew University of Jerusalem, Jerusalem, Israel.

Introduction: Contemporary pediatric data on pouch outcomes are sparse, especially in the era of laparoscopic surgeries. We aimed to assess outcomes and predictors in children with ulcerative colitis/inflammatory bowel disease (IBD)-unclassified who underwent colectomy and ileal pouch-anal anastomosis.

Methods: This was a multicenter retrospective cohort study from 17 IBD centers affiliated with the pediatric IBD Porto group of ESPGHAN. An electronic REDcap system was used to collate baseline characteristics, demographic, clinical, management and surgical data, short- and long-term outcomes, and to identify potential predictors of pouch outcome.

Results: Of the 129 patients included, 86 (67%) developed pouchitis during follow-up of median 40 months (interquartile range 26-72), of whom 33 (26%) with chronic pouchitis. Patients operated on by surgeons performing <10 pouch surgeries/year had a higher rate of chronic pouchitis (11/27 [41%] vs 8/54 [15%], P = 0.013) on both univariable and multivariable analyses and also associated with time to pouchitis (P = 0.018) and chronic pouchitis (P = 0.020). At last follow-up, overall pouch performance was rated good/excellent in 86 (74%) patients. Time from colectomy to pouch formation was not associated with pouch outcomes. Despite higher rate of nonsevere surgical complications among children undergoing colectomy at <10 years of age (7/16 [44%] vs 10/92 [11%], P = 0.003), functional outcome and pouchitis rate did not differ.

Conclusions: Pouchitis rate in children with ulcerative colitis/IBD unclassified is high. Surgeon experience is the major modifiable risk factor for pouch outcome. Our analyses suggest that pouch surgery can also be performed successfully in young children.
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http://dx.doi.org/10.1097/MPG.0000000000002805DOI Listing
September 2020

Do Antibiotics Reduce the Incidence of Infections After Percutaneous Endoscopic Gastrostomy Placement in Children?

J Pediatr Gastroenterol Nutr 2020 07;71(1):23-28

Pediatric Gastroenterology.

Objective: Percutaneous endoscopic gastrostomy (PEG) provides a long-term solution for tube dependency. Pediatric guidelines recommend prophylactic antibiotic treatment (ABT) based on adult studies.

Aim: To compare wound infection and other complications in children receiving a PEG with and without prophylactic ABT.

Methods: Retrospective study including children 0 to 18 years undergoing PEG placement. Patients with (2010-2013) and without (2000-2010) ABT were compared with respect to the occurrence of wound infection and other complications.

Results: In total, 297 patients were included (median age 2.9 years, 53% boys). Patients receiving ABT per PEG protocol (n = 78) had a similar wound infection rate (17.9% vs 21%, P = 0.625), significantly less fever (3.8% vs 14.6%, P = 0.013), leakage (0% vs 9.1%, P = 0.003) and shorter hospital admission (2 vs 4 days, P = 0.000), but more overgranulation (28.2% vs 8.7%, P = 0.000) compared with those without (n = 219). Patients receiving any ABT, per PEG protocol or clinical indication (n = 115), had similar occurrence of wound infection (19.1% vs 20.9%, P = 0.768), fever (7.8% vs 14.3%, P = 0.100) and leakage (3.5% vs 8.8%, P = 0.096), a significantly shorter hospital admission (3 vs 4 days, P = 0.000), but more overgranulation (21.7% vs 8.8%, P =0.003) compared with those without (n = 182).

Conclusions: Prophylactic ABT does not seem to reduce the occurrence of wound infection but it might be beneficial with respect to fever, leakage and duration of hospital admission, but not overgranulation. A randomized controlled trial is needed to confirm our results.
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http://dx.doi.org/10.1097/MPG.0000000000002709DOI Listing
July 2020

Long-term efficacy of clinical hunger provocation to wean feeding tube dependent children.

Clin Nutr 2020 09 24;39(9):2863-2871. Epub 2019 Dec 24.

Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Pediatric Gastroenterology, Amsterdam, the Netherlands; Emma Children's Hospital, Amsterdam UMC, Vrije Universiteit Amsterdam, Pediatric Gastroenterology, Amsterdam, the Netherlands.

Background & Aims: The incidence of tube feeding dependency seems to increase worldwide, and these children may remain on prolonged tube feeding for many months to years. The multidisciplinary clinical hunger provocation (CHP) program is an intensive inpatient intervention of usually 2-3 weeks, aimed at weaning children from tube feeding. CHP has been proven highly effective on the short term (80-86%), particularly when applied before the age of two years but long-term data are lacking. The aims of our study were to determine the long-term efficacy of the CHP program and factors associated with success or failure and to assess anthropometrics, feeding behavior, and medical outcomes at long-term follow-up.

Methods: All tube-dependent children who underwent CHP at a tertiary hospital in Amsterdam, the Netherlands, between 2001 and 2014, and had a minimum follow-up of 12-months in 2015, were eligible to participate in this retrospective cohort study. During the CHP program, tube feeding is ceased stepwise to create appetite, according to a strict protocol. The program was defined successful if patients achieved oral intake and could be fully weaned from tube feeding following the CHP program. Acute malnourishment was defined as weight for height <2 SD or loss of >1 SD within 3 months, chronic malnourishment as height for age <2 SD and both acute and chronic malnourishment as both a height for age and weight for height <2 SD. Long-term efficacy (tube free at varying follow-up periods), anthropometrics (height for age, weight for height), feeding behavior and medical outcomes were assessed by a structured cross-sectional parental interview.

Results: In total, 57 patients were admitted to the CHP program. Fifty-two patients could be contacted of whom 42 participated in the study (response rate 81%) with a median age at admittance of 19 (IQR 13-22) months (62% female). The program was initially successful in 36/42 (86% (Bca CI 95% 75.0-95.2)) patients. A younger age upon initiation of tube feeding was negatively correlated with success (p 0.016). At follow-up, a median period of 67.0 (IQR 37.0-101.5) months after discharge, long-term efficacy was 32/41 (78% (Bca CI 95% 64.1-90.0)) (1 missing data). Patients with a successful CHP had beneficial outcomes compared to those with an unsuccessful CHP, showing less selective eating behavior (p 0.025), nocturnal feeding (p 0.044), forced feeding (p 0.044) and hospital admissions (p 0.028). However, 44% of successfully weaned patients fulfilled the criteria for malnourishment at long-term follow-up (13% acute, 22% chronic, and 9% both acute and chronic (compared to 22% at admittance: 13% acute, 6% chronic, and 3% both)). 59.4% of successfully weaned patients showed signs of developmental delays or were diagnosed with new medical diagnoses (43.8%) at long-term follow-up.

Conclusions: The multidisciplinary CHP is a highly effective short-term (86%) and long-term (78%) intervention to wean young children from tube feeding, with beneficial feeding outcomes. However, at long-term follow-up, many successfully weaned patients were malnourished, showed signs of developmental delay, and were diagnosed with new medical diagnoses. For these reasons, patients should be monitored carefully during and after tube weaning, also after successful CHP. Tube dependency might be an early expression of medical diagnoses.
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http://dx.doi.org/10.1016/j.clnu.2019.12.021DOI Listing
September 2020

Bowel ultrasound measurements in healthy children - systematic review and meta-analysis.

Pediatr Radiol 2020 04 14;50(4):501-508. Epub 2019 Dec 14.

Pediatric Gastroenterology, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands.

Background: Ultrasound (US) is a noninvasive method of assessing the bowel that can be used to screen for bowel pathology, such as Inflammatory Bowel Disease, in children. Knowledge about US findings of the bowel in healthy children is important for interpreting US results in cases where disease is suspected.

Objective: To assess the bowel wall thickness in different bowel segments in healthy children and to assess differences in bowel wall thickness among pediatric age categories.

Materials And Methods: We conducted a systematic search in the PubMed, Embase, Cochrane, and CINAHL databases for studies describing bowel wall thickness measured by transabdominal US in healthy children. We excluded studies using contrast agent. We calculated the pooled mean and standard deviation scores and assessed differences among age categories (0-4 years, 5-9 years, 10-14 years, 15-18 years), first with analysis of variance (ANOVA) and further with subsequent Student's t-tests for independent samples, corrected for multiple testing.

Results: We identified 191 studies and included 7 of these studies in the systematic review. Reported bowel wall thickness values ranged from 0.8 mm to 1.9 mm in the small bowel and from 1.0 mm to 1.9 mm in the colon. The mean colonic bowel wall thickness is larger in children ages 15-19 years compared to 0-4 years (range in difference: 0.3-0.5 mm [corrected P<0.02]).

Conclusion: The reported upper limit of bowel wall thickness in healthy children is 1.9 mm in the small bowel and the colon, and mean thickness increases slightly with age in jejunum and colon. These values can be used as guidance when screening for bowel-related pathology in children.
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http://dx.doi.org/10.1007/s00247-019-04567-2DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7067709PMC
April 2020

[Foreign body ingestion in children].

Ned Tijdschr Geneeskd 2019 11 7;163. Epub 2019 Nov 7.

Emma Kinderziekenhuis, Amsterdam UMC, locatie AMC-UvA, Afd. Kinder-mdl, Amsterdam.

Foreign body ingestion occurs frequently in children and may lead to severe complications and mortality. In this article, three cases are presented. A 2-year-old boy swallowed a plastic toy helmet. He had no symptoms and physical examination was normal, and the toy was found in the stool within three days. Similarly, a 6-year-old girl swallowed two magnets and was asymptomatic. Physical examination was normal and a radiograph showed a foreign body which had passed the stomach. Due to the location, endoscopic removal by gastroduodenoscopy was not possible and surgical removal unnecessary. The magnets were secreted in the stool within two days. A 10-year-old boy with VACTERL association and psychiatric history, swallowed a button battery. After a delay in presentation, a radiograph showed a button battery mid-esophageal, which was endoscopically removed. He also needed dilatation due to esophageal stenosis. The above cases are all illustrative of the topic covered in the guideline 'Ingestion of foreign bodies in children aged 0-18 years', which was developed on behalf of the Dutch Pediatric Association and published in March 2019.
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November 2019

Premedication with intravenous steroids does not influence the incidence of infusion reactions following infliximab infusions in pediatric inflammatory bowel disease patients-a case-control study.

Eur J Clin Pharmacol 2019 Oct 22;75(10):1445-1450. Epub 2019 Jul 22.

Emma Children's Hospital, Amsterdam UMC, Pediatric Gastroenterology, Vrije Universiteit Amsterdam, Amsterdam, The Netherlands.

Purpose: Infusion reactions (IR) are commonly described side effects of infliximab (IFX) infusions, often leading to discontinuation of IFX. This study aimed to investigate the influence of steroid premedication (PM) on incidence of IR in pediatric inflammatory bowel disease (PIBD) patients receiving IFX.

Methods: A case-control study in two tertiary centers in Amsterdam, The Netherlands, including PIBD patients receiving IFX. PM with steroids was part of standard care in one center (PM+) but not in the other center (PM-). Acute IR were divided into mild/severe reactions and in grade 1/2/3/4 for detailed exploration. Differences between subgroups were assessed with the T or chi-square test. Multivariate logistic regression was used to assess associations between PM and IR incidence, correcting for co-medication usage.

Results: We included 226 patients (91 PM+, 50% male, mean age at onset of IBD 12.7 years), receiving 3433 infusions. There was no difference between the PM+ and PM- subgroups in incidence of IR (14.3% vs. 17.0% of patients, p = 0.58) and in percentage of infusions followed by IR (1.4% in both subgroups). The OR of developing IR when using PM was 1.06 (95% CI 0.49-2.27, p = 0.89), and the OR of developing a grade 3 or 4 IR when using PM was 0.90 (95% CI 0.24-3.39, p = 0.88) when correcting for co-medication usage.

Conclusion: The incidence of IR was low, and premedication with steroids did not decrease the incidence of IR in this cohort of PIBD patients receiving IFX. Our results indicate that PM with steroids is not indicated in PIBD to prevent IR.
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http://dx.doi.org/10.1007/s00228-019-02715-zDOI Listing
October 2019

Diagnostic Accuracy of Transabdominal Ultrasound in Detecting Intestinal Inflammation in Paediatric IBD Patients-a Systematic Review.

J Crohns Colitis 2019 Dec;13(12):1501-1509

Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Pediatric Gastroenterology, Amsterdam, The Netherlands.

Background And Aims: Currently used non-invasive tools for monitoring children with inflammatory bowel disease [IBD], such as faecal calprotectin, do not accurately reflect the degree of intestinal inflammation and do not provide information on disease location. Ultrasound [US] might be of added value. This systematic review aimed to assess the diagnostic test accuracy of transabdominal US in detecting intestinal inflammation in children with IBD in both diagnostic and follow-up settings.

Methods: We systematically searched PubMed, Embase [Ovid], Cochrane Library, and CINAHL [EBSCO] databases for studies assessing diagnostic accuracy of transabdominal US for detection of intestinal inflammation in patients diagnosed or suspected of IBD, aged 0-18 years, with ileo-colonoscopy and/or magnetic resonance enterography [MRE] as reference standards. Studies using US contrast were excluded. Risk of bias was assessed with QUADAS-2.

Results: The search yielded 276 records of which 14 were included. No meta-analysis was performed, because of heterogeneity in study design and methodological quality. Only four studies gave a clear description of their definition for an abnormal US result. The sensitivity and specificity of US ranged from 39-93% and 90-100% for diagnosing de novo IBD, and 48-93% and 83-93% for detecting active disease during follow-up, respectively.

Conclusions: The diagnostic accuracy of US in detecting intestinal inflammation as seen on MRE and/or ileo-colonoscopy in paediatric IBD patients remains inconclusive, and there is currently no consensus on defining an US result as abnormal. Prospective studies with adequate sample size and methodology are needed before US can be used in the diagnostics and monitoring of paediatric IBD.
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http://dx.doi.org/10.1093/ecco-jcc/jjz085DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7142400PMC
December 2019

Quality of life and colorectal function in Crohn's disease patients that underwent ileocecal resection during childhood.

Eur J Pediatr 2019 Sep 20;178(9):1413-1421. Epub 2019 Jul 20.

Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital, Academic Medical Center, Meibergdreef 9 (room C2-312), 1105 AZ, Amsterdam, The Netherlands.

Psychosocial and functional outcomes after intestinal resection in pediatric Crohn's disease (CD) are lacking. Therefore, we (I) assessed health-related quality of life (HRQOL), colorectal function, and satisfaction with surgery and (II) investigated their relationship with surgical outcomes, after ileocecal resection for CD. Crohn's patients that underwent ileocecal resection during childhood were included. HRQOL and colorectal function were assessed using SF-36 and COREFO, respectively, and compared with reference values. Satisfaction was scored on a 5-point Likert scale. In total, 80 patients (50% male, median age 23.0 years) were included. Physical HRQOL was impaired (SF-36 [mean]: CD, 47 vs. general, 54; p < 0.001), while mental HRQOL was similar to that in the general population. Overall colorectal function was impaired (COREFO [mean]: CD, 12.6 vs. normal, 7.2; p < 0.001). Worse colorectal function was associated with increasing clinical disease activity and longer interval since resection. Majority of patients was satisfied with surgery (81% satisfied/very satisfied, 11% neither satisfied nor dissatisfied, 8% dissatisfied/very dissatisfied). Decreased satisfaction with surgery was associated with increased clinical disease activity but not related to colorectal function.Conclusions: Physical HRQOL and colorectal function in CD patients who underwent ileocecal resection during childhood seem impaired and related to adverse surgical outcomes. This emphasizes the need for post-operative monitoring and prophylactic therapies. What is Known: • Up to 25% of pediatric-onset Crohn's disease (CD) patients undergo an intestinal resection within 5 years from diagnosis. • Many children and adults with CD experience disruption of their daily activities and health-related quality of life (HRQOL). What is New: • Physical HRQOL and colorectal function are impaired in patient with CD that underwent ileocecal resection during childhood. • Increasing clinical disease activity, a longer interval since surgery, severe complications related to surgery, and recurrent surgeries are all associated with worse colorectal function.
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http://dx.doi.org/10.1007/s00431-019-03427-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6694081PMC
September 2019

Health related quality of life of infants and children with avoidant restrictive food intake disorder.

Int J Eat Disord 2019 04 7;52(4):410-418. Epub 2019 Feb 7.

Department of Pediatric Gastroenterology, Hepatology, and Nutrition, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands.

Objective: The aim of this study was to compare health related quality of life (HRQOL) in infants and children with avoidant restrictive food intake disorder (ARFID) to healthy and chronically ill controls.

Method: A cross-sectional study was conducted in children who meet ARFID criteria at our tertiary care pediatric feeding clinic (September 2014 to July 2016). Before consultation, parents of patients (n = 100) were asked to complete questionnaires to determine HRQOL: the TNO-AZL Preschool Children Quality of Life (0-5 years), and "Pediatric Quality of Life Inventory" (6-7 and 8-10 years). HRQOL of ARFID patients was compared to both healthy (0-5 years n = 241; 6-7 years n = 61; 8-10 years n = 192) and chronically ill (0-5 years n = 79; 6-7 years n = 11; 8-10 years n = 26) controls.

Results: The prevalence of ARFID was 64%. HRQOL of ARFID patients aged 0-5 years (n = 37) was significantly lower on 6/12 scales (appetite, lungs, stomach, motor functioning, positive mood and liveliness) compared to healthy controls (P < .01), and on 4/12 scales (appetite, stomach, motor functioning, and liveliness) compared to chronically ill controls (P < .01). The ARFID patients scored significantly better on the problem behavior scale compared to healthy and chronically ill controls (P < .01). ARFID patients aged 6-7 (n = 9) had significantly lower scores in 3/6 scales (total score, psychosocial health, and school functioning) (P < .01), and aged 8-10 (n = 2) had a significantly lower school functioning scale (P < .01) compared to healthy controls.

Conclusion: HRQOL of children with ARFID is decreased on multiple scales. The effect on HRQOL should be incorporated in clinical practice, and clinical studies should add HRQOL as an outcome measure.
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http://dx.doi.org/10.1002/eat.23037DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6594067PMC
April 2019

Serious complications after button battery ingestion in children.

Eur J Pediatr 2018 Jul 2;177(7):1063-1070. Epub 2018 May 2.

Emma Children's Hospital, Academic Medical Center, Amsterdam, Netherlands.

Serious and fatal complications after button battery ingestion are increasing worldwide. The aim of this study is to describe serious complications after battery ingestion in children in the Netherlands.All pediatric gastroenterologists in the Netherlands performing upper endoscopies were asked to report all serious complications after battery ingestion in children (0-18 years) between 2008 and 2016 retrospectively.Sixteen serious complications were reported: death after massive bleeding through esophageal-aortal fistula (n = 1), esophageal-tracheal fistula (n = 5), stenosis after (suspected) perforation and mediastinitis (n = 5), (suspected) perforation and mediastinitis (n = 3), vocal cord paralysis (n = 1), and required reintubation for dyspnea and stridor (n = 1). The median time interval between ingestion and presentation was 5 (IQR 2-258) h. All children were ≤ 5 (median 1.4; IQR 0.9-2.1) years. Vomiting (31.3%), swallowing/feeding problems (31.3%), and fever (31.3%) were the most common presenting symptoms; however, 18.8% of the patients were asymptomatic (n = 1 missing). All batteries were button batteries (75% ≥ 20 mm; 18.8% < 20 mm; n = 1 missing). The batteries were removed by esophagogastroduodenoscopy (50%) and rigid endoscopy (37.5%) or surgically (12.5%).

Conclusion: Sixteen serious complications occurred after small and large button batteries ingestion between 2008 and 2016 in both symptomatic and asymptomatic children in the Netherlands. Therefore, immediate intervention after (suspected) button battery ingestion is required. What is Known: • Button battery ingestion may result in serious and fatal complications. • Serious and fatal complications after button battery ingestion are increasing worldwide. What is New: • Sixteen serious complications after button battery ingestion occurred during 2008-2016 in children in the Netherlands. • Serious complications were also caused by small batteries (< 20 mm) in the Netherlands and also occurred in asymptomatic Dutch children.
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http://dx.doi.org/10.1007/s00431-018-3154-6DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5997112PMC
July 2018

Diet and Anthropometrics of Children With Inflammatory Bowel Disease: A Comparison With the General Population.

Inflamm Bowel Dis 2018 07;24(8):1632-1640

Department of Pediatric Gastroenterology, Emma Children's Hospital, Academic Medical Center, Amsterdam, the Netherlands.

Background: There is a lack of knowledge regarding the diet of children with inflammatory bowel disease (IBD). Therefore, we investigated dietary intake in pediatric IBD compared to the general population and assessed anthropometrics and food avoidance.

Methods: In this cross-sectional cohort study, patients younger than 18 years with IBD were included (2014-2017). Dietary intake (total energy, macro nutrients, food types) was assessed using a Food Frequency Questionnaire (FFQ) and compared with a matched reference population (n = 306).

Results: There were 102 patients included (55% male, median age 15.0 years). Median height Z-score was -0.39 (interquartile range [IQR]: -1.17 to 0.26). Growth failure (height Z-scores<-1.64) was present in 11% of patients. Median BMI Z-score was 0.36 (IQR: -0.70 to 0.96). Acute malnutrition (weight-for-height Z-score<-2) was present in 3% of patients. Energy intake was lower in pediatric IBD compared to the reference population (mean kilojoule/day: 8286 vs 9794, P < 0.001). Protein intake did not differ, while fat intake was higher in pediatric IBD (mean gram/day: 101 vs 91, P < 0.001), with higher intake of vegetable oils/fats. Carbohydrate intake was lower in pediatric IBD (mean gram/day: 267 vs 305, P < 0.001), with lower intake of food types high in sugar. Food avoidance was reported in 53% of patients, with frequent avoidance of spicy (46%), high-fat food (30%), and dairy (30%).

Conclusions: The diet of children with IBD differs from the general pediatric population, with lower energy intake and high rates of food avoidance. Evaluation of the dietary intake alongside anthropometrics may be important to prevent nutrition deficiencies and promote health.
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http://dx.doi.org/10.1093/ibd/izy027DOI Listing
July 2018

Prevalence and side effects of pediatric home tube feeding.

Clin Nutr 2019 02 15;38(1):234-239. Epub 2018 Feb 15.

Department of Pediatric Gastroenterology, Emma Children's Hospital, Box 22666, 1100 DD, Amsterdam, The Netherlands.

Background & Aims: Tube feeding ensures growth, but can have negative effects on health and psychosocial functioning, resulting in health related costs. The aims of this study were to determine the prevalence of pediatric home tube feeding in the Netherlands and to assess the clinical characteristics of tube fed children and side effects of tube feeding.

Methods: The prevalence of pediatric home tube feeding was calculated using data (2010-2014) of both the Medicines and Devices Information Project of the National Health Care Institute, and Statistics Netherlands. Subsequently, a cross-sectional parental online questionnaire was used to obtain data regarding clinical characteristics of tube fed children and side effects of tube feeding. Children aged ≤17 years receiving tube feeding ≥2 weeks were included.

Results: The prevalence of pediatric home tube feeding was 83-92:100,000 children/year. Parents of 279 children (53% boys) completed the questionnaire. Most children (88%) had ≥1 medical diagnosis, of which congenital abnormalities (42%), perinatal problems (38%) and neurologic diseases (16%) were most common. They had gastrostomy (60%), nasogastric (33%), or other tube types (7%). Parents of most children (74%) mentioned ≥1 side effect due to tube feeding. Vomiting (37%), lack of appetite (29%), and gagging (29%) were reported most frequently. Nasogastric tube placement resolved in negative experiences (94%).

Conclusions: The prevalence of pediatric home tube feeding varies between 83 and 92:100,000 children/year in the Netherlands. These children are characterized by various underlying medical diagnoses. Side effects of tube feeding are frequently reported by parents. Further studies should focus on methods reducing side effects.
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http://dx.doi.org/10.1016/j.clnu.2018.01.027DOI Listing
February 2019

Efficacy of Home Telemonitoring versus Conventional Follow-up: A Randomized Controlled Trial among Teenagers with Inflammatory Bowel Disease.

J Crohns Colitis 2018 Mar;12(4):432-441

Paediatric Gastroenterology, Hepatology and Nutrition, Department of Paediatrics, University of Groningen, University Medical Center Groningen, Hanzeplein, Groningen, The Netherlands.

Background And Aims: Conventional follow-up of teenagers with inflammatory bowel diseases [IBD] is done during scheduled outpatient visits regardless of how well the patient feels. We designed a telemonitoring strategy for early recognition of flares and compared its efficacy with conventional follow-up.

Methods: We used a multicentre randomized trial in patients aged 10-19 years with IBD in clinical remission at baseline. Participants assigned to telemonitoring received automated alerts to complete a symptom score and send a stool sample for measurement of calprotectin. This resulted in an individual prediction for flare with associated treatment advice and test interval. In conventional follow-up the health check interval was left to the physician's discretion. The primary endpoint was cumulative incidence of disease flares. Secondary endpoints were percentage of participants with a positive change in quality-of-life and cost-effectiveness of the intervention.

Results: We included 170 participants [84 telemonitoring; 86 conventional follow-up]. At 52 weeks the mean number of face-to-face visits was significantly lower in the telemonitoring group compared to conventional follow-up [3.6 vs 4.3, p < 0.001]. The incidence of flares [33 vs 34%, p = 0.93] and the proportion of participants reporting positive change in quality-of-life [54 vs 44%, p = 0.27] were similar. Mean annual cost-saving was €89 and increased to €360 in those compliant to the protocol.

Conclusions: Telemonitoring is as safe as conventional follow-up, and reduces outpatient visits and societal costs. The positive impact on quality-of-life was similar in the two groups. This strategy is attractive for teenagers and families, and health professionals may be interested in using it to keep teenagers who are well out of hospital and ease pressure on overstretched outpatient services.

Trial Registration: NTR3759 [Netherlands Trial Registry].
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http://dx.doi.org/10.1093/ecco-jcc/jjx169DOI Listing
March 2018

Diagnostic test strategies in children at increased risk of inflammatory bowel disease in primary care.

PLoS One 2017 6;12(12):e0189111. Epub 2017 Dec 6.

Department of General Practice, University of Groningen, University Medical Center Groningen, Groningen, the Netherlands.

Background: In children with symptoms suggestive of inflammatory bowel disease (IBD) who present in primary care, the optimal test strategy for identifying those who require specialist care is unclear. We evaluated the following three test strategies to determine which was optimal for referring children with suspected IBD to specialist care: 1) alarm symptoms alone, 2) alarm symptoms plus c-reactive protein, and 3) alarm symptoms plus fecal calprotectin.

Methods: A prospective cohort study was conducted, including children with chronic gastrointestinal symptoms referred to pediatric gastroenterology. Outcome was defined as IBD confirmed by endoscopy, or IBD ruled out by either endoscopy or unremarkable clinical 12 month follow-up with no indication for endoscopy. Test strategy probabilities were generated by logistic regression analyses and compared by area under the receiver operating characteristic curves (AUC) and decision curves.

Results: We included 90 children, of whom 17 (19%) had IBD (n = 65 from primary care physicians, n = 25 from general pediatricians). Adding fecal calprotectin to alarm symptoms increased the AUC significantly from 0.80 (0.67-0.92) to 0.97 (0.93-1.00), but adding c-reactive protein to alarm symptoms did not increase the AUC significantly (p > 0.05). Decision curves confirmed these patterns, showing that alarm symptoms combined with fecal calprotectin produced the diagnostic test strategy with the highest net benefit at reasonable threshold probabilities.

Conclusion: In primary care, when children are identified as being at high risk for IBD, adding fecal calprotectin testing to alarm symptoms was the optimal strategy for improving risk stratification.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0189111PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5718464PMC
December 2017

Parental Distress and Quality of Life in Pediatric Inflammatory Bowel Disease: Implications for the Outpatient Clinic.

J Pediatr Gastroenterol Nutr 2018 04;66(4):630-636

Department of Pediatric Gastroenterology.

Objectives: The interrelation between the course of inflammatory bowel disease (IBD) in children and parent's distress, and the subsequent impact this may have on health-related quality of life (HRQOL) of the child is unclear. Therefore, we investigated patient's HRQOL and parental distress and the association between the course of IBD, parental distress, and HRQOL of pediatric patients with IBD.

Methods: Pediatric patients with IBD (8-18 years) and parents were invited. Patients completed the Pediatric Quality of Life Inventory, and parents the Distress Thermometer for Parents, simultaneously. Disease course was expressed as current clinical disease activity or months since last IBD flare. Patient's HRQOL and parental distress were compared to healthy controls.

Results: In total, 87 patients (71% response rate, 59% boys, median age 15.2 years) and parents were included. Patients had an impaired total HRQOL (β = 0.125, P = 0.010), driven by lower physical (0.196, P = 0.001) and school (β = 0.232, P < 0.001) functioning. Parents of children with IBD exhibited comparable levels of distress to parents of healthy children on the total problem and most subdomain problem scores (practical, social, emotional, physical, and cognitive), yet experienced more frequent parenting problems (P = 0.025). More severe disease course (months since last IBD flare) was indirectly associated, through parental distress, with decreased HRQOL of patients.

Conclusions: Worse disease course is directly associated with increased distress of parents and indirectly with lower HRQOL of children and adolescents with IBD. Distress of parents may be considered in management of pediatric IBD to improve HRQOL of children.
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http://dx.doi.org/10.1097/MPG.0000000000001756DOI Listing
April 2018

Less Anti-infliximab Antibody Formation in Paediatric Crohn Patients on Concomitant Immunomodulators.

J Pediatr Gastroenterol Nutr 2017 10;65(4):425-429

*Department of Paediatric Gastroenterology, University Medical Centre-Wilhelmina Children's Hospital, Utrecht†Department of Paediatric Gastroenterology, University Medical Centre Groningen, University of Groningen, Groningen‡Department of Paediatric Gastroenterology, Maxima Medical Centre, Veldhoven§Department of Paediatric Gastroenterology, Radboud University Medical Centre-Amalia Children's Hospital, Nijmegen||Department of Paediatric Gastroenterology, Emma Children's Hospital, Academic Medical Center, Amsterdam¶Department of Paediatric Gastroenterology, Erasmus Medical Centre-Sophia Children's Hospital, Rotterdam, The Netherlands.

Objectives: To evaluate the effect of immunomodulators on formation of antibodies to infliximab (ATI) in paediatric patients with Crohn disease (CD) and the association of ATI and loss of response.

Methods: Retrospective multicentre observational study (January 2009-December 2014) among Dutch children with CD treated with infliximab (IFX). ATI formation was analysed with Chi-square test and time-to-ATI formation with Kaplan-Meier and log-rank test.

Results: A total of 229 children were identified. ATIs were measured in 162 patients (70.7%) and 25 (15%) developed ATIs: 6 of 62 (10%) on continuous combined immunosuppression (CCI), 11 of 81 (14%) on early combined immunosuppression (ECI), and 8 of 19 (42%) on IFX monotherapy. ATI formation was higher in patients on IFX monotherapy compared to CCI (P = 0.003) and ECI (P = 0.008), whereas no significant difference was found between CCI and ECI. Sixteen out of 25 patients (64%) with ATIs had loss of response, compared with 32 of 137 patients (19%) without ATIs (P < 0.00002, log rank 0.02). Among patients treated with ECI, 10 of 55 (18%) developed ATIs within the first 12 months, compared to 1 of 26 (4%) after more than 12 months.

Conclusions: In children with CD combination therapy is associated with significant reduction of antibody formation and prolonged effectivity compared to IFX monotherapy. ECI for at least 12 months, followed by IFX monotherapy, may be an equally effective alternative to CCI.
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http://dx.doi.org/10.1097/MPG.0000000000001551DOI Listing
October 2017

Do Children and Adolescents With Inflammatory Bowel Disease Complete Clinical Disease Indices Similar to Physicians?

J Pediatr Gastroenterol Nutr 2018 03;66(3):410-416

Department of Pediatric Gastroenterology, Emma Children's Hospital/Academic Medical Center, Amsterdam, The Netherlands.

Objectives: The degree to which children and adolescents with inflammatory bowel disease (IBD) complete clinical disease activity indices in accordance with their physician is indefinite. Therefore, we investigated the agreement between patient- and physician-based clinical indices in children and adolescents with a previous diagnosis of IBD.

Methods: In this cross-sectional study, IBD patients (8-18 years) were included prospectively. Patients completed a patient-based short Pediatric Crohn's Disease Activity Index (shPCDAI) for Crohn disease or the Pediatric Ulcerative Colitis Activity Index (PUCAI) for ulcerative or indeterminate colitis. Physicians completed the original physician-based shPCDAI or PUCAI. Agreement was calculated with linear weighted kappa.

Results: In total, 154 pairs of clinical indices were collected: 89 pairs of shPCDAI's (median age at assessment 15.6 years, 61% men) and 55 pairs of PUCAI's (median age at assessment 14.0 years, 44% men). The shPCDAI disease activity category only fairly agreed between patient- and physician-based indices (kappa: 0.40 [95% confidence interval 0.24-0.55], P < 0.001), with perfect agreement in 58% of pairs. In the majority of disagreement (81%), patients scored in a higher shPCDAI disease activity category. The PUCAI disease activity category substantially agreed between patient- and physician-based indices (kappa: 0.64 [95% confidence interval 0.45-0.83], P < 0.001), with perfect agreement in 78% of pairs. In the majority of disagreement (75%), patients scored in a higher PUCAI disease activity category.

Conclusions: Patient- and physician-based shPCDAI and PUCAI do not always agree, particularly the shPCDAI, and therefore, should not be interpreted equivalently in management and research on children and adolescents with IBD.
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http://dx.doi.org/10.1097/MPG.0000000000001712DOI Listing
March 2018

The child's perspective on discomfort during medical research procedures: a descriptive study.

BMJ Open 2017 Aug 1;7(7):e016077. Epub 2017 Aug 1.

Department of Ethics and Philosophy, Erasmus University Medical Centre, Rotterdam, The Netherlands.

Objective: The evaluation of discomfort in paediatric research is scarcely evidence-based. In this study, we make a start in describing children's self-reported discomfort during common medical research procedures and compare this with discomfort during dental check-ups which can be considered as a reference level of a 'minimal discomfort' medical procedure. We exploratory study whether there are associations between age, anxiety-proneness, gender, medical condition, previous experiences and discomfort. We also describe children's suggestions for reducing discomfort.

Design: Cross-sectional descriptive study.

Setting: Paediatric research at three academic hospitals.

Patients: 357 children with and without illnesses (8-18 years, mean=10.6 years) were enrolled: 307 from paediatric research studies and 50 from dental care.

Main Outcome Measures: We measured various generic forms of discomfort (nervousness, annoyance, pain, fright, boredom, tiredness) due to six common research procedures: buccal swabs, MRI scans, pulmonary function tests, skin prick tests, ultrasound imaging and venepunctures.

Results: Most children reported limited discomfort during the research procedures (means: 1-2.6 on a scale from 1 to 5). Compared with dental check-ups, buccal swab tests, skin prick tests and ultrasound imaging were less discomforting, while MRI scans, venepunctures and pulmonary function tests caused a similar degree of discomfort. 60.3% of the children suggested providing distraction by showing movies to reduce discomfort. The exploratory analyses suggested a positive association between anxiety-proneness and discomfort.

Conclusions: The findings of this study support the acceptability of participation of children in the studied research procedures, which stimulates evidence-based research practice. Furthermore, the present study can be considered as a first step in providing benchmarks for discomfort of procedures in paediatric research.
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http://dx.doi.org/10.1136/bmjopen-2017-016077DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5642655PMC
August 2017

Application of Population Pharmacokinetic Modeling for Individualized Infliximab Dosing Strategies in Crohn Disease.

J Pediatr Gastroenterol Nutr 2017 12;65(6):639-645

Stanford Children's IBD Center, Division of Gastroenterology, Department of Pediatrics, Stanford University School of Medicine, Stanford, CA.

Objectives: The pharmacokinetics of infliximab (IFX) is highly variable in children with Crohn disease (CD), and a one-size-fits-all approach to dosing is inadequate. Model-based drug dosing can help individualize dosing strategies. We evaluated the predictive performance and clinical utility of a published population pharmacokinetic model of IFX in children with CD.

Methods: Within a cohort of 34 children with CD who had IFX trough concentrations measured, the pharmacokinetics of each patient was estimated in NONMEM using a published population pharmacokinetic model. Infliximab concentrations were then predicted based on each patient's dosing history and compared with actual measured concentrations (n = 59). In addition, doses 5 to 10 mg/kg and dosing intervals every 4 to 8 weeks were simulated in each patient to examine dose-trough relationships.

Results: Predicted concentrations were within ±1.0 μg/mL of actual measured concentrations for 88% of measurements. The median prediction error (ie, measure of bias) was -0.15 μg/mL (95% confidence interval -0.37 to -0.05 μg/mL) and absolute prediction error (ie, measure of precision) was 0.26 μg/mL (95% confidence interval 0.15 to 0.40 μg/mL). At standard maintenance dosing of 5 mg/kg every 8 weeks, a trough >3 μg/mL was predicted to be achieved in 32% of patients. To achieve a trough >3 μg/mL, a dosing interval ≤every 6 weeks was predicted to be required in 29% of patients.

Conclusions: A published IFX population pharmacokinetic model demonstrated accurate predictive performance in a pediatric CD population. Individualized IFX dosing strategies in children with CD will be critical to consistently achieve trough concentrations associated with optimal outcomes.
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http://dx.doi.org/10.1097/MPG.0000000000001620DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5670026PMC
December 2017

Development, prevention, and treatment of feeding tube dependency.

Eur J Pediatr 2017 Jun 13;176(6):683-688. Epub 2017 Apr 13.

Emma Children's Hospital, Box 22666, 1100 DD, Amsterdam, The Netherlands.

Enteral nutrition is effective in ensuring nutritional requirements and growth. However, when tube feeding lasts for a longer period, it can lead to tube dependency in the absence of medical reasons for continuation of tube feeding. Tube-dependent children are unable or refuse to start oral activities and they lack oral skills. Tube dependency has health-, psychosocial-, and economy-related consequences. Therefore, the transition to oral feeding is of great importance. However, this transition can be very difficult and needs a multidisciplinary approach. Most studies for treatment of tube dependency are based on behavioral interventions, such as family therapy, individual behavior therapy, neuro-linguistic programming, and parental anxiety reduction. Furthermore, oral motor therapy and nutritional adjustments can be helpful in tube weaning. The use of medication has been described in the literature. Although mostly chosen as the last resort, hunger-inducing methods, such as the Graz-model and the Dutch clinical hunger provocation program, are also successful in weaning children off tube feeding.

Conclusion: The transition from tube to oral feeding is important in tube-dependent children but can be difficult. We present an overview for the prevention and treatment of tube dependency. What is known: • Longer periods of tube feeding can lead to tube dependency. • Tube weaning can be very difficult. What is new: • Weaning as soon as possible and therefore referral to a multidisciplinary team are recommended. • An overview of treatment options for tube dependency is presented in this article.
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http://dx.doi.org/10.1007/s00431-017-2908-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5432583PMC
June 2017

Humoral Immunodeficiency with Hypotonia, Feeding Difficulties, Enteropathy, and Mild Eczema Caused by a Classical Mutation.

Front Pediatr 2017 27;5:37. Epub 2017 Feb 27.

Department of Pediatric Hematology, Immunology, Rheumatology and Infectious Diseases, Emma Children's Hospital, Academic Medical Center (AMC), University of Amsterdam, Amsterdam, Netherlands; Department of Clinical Genetics, Academic Medical Center (AMC), University of Amsterdam, Amsterdam, Netherlands.

We describe here the case of a boy who presented with pulmonary infections, feeding difficulties due to velopharyngeal insufficiency and gastroesophageal reflux, myopathy, and hypotonia soon after birth. Later, he was also found to have an elevated immunoglobulin (Ig) E and mild eczema and was diagnosed with inflammatory bowel disease. Further immunological screening at the age of 7 years showed low B and NK cell numbers but normal CD4 and CD8 T cells and notably, normal numbers of CD4 regulatory T (Treg) cells. Serum IgG, IgA, and IgM were low to normal, but he had a deficient response to a pneumococcal polysaccharide vaccine and thus a humoral immunodeficiency. To our surprise, whole exome sequencing revealed a mutation in , encoding an essential transcription factor for the development and function of Treg cells. This classical mutation is associated with immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome. Further studies indeed showed defective function of Treg cells despite normal FOXP3 protein expression and nuclear localization. The boy underwent hematopoietic stem cell transplantation at 11 years of age and despite the temporary development of diabetes while on prednisone is now doing much better, IgE levels have declined, and his fatigue has improved. This case illustrates that a classical pathogenic mutation in can lead to a clinical phenotype where the diagnosis of IPEX syndrome was never considered because of the lack of diabetes and the presence of only mild eczema, in addition to the normal Treg cell numbers and FOXP3 expression.
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http://dx.doi.org/10.3389/fped.2017.00037DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5326763PMC
February 2017

Transverse Myelitis as Manifestation of Celiac Disease in a Toddler.

Pediatrics 2017 Mar 2;139(3). Epub 2017 Feb 2.

Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital, Amsterdam, Netherlands.

We present a 17-month-old girl with rapidly progressive unwillingness to sit, stand, play, and walk. Furthermore, she lacked appetite, vomited, lost weight, and had an iron deficiency. Physical examination revealed a cachectic, irritable girl with a distended abdomen, dystrophic legs with paraparesis, disturbed sensibility, and areflexia. An MRI scan revealed abnormal high signal intensity on T2-weighted images in the cord on the thoracic level, without cerebral abnormalities, indicating transverse myelitis (TM). Laboratory investigations revealed elevated immunoglobulin A antibodies against gliadin (1980.0 kU/L; normal, 0-10.1 kU/L) and tissue transglutaminase (110.0 kU/L; normal, 0-10.1 kU/L). Gastroscopy revealed villous atrophy in the duodenal biopsies and lymphocytic gastritis according to Marsh IIIb, compatible with celiac disease (CD). After the start of a gluten free diet and methylprednisolone, she recovered completely. To our knowledge, this is the first pediatric case of TM as manifestation of CD. We suggest that all children with TM or other neurologic manifestations of unknown origin should be screened for CD.
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http://dx.doi.org/10.1542/peds.2016-1381DOI Listing
March 2017

Complications and Disease Recurrence After Primary Ileocecal Resection in Pediatric Crohn's Disease: A Multicenter Cohort Analysis.

Inflamm Bowel Dis 2017 02;23(2):272-282

*Department of Pediatric Gastroenterology and Nutrition, Emma Children's Hospital, Academic Medical Center, Amsterdam, the Netherlands; †Department of Pediatric Gastroenterology, Erasmus MC-Sophia Children's Hospital, Rotterdam, the Netherlands; ‡Department of Pediatric Gastroenterology, University Medical Centre Groningen, University of Groningen, Groningen, the Netherlands; §Department of Pediatric Gastroenterology, University Medical Center Utrecht, Utrecht, the Netherlands; ‖Department of Pediatrics, Leiden University Medical Center, Leiden, the Netherlands; ¶Department of Pediatric Gastroenterology, Radboud University Medical Center, Nijmegen, the Netherlands; **Department of Pediatric Gastroenterology, VU University Medical Center, Amsterdam, the Netherlands; ††Department of Pediatrics, Amphia Hospital, Breda, the Netherlands; ‡‡Department of Pediatric Surgery, Academic Medical Center, Amsterdam, the Netherlands; and §§Department of Pediatric Surgery, Erasmus MC-Sophia Children's Hospital, Rotterdam, the Netherlands.

Background: Studies on the outcome of ileocecal resection in pediatric Crohn's disease (CD) have a limited follow-up and fail to assign predictors of adverse outcomes. Therefore, we aimed to investigate (I) the complication and disease recurrence rates and (II) identify risk factors for these adverse outcomes after ileocecal resection for pediatric CD.

Methods: This is a retrospective cohort analysis of all children (<18 years) that underwent ileocecal resection as first intestinal resection for CD derived from 7 tertiary hospitals in the Netherlands (1990-2015). Risk factors were identified using multivariable analysis.

Results: In total, 122 children were included (52% male; median age 15.5 years [interquartile range 14.0-16.0]). Severe postoperative complications rate was 10%. Colonic disease (odds ratio: 5.6 [95% confidence interval {CI}: 1.3-26.3], P = 0.024), microscopically positive resection margins (odds ratio: 10.4 [95% CI: 1.1-100.8] P = 0.043), and emergency surgery (odds ratio: 6.8 [95% CI: 1.1-42.2], P = 0.038) were risk factors for severe complications. Clinical and surgical recurrence rates after 1, 5 and 10 years were 19%, 49%, 71% and 2%, 12%, 22%, respectively. Female sex (hazard ratio [HR]: 2.1 [95% CI: 1.1-3.8], P = 0.023) was a risk factor for clinical recurrence, whereas ileocecal disease (HR: 3.9 [95% CI: 1.2-12.5], P = 0.024) and microscopically positive resection margins (HR: 9.6 [95% CI: 1.2-74.5], P = 0.031) were risk factors for surgical recurrence. Immediate postoperative therapy reduced the risk of both clinical (HR: 0.3 [95% CI: 0.1-0.6], P = 0.001) and surgical (HR: 0.5 [95% CI: 0.1-0.9], P = 0.035) recurrence.

Conclusions: Ileocecal resection is an effective and durable treatment of pediatric CD, although postoperative complications occur frequently. Postoperative therapy may be started immediately to prevent disease recurrence.
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http://dx.doi.org/10.1097/MIB.0000000000000999DOI Listing
February 2017

Diagnostic Accuracy of Fecal Calprotectin for Pediatric Inflammatory Bowel Disease in Primary Care: A Prospective Cohort Study.

Ann Fam Med 2016 09;14(5):437-45

Department of General Practice, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands

Purpose: In specialist care, fecal calprotectin (FCal) is a commonly used noninvasive diagnostic test for ruling out inflammatory bowel disease (IBD) in children with chronic gastrointestinal symptoms. The aim of this study was to evaluate the diagnostic accuracy of FCal for IBD in symptomatic children in primary care.

Methods: We studied 2 prospective cohorts of children with chronic diarrhea, recurrent abdominal pain, or both: children initially seen in primary care (primary care cohort) and children referred to specialist care (referred cohort). FCal (index test) was measured at baseline and compared with 1 of the 2 reference standards for IBD: endoscopic assessment or 1-year follow-up. Physicians were blinded to FCal results, and values greater than 50 μg/g feces were considered positive. We determined specificity in the primary care cohort and sensitivity in the referred cohort.

Results: None of the 114 children in the primary care cohort ultimately received a diagnosis of IBD. The specificity of FCal in the primary care cohort was 0.87 (95% CI, 0.80-0.92). Among the 90 children in the referred cohort, 17 (19%) ultimately received a diagnosis of IBD. The sensitivity of FCal in the referred cohort was 0.99 (95% CI, 0.81-1.00).

Conclusions: The findings of this study suggest that a positive FCal result in children with chronic gastrointestinal symptoms seen in primary care is not likely to be indicative of IBD. A negative FCal result is likely to be a true negative, which safely rules out IBD in children in whom a primary care physician considers referral to specialist care.
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http://dx.doi.org/10.1370/afm.1949DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5394359PMC
September 2016

Relationship of clinical symptoms with biomarkers of inflammation in pediatric inflammatory bowel disease.

Eur J Pediatr 2016 Oct 29;175(10):1335-42. Epub 2016 Aug 29.

Department of Pediatric Gastroenterology and Nutrition, Academic Medical Center, Meibergdreef 9, 1105 AZ, Amsterdam, The Netherlands.

Unlabelled: In adult inflammatory bowel disease (IBD) patients, there is a strong discrepancy between symptoms and biomarkers of inflammation. Data on pediatric IBD patients are conflicting. Therefore, we aimed to investigate the relationship between clinical symptoms and biomarkers of inflammation in pediatric IBD. Patients aged <18 years with previously diagnosed Crohn's disease (CD) or ulcerative colitis (UC) were included. Clinical disease activity was determined using the abbreviated Pediatric CD Activity Index (aPCDAI) or Pediatric UC Activity Index (PUCAI). Biochemical disease activity was assessed using fecal calprotectin (FC) and C-reactive protein (CRP). In total, 127 patients (62 male; median age 14.9 years) were included (82 CD, 45 UC). FC correlated weakly with total aPCDAI score (r s = 0.32; 95 % CI 0.12-0.51; p = 0.003) and total PUCAI score (r s = 0.36; 95 % CI 0.07-0.62; p = 0.015). Only aPCDAI components abdominal examination and perirectal disease and PUCAI component activity level had a significant correlation with levels of FC. CRP correlated weakly with total aPCDAI score (r s = 0.28; 95 % CI 0.05-0.46; p = 0.012) and aPCDAI components abdominal examination and activity level. No significant correlation was observed between CRP and total PUCAI score (r s = 0.01; 95 % CI -0.34-0.29; p = 0.961) or individual PUCAI components.

Conclusion: There is a strong discrepancy between clinical symptoms and biomarkers of inflammation in children with IBD.

What Is Known: • A substantial proportion of asymptomatic pediatric inflammatory bowel disease (IBD) patients have elevated biomarkers of inflammation. • There is a strong discrepancy between symptoms and biomarkers of inflammation in adults with IBD. What is New: • Clinical symptoms are only weakly associated with levels of fecal calprotectin and serum C-reactive protein in children and adolescents with previously diagnosed IBD. • Similarly to adult IBD patients, there is a strong discrepancy between clinical symptoms and biomarkers of inflammation in children with IBD.
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http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5031739PMC
http://dx.doi.org/10.1007/s00431-016-2762-2DOI Listing
October 2016

Evaluation of point-of-care test calprotectin and lactoferrin for inflammatory bowel disease among children with chronic gastrointestinal symptoms.

Fam Pract 2017 08;34(4):400-406

Department of General Practice, University of Groningen, University Medical Center Groningen, PO Box 196, 9700 AD Groningen, The Netherlands.

Background: Faecal calprotectin is considered to be a valid test for ruling out inflammatory bowel disease (IBD) in children with chronic gastrointestinal symptoms in specialist care. In contrast, faecal lactoferrin has higher specificity. The recent availability of both as point-of-care tests (POCTs) makes them attractive for use in primary care.

Objective: To evaluate the test characteristics of calprotectin and lactoferrin POCTs for diagnosing IBD in symptomatic children.

Methods: We defined two prospective cohorts of children with chronic gastrointestinal symptoms: (i) children presenting to primary care (primary care cohort); (ii) children referred for specialist care (referred cohort). Baseline POCT results were compared with the outcome of either endoscopic assessment or 12 months follow-up. Clinicians were blinded to the POCT results.

Results: In the primary care cohort, none of the 114 children had IBD, and the calprotectin and lactoferrin POCTs had specificities of 0.95 (0.89-0.98) and 0.98 (0.93-0.99), respectively. In the referred cohort, 17 of the 90 children had IBD: the sensitivity of POCT calprotectin and POCT lactoferrin were both 0.94 (0.72-0.99); and the specificity was 0.93 (0.84-0.97) and 0.99 (0.92-1.00), respectively. The POCT calprotectin could reduce the referral rate by 76% and POCT lactoferrin by 81%, while missing one child with IBD (6%).

Conclusion: A diagnostic test strategy in primary care using a simple POCT calprotectin or lactoferrin has the potential to reduce the need for referral for further diagnostic work-up in specialist care, with a low risk of missing a child with IBD.
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http://dx.doi.org/10.1093/fampra/cmw079DOI Listing
August 2017