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Biomedical subjects

M A Benninga

Publications and source records attributed to M A Benninga.

At least 19 recordsLinked to original sources
PubMed2006

[Takayasu arteritis: a chronic vasculitis that is rare in children].

Takayasu arteritis was diagnosed in two children, a 15-year-old girl and a 10-year-old boy. The girl had suffered from fatigue, malaise, abdominal pain and weight loss for several months, but no clear cause could be found. A few weeks later, when a blood pressure of 222/155 mmHg was measured, the possibility of renal artery stenosis was considered and imaging studies revealed indications for Takayasu's disease. The patient was given methylprednisolone followed by a combination of prednisone and, initially, cyclophosphamide, later methotrexate. This resulted in a clinical remission of the inflammatory process. The boy presented with increasing fatigue and variable episodes of fever. After 3 years, sarcoidosis or Castleman's disease were considered. Imaging studies revealed aortic stenosis. He underwent stenting of the involved vessel segment. Takayasu arteritis is a chronic vasculitis of unknown origin, affecting mainly the aorta and its main branches. As a result of the inflammation, stenosis, occlusion or dilatation of the involved vessels may occur and cause a wide range of symptoms. Especially in the early phase, the symptoms often are non-specific. One should look for hypertension, blood pressure differences between the two arms, decreased peripheral pulsation or bruits over the aorta and its major branches. Radiological examination may consist ofangiography, magnetic resonance imaging or CT-scans. Treatment consists of corticosteroids and other immunosuppressants, such as cyclophosphamide, methotrexate, azathioprine, and antagonists of tumour-necrosis factor alpha. In addition, balloon dilatation or stenting is often necessary.

Adolescent↗
PubMed2006

Pharmacogenetics of thiopurine therapy in paediatric IBD patients.

BACKGROUND: Azathioprine is widely used in the treatment of children with inflammatory bowel disease. The occurrence and type of adverse events to azathioprine may be related to thiopurine S-methyltransferase (TPMT) enzyme activity and to inosine triphophate pyrophosphatase (ITPase) deficiency. AIM: Investigate frequencies of functional TPMT polymorphisms and ITPA polymorphisms and their association with the occurrence of adverse events during azathioprine therapy in a paediatric inflammatory bowel disease population. METHODS: Seventy-two azathioprine treated paediatric inflammatory bowel disease patients, 47% girls, mean age 12.5 years (range 6.5-17.5), were assessed for TPMT and ITPA polymorphisms and for adverse events. The relation between polymorphisms and adverse events is evaluated. RESULTS: Of all azathioprine treated patients, 11 experienced an adverse event for which azathioprine was stopped: pancreatitis (n = 4), leucopenia (n = 2) and 'general malaise' (n = 5). Of the 11 patients who stopped azathioprine because of adverse events, 10 had wild-type alleles for all investigated genotypes. Genotyping of ITPA 94C>A polymorphisms showed that two patients were homozygous, both tolerated azathioprine well. CONCLUSIONS: No association of functional ITPA and TPMT polymorphisms and the occurrence of azathioprine related adverse events could be detected. Pharmacogenetic assessment prior to thiopurine therapy does not seem warranted.

Adolescent↗
PubMed2006

Diagnostic tests in Hirschsprung disease: a systematic review.

OBJECTIVE: We conducted a systematic review to determine and compare the diagnostic accuracy of contrast enema (CE), anorectal manometry (ARM) and rectal suction biopsy (RSB) in infants suspected of Hirschsprung disease. DESIGN: This is a systematic review. DATA SOURCES: Articles were identified through electronic searches in Medline, EMBASE.com and Cochrane Controlled Trials Register. Searches were limited to articles published after 1966 in PubMed and after 1980 in EMBASE.com. STUDY SELECTION: Studies were included if infants underwent at least one of the following tests: CE, ARM or RSB, followed by full-thickness biopsy and/or clinical follow-up as the reference standard. DATA EXTRACTION: Two reviewers independently assessed the methods of data collection, patient selection, blinding and prevention of verification bias and description of the test protocol and reference standard. Data to construct 2 x 2 tables were abstracted for each test. RESULTS: Twenty-four studies met our inclusion criteria, but 2 studies were subsequently excluded for statistical analysis because data was missing to construct the 2 x 2 table. RSB (14 studies for a total of 993 patients) was the most accurate test, having both the highest mean sensitivity (93%; 95% confidence interval [CI], 88%-95%) and mean specificity (98%; 95% CI, 95%-99%). Sensitivity and specificity of ARM (9 studies for a total of 400 patients) were similar to those of RSB (91% vs 93%, P = 0.73 and 94% vs 98%, P = 0.08, respectively). Sensitivity and specificity of CE (12 studies for a total of 425 patients) were significantly lower than those of RSB and ARM, with mean sensitivity and mean specificity of 70% and 83%, respectively. CONCLUSIONS: RSB and ARM are the most accurate tests in the diagnostic workup of Hirschsprung disease.

Anal Canal↗
PubMed2006

Long-term results of boerema anterior gastropexy in children.

OBJECTIVES: To analyze the long-term results of Boerema anterior gastropexy using the Visick grading system in a university teaching hospital. METHODS: Retrospective review of 247 children with severe or complicated gastroesophageal reflux disease (GERD). Patients who underwent primary gastropexy between 1990 and 2001 were divided in 3 groups: (A) neurologic impaired patients (n = 83); (B) esophageal atresia patients (n = 36); and (C) patients without underlying disease (n = 128). The median length of follow-up after gastropexy was 7 years and 8 months (range, 1.5-13 years). Follow-up was carried out using a standardized questionnaire and was obtained by telephone. Postoperative recurrence of symptoms during follow-up was considered as failure of the operation. RESULTS: Postoperative complications occurred in 20 patients (24%) in group A, in 13 patients (36%) in group B and in 23 patients (18%) in group C. Thirty-three patients died during the follow-up period, 5 of whom during the postoperative period. Six children could not be traced; thus, 208 were available for long-term follow-up. Postoperative failures occurred in 12 patients (23%) in group A, in 11 patients (31%) in group B and in 17 patients in group C (14%). Reoperation for recurrence of symptoms due to GERD was performed in 6 patients in group A, in 7 patients in group B and in 2 patients in group C. The final outcome at the time of interview was successful in 81% of group A, in 88% of group B and in 91% of group C. CONCLUSIONS: In the long term, Boerema anterior gastropexy is an effective operation for complicated GERD in children without underlying disease as well as in neurologically impaired children and patients with esophageal atresia.

Adolescent↗
PubMed2006

Epidemiology of childhood constipation: a systematic review.

OBJECTIVE: A systematic review of the published literature was performed to assess the prevalence, incidence, natural history, and comorbid conditions of functional constipation in children. METHODS: Articles were identified through electronic searches in Medline, Embase, Cochrane Central Library, Cinhal and PsychInfo databases. Study selection criteria included: (1) epidemiology studies of general population, (2) on the prevalence of constipation without obvious organic etiology, (3) in children from 0 to 18 yr old, and (4) published in English and full manuscript form. RESULTS: Eighteen studies met our inclusion criteria. The prevalence of childhood constipation in the general population ranged from 0.7% to 29.6% (median 8.9; inter quartile range 5.3-17.4). The prevalence of constipation defined as defecation frequency of < 3/wk varied from 0.7% to 29.6% (median 10.4; inter quartile range 1.3-21.3). Identified studies originated from North America (N = 4), South America (N = 2), Europe (N = 9), the Middle-East (N = 1), and Asia (N = 2). Variance of gender specific prevalence was reported in seven studies and five of seven studies reported no significant difference between boys and girls. The age group in which constipation is most common could not be assessed with certainty. Socioeconomic factors were not found to be associated with constipation. CONCLUSION: Childhood constipation is a common problem worldwide. Most studies report similar prevalence rates for boys and girls. Large epidemiologic studies with the use of generally accepted diagnostic criteria are needed to define the precise prevalence of constipation.

Adolescent↗
PubMed2005

[Video-capsule endoscopy in children with unexplained symptoms of the small intestine].

Video-capsule endoscopy was used in 4 children with unexplained symptoms of the small intestine. Each patient swallowed a capsule of 11 by 27 mm, which contained a camera that takes 2 images per second (in children aged less than 8 years, the capsule was placed in the duodenum under sedation). In a 3-year-old girl with rectal bleeding following partial resection of the colon and small intestine, ulcers were seen proximal to the ileorectal anastomosis. In a 14-year-old boy with Crohn's disease and an abnormal growth curve, multiple stenoses of the small intestine were seen. In an 8-year-old boy with rectal bleeding, a solitary polypoid mass was seen that, upon operation, appeared to be the result of a partially invaginated Meckel's diverticulum. In a 17-year-old boy with weight loss, rectal bleeding and colitis, abnormalities were seen that were consistent with Crohn's disease. Patients were treated based on the endoscopic results and subsequently recovered. Video-capsule endoscopy is non-invasive and painless and provides better images of the small intestine than a standard endoscopic and radiological examination.

Adolescent↗
PubMed2005

[The treatment of chronic hepatitis C virus infection in children].

A 9-year-old boy, a 15-year-old boy, and a 6-year-old girl were infected with chronic hepatitis C virus (HCV). They had no physical complaints and a virus genotype that was favourable to treatment with peginterferon-alpha and ribavirin. The younger boy and the girl had liver fibrosis and were treated for 6 months; the virus was eradicated from the boy's plasma and the fibrosis diminished, while the girl's plasma virus was again present shortly after the end of treatment. In the older boy with no fibrosis, treatment was temporarily suspended due to behaviour problems. HCV infection is a frequent cause of chronic hepatitis in children. A better understanding of its natural history, improvements in the efficacy of treatment, and more favourable outcomes seen in children compared with adults have gradually changed the consideration to treat children with chronic HCV infection over the last 10 years. The decision whether or not to treat depends primarily on the degree of liver damage, virus genotype, and the psychological condition and motivation of the patient. Screening patients at risk for chronic HCV infection and careful follow-up for liver damage in those with HCV infection have become even more important given the new insights regarding treatment.

Adolescent↗
PubMed2005

[Defecation disorders in children: treatment with colonic irrigation through an appendicostomy].

In 3 patients with serious constipation or faecal incontinence the appendix was sewn open-ended into the abdominal wall (Malone-stoma) allowing antegrade bowel washouts. The patients were a 4-year-old boy with Hirschsprung's disease, a 15-year-old boy with chronic functional constipation and encopresis, and a 5.5-year-old boy with incontinence associated with a meningomyelocele. Following the procedure they were free from bowel problems. Faecal incontinence has a major detrimental effect on the psychosocial development of children. Severe constipation and faecal incontinence which do not optimally respond to conventional therapy can be treated by this procedure. Treatment is effective in around three-quarters of these children. Complications occur in an equal number of children, stenosis being the most frequently seen.

Adolescent↗
PubMed2005

Functional constipation in infants: a follow-up study.

Our objective of this study was to describe the clinical course of severe functional constipation in early childhood. Eligible patients were 47 children (60% boys; median age, 3.5 months) who had constipation in their first year of life. Follow-up data were obtained through a standardized questionnaire. Success was defined as a period of at least 4 weeks with > or =3 painless bowel movements per week. Six months after initial evaluation, 69% of the children were recovered. After initial success, a relapse occurred in 15% of the children within 3 years. A duration of symptoms <3 months before referral correlated significantly with better outcome. We conclude that most infants with severe constipation evaluated at a tertiary center are recovered after 6 months. Early therapeutic intervention may beneficially contribute to the resolution of constipation.

Cathartics↗
PubMed2005

The prevalence of encopresis in a multicultural population.

BACKGROUND: Population-based studies on the prevalence of encopresis in children are scarce and generally outdated. Prevalence estimates based on clinical studies are unreliable because parents tend to be reticent to seek medical help for this problem. Professional help is necessary, however, because encopresis can lead to serious psychosocial health problems. The authors examined the prevalence of encopresis in children, the frequency of visits made to general practitioners for encopresis and the psychosocial health problems of encopretic children. METHODS: This population-based study involved 13,111 parents and their 5- to 6-year-old children and 9,780 parents and their 11- to 12-year-old children, all residents of Amsterdam, the Netherlands. RESULTS: The prevalence of encopresis was 4.1% in the 5-to-6 age group and 1.6% in the 11-to-12 age group. Encopresis was more frequent among boys and children from the very depressed areas of the city. Encopresis was less frequent among Moroccan and Turkish children. A defecation frequency of less than three per week was found in 3.8% of the 5- to 6-year-olds and 10.1% of the 11- to 12-year-olds with encopresis. Only 37.7% of the 5- to 6-year-olds and 27.4% of the 11- to 12-year-olds who had encopresis had ever been taken to see a doctor for this problem. Psychosocial problems were far more common among children with encopresis than among normal children. CONCLUSIONS: Encopresis is a common condition that is often associated with psychosocial health disorders but only a small proportion of the children with encopresis are taken to a general practitioner to discuss their problem.

Child↗
PubMed2005

Assessment of the rectoanal inhibitory reflex in preterm infants with delayed meconium passage.

BACKGROUND: There is an inverse relationship between gestational age, birth weight and the time of first neonatal bowel movement. The authors hypothesized that delayed passage of meconium might result from a delayed maturation of the recto-anal inhibitory reflex (RAIR) in premature infants. OBJECTIVE: To evaluate whether the RAIR is absent in very preterm infants 28-32 weeks postmenstrual age with delayed meconium production. STUDY DESIGN: Anorectal manometry was performed in 10 preterm infants (seven male) with delayed meconium production (no meconium in the first 48 hours). Median postmenstrual age was 30 weeks (28-31 weeks). Birth weight ranged from 780 to 1930 g (median, 1395 g). A micromanometric assembly (outer diameter, 2.0 mm) was used which incorporated a 1.5-cm-long sleeve sensor for measurement of resting anal sphincter pressure and relaxation. Four side-holes recorded anal and rectal pressures. Rectal distension was performed with direct air insufflation to elicit the RAIR. RESULTS: The time from birth to passage of meconium ranged from 48 to 105 hours (median, 82 hours). The mean anal sphincter pressure, rectal pressure, and anal sphincter oscillation frequency were 22.0 +/- 5.0 mm Hg, 6.9 +/- 2.0 mm Hg, and 9.8 +/- 1.9/min, respectively. A normal RAIR was elicited in all infants. CONCLUSION: Anorectal manometry recordings in premature infants with delayed passage of meconium showed normal anorectal pressures and a normal RAIR, suggesting that delayed meconium passage is not related to the absence of a RAIR.

Anal Canal↗
PubMed2005

Interstitial cells of Cajal are involved in the afferent limb of the rectoanal inhibitory reflex.

BACKGROUND AND AIMS: Interstitial cells of Cajal (ICC) have been shown to be involved in nitrergic neurotransmission of the lower oesophageal sphincter and pylorus. Here we studied the role of ICC and nitric oxide (NO) in the inhibitory neurotransmission of the murine internal anal sphincter (IAS). METHODS: The rectoanal inhibitory reflex, rectal compliance, and relaxation of the isolated IAS to electrical stimulation were measured in controls, KIT (W)/KIT (Wv) mice, and neuronal NO synthase (nNOS) deficient mice. In addition, we evaluated the effect of blockade of nNOS using N-nitro-L-arginine methyl ester. Distribution of nNOS positive neurones and ICC in the IAS was assessed immunohistochemically. RESULTS: KIT positive ICC were present in a dense network in the IAS of controls but not in KIT (W)/KIT(Wv) mice. Relaxation of IAS muscle strips induced by electrical stimulation was diminished in nNOS-/- mice but not in KIT (W)/KIT (Wv) mice. Blockade of NOS reduced the relaxation of IAS muscle strips in both mice. Relaxation of the IAS to rectal distension was significantly diminished in KIT (W)/KIT (Wv) mice and nNOS deficient mice. In concert, in vivo blockade of NOS attenuated the relaxation of the IAS in controls. No significant difference in compliance was found. CONCLUSION: The inhibitory innervation of the IAS and the rectoanal inhibitory reflex are mediated by NO and the rectoanal inhibitory reflex requires an intact network of ICC in the IAS. Thus both loss of nitrergic innervation and deficiency of ICC lead to impaired anal relaxation and may play an important role in rectal evacuation disorders.

Anal Canal↗
PubMed2004

[Dientamoeba fragilis: possibly an important cause of persistent abdominal pain in children].

OBJECTIVE: To determine the effect of an antiprotozoic treatment on children with persistent abdominal pain and infection with Dientamoeba fragilis. DESIGN: Retrospective. METHOD: A total of 43 children with D. fragilis infection and persistent gastrointestinal complaints were included in the study. Of these 27 were treated with clioquinol and 16 with a nitroimidazole drug: metronidazole or tinidazole. The parasitological and clinical effects of the treatment were assessed. RESULTS: In 33 of the 43 (77%) children, no parasites were detected during follow-up with a triple faeces test: 22/27 following treatment with clioquinol and 11/16 following treatment with a nitroimidazole drug. In 27 of the 33 (82%) children with a negative follow-up result, gastrointestinal complaints were considerably less or had completely disappeared. In 2 of the 10 (20%) children in which D. fragilis had not disappeared in the follow-up period, the complaints were less or had disappeared. CONCLUSION: Effective treatment of D. fragilis infection in children with longstanding gastrointestinal complaints often resulted in a reduction or disappearance of the complaints.

Abdominal Pain↗
PubMed2004

Children with constipation: what happens to them when they grow up?

Constipation and soiling are common complaints in childhood and often cause distress to the children and their parents. The treatment for these symptoms is oral and sometimes rectal laxatives or behavioural therapy. Until now, there have been few randomized controlled trials to evaluate the efficacy of the different treatment regimens. Moreover, there is a lack of information regarding what happens to these children with constipation when they grow up. A recent long-term follow-up study in The Netherlands showed that 30% of the children followed beyond puberty continued to have severe complaints of constipation such as a low defecation frequency and soiling. Long-lasting support and the development of improved therapeutic regimens are necessary to treat these difficult patients more effectively.

Child↗
PubMed2004

Health related quality of life in disorders of defecation: the Defecation Disorder List.

BACKGROUND: Constipation and encopresis frequently cause problems with respect to emotional wellbeing, and social and family life. Instruments to measure Health Related Quality of Life (HRQoL) in these disorders are not available. METHODS: A disease specific HRQoL instrument, the "Defecation Disorder List" (DDL) for children with constipation or functional non-retentive faecal soiling (FNRFS) was developed using accepted guidelines. For each phase of the process, different samples of patients were used. The final phase of development included 27 children. Reliability was assessed in two ways: internal consistency of domains with Cronbach's alpha, and test-retest reliability with intra-class correlation coefficients (ICC). To assess validity, comparable items and domains were correlated with Tacqol, a generic HRQoL instrument for children (TNO-AZL). RESULTS: In the final phase of the development, 27 children completed the instrument. It consisted of 37 items in four domains. The response rate was 96%. Reliability was good for all domains, with Cronbach's alpha values ranging from 0.61 to 0.76. Measures of test-retest stability were good for all four domains with ICCs ranging from 0.82 to 0.92. Validity based on comparison with the Tacqol instrument was moderate. CONCLUSION: The DDL is promising as a measure of HRQoL in childhood defecation disorders.

Adolescent↗