Authors: Giersiepen K, Lelgemann M, Stuhldreher N, Ronfani L, Husby S, Koletzko S, Korponay-Szabó IR,
Abstract
OBJECTIVE:: The aim of this study was to summarise the evidence from 2004 to September 2009 on the performance of laboratory-based serological and point of care (POC) tests for diagnosing coeliac disease (CD) in children using histology as reference standard. PATIENTS AND METHODS:: We searched MEDLINE and EMBASE for studies reporting on children for tests based on IgA and IgG anti-gliadin (AGA), endomysial (EmA), anti-transglutaminase-2 (TG2), and anti-deamidated gliadin peptides (DGP) antibodies or POC tests. For inclusion, histological analysis of duodenal biopsies and sensitivity and specificity for index tests had to be reported. Data were pooled and summary measures calculated for sensitivity, specificity, positive and negative likelihood ratios (“LR+”, “LR-”), and diagnostic odds ratios (DOR). In case of elevated statistical heterogeneity, studies reaching 90% sensitivity or specificity were reported. RESULTS:: A total of 2510 articles were reviewed; 16 entered meta-analysis, reporting on 3110 patients (1876 with CD, 1234 without CD). For IgA-EmA, sensitivity was ≥90% in 7/11 studies and pooled specificity 98.2%. For IgA-anti-TG2, 11/15 studies yielded sensitivities ≥90% and 13/15 specificities ≥90%. For IgA-DGP, sensitivity ranged between 80.7% and 95.1% (specificity 86.3%-93.1%); for IgG-DGP between 80.1% and 98.6% (specificity 86.0-96.9%). IgA-EmA had the highest pooled DOR (554) and LR+ (31.8) for a laboratory test, followed by IgA-anti-TG2, IgG-DGP, IgA-DGP and IgA-AGA. POC tests showed a pooled sensitivity of 96.4% for IgA-TG2 (specificity 97.7%). CONCLUSIONS:: IgA-EmA and IgA-anti-TG2 tests appear highly accurate to diagnose CD. IgG-anti-DGP tests may help in excluding CD. IgA-AGA and IgA-DGP tests show inferior accuracy. POC tests may achieve high accuracy in the hands of experienced readers, but IgA-anti-TG2/EmA were superior.
PMID: 22266486 [PubMed - as supplied by publisher]
Authors: Borowitz D, Stevens C, Brettman LR, Campion M, Wilschanski M, Thompson H,
Abstract
OBJECTIVES:: Patients with cystic fibrosis (CF) who have exocrine pancreatic insufficiency (EPI) require treatment with pancreatic enzyme replacement therapy (PERT) to maintain adequate nutrition and age-appropriate growth and weight gain. Liprotamase, a nonporcine, highly purified biotechnology-derived PERT, has demonstrated significant efficacy in fat and protein malabsorption in patients with EPI compared to placebo. This study of liprotamase is the first ever long-term trial of a PERT to evaluate safety and nutritional parameters. METHODS:: This phase III 12-month open-label trial assessed the safety, tolerability, and long-term nutritional effects of liprotamase treatment in patients with CF and EPI 7 years and older. All of the patients were required to discontinue their long-term use of porcine PERTs at the time of enrollment. Dosing started at 1 capsule of liprotamase (32,500 US Pharmacopoeia (USP) units crystallized cross-linked lipase, 25,000 USP units crystallized protease, and 3,750 USP units amorphous amylase) per meal or snack; dose could be increased based on protocol-defined parameters. RESULTS:: A total of 215 subjects were enrolled and 214 received at least 1 dose of liprotamase (mean 5.5 capsules per day). During the study period, height, weight, and body mass index z scores and lung function as measured by forced expiratory volume in 1 second were stable. There were no clinically meaningful changes in laboratory tests, including levels of fat-soluble vitamins. Liprotamase was well tolerated without any significant safety concerns. Adverse events, primarily gastrointestinal, led to treatment discontinuation for 36 subjects (16.8%), most within the first 3 months. CONCLUSIONS:: Treatment with a mean of 5.5 capsules of liprotamase per day, during meals and snacks, for up to 12 months was safe, well tolerated, and associated with age-appropriate growth and weight gain or weight maintenance in subjects with CF-related EPI.
PMID: 22266487 [PubMed - as supplied by publisher]
BACKGROUND AND AIMS: : Solitary rectal ulcer syndrome (SRUS) is an uncommon but troublesome and easily misdiagnosed condition of childhood. We have reviewed the presentation and outcome following conservative management of a group of children with SRUS attending a single national paediatric gastrointesinal referral unit.
METHODS: : Eight children were identified with histology-proven SRUS. Chart review was conducted for relevant history and examination at diagnosis. Patients were contacted to assess success of treatment at the time of follow-up.
RESULTS: : Symptoms at presentation included repeated prolonged and ineffectual straining at stool, passage of blood/mucous per rectum, diarrhoea, and constipation. Most children were referred with suspected constipation, diarrhoea, or inflammatory bowel disease. On the basis of retrospective chart review, 7 of 8 children responded well to conservative management (behavioural modification programme involving reduction of time spent straining at defecation). The child failing treatment could not comply with advice because of comorbid autism. Six of the initial responders were available for follow-up. Four were asymptomatic. Two had relapsed and were not compliant with the management programme.
DISCUSSION/CONCLUSIONS: : SRUS can masquerade as more common childhood intestinal conditions such as inflammatory bowel disease or constipation. A biopsy is required for diagnosis, because ulceration may not be apparent at the time of endoscopy. Most patients with SRUS in childhood have a satisfactory outcome using a simple behavioural modification approach. Ongoing follow-up to reinforce behavioural modification is important and may avoid long-term, treatment-resistant disease into adulthood.
Authors: Wali PD, Loveridge-Lenza B, He Z, Horvath K
Abstract
OBJECTIVES: : The fecal pancreatic elastase-1 (FE-1) test is considered a simple, noninvasive, indirect measure of pancreatic function. We aimed to evaluate the performance of the FE-1 test compared with the direct pancreatic function test (PFT) with secretin stimulation in children.
METHODS: : Data of 70 children (6 months-17 years of age) who had both FE-1 test and PFT were analyzed.
RESULTS: : The average FE-1 concentration was 403 ± 142 μg/g. Eleven children had concentrations below 200 μg/g, 23 between 201 to 500 μg/g, and 36 were above 500 μg/g. The average pancreatic elastase activity measured on direct stimulation was 49.1 ± 38.6 μmol · min · ml and 11 children had activity below the established cutoff (10.5 μmol · min · ml). Among the 11 children with pathologic PFT, 7 had normal FE-1, 4 were in the intermediate range (201-500 μg/g), and none were in the low range (<200 μg/g). Among the 59 children with normal direct PFT 11 (19%) had pathologic (<200 μg/g) and 19 (32%) had intermediate FE-1 tests. Twenty-nine children had both normal FE-1 concentration and normal PFT, giving a negative predictive value of 80%. The correlation between pancreatic elastase activity and FE-1 concentration was poor (r = 0.190). The sensitivity of the FE-1 test was found to be 41.7%, whereas the specificity was 49.2%. The positive predictive value of the FE-1 test was only 14%.
CONCLUSIONS: : The FE-1 test is a simple, noninvasive, indirect method; however, ordering physicians should be aware of its limitations. It can give false-positive results and has low sensitivity in children with mild pancreatic insufficiency without cystic fibrosis and in those with isolated pancreatic enzyme deficiencies.
INTRODUCTION:: Esophageal strictures in children are serious and require specialized care. Peptic stricture is most common in our context, caustic and congenital strictures are rare. OBJECTIVE:: This study reports our experience in esophageal endoscopic dilation while specifying the causes of esophageal strictures as well as their response to endoscopic treatment. PATIENTS AND METHODS:: This is cases study over a period of 7 years, during which 60 cases of esophageal stricture were treated with endoscopic dilation by Savary-Gilliard bougies. Our patients were divided into three groups: Group A for peptic strictures (52 patients), Group B for caustic strictures (4 patients) and Group C for strictures subsequent to esophageal atresia surgery (4 patients). RESULTS:: The age of patients is between 10 months and 17 years. Dysphagia was the main symptom and was the major reason for consultation. Two hundred forty-seven dilation sessions were performed, with an average of four sessions per patient ranging from 1 to 15 sessions. The maximum score was observed in group B (50%). Expansion was performed under deep sedation. We had two esophageal perforations: one in group A and one in group B. A good response was obtained in group A (70%) and in group B (50%). The patients in group C still required repeated sessions. CONCLUSION:: Esophagus endoscopic dilation is an effective technique especially in peptic stricture with no need of surgery in some cases. In addition, perforation was rare in this group.
PMID: 22270040 [PubMed - as supplied by publisher]
ABSTRACT: Esophageal strictures are common pediatric diseases and dilations are the most spread procedure, difficult to perform and with possible important complicationsAbout the stricture etiology the authors report a special series of peptic strictures, in patients coming from rural areas with possible delayed GERD diagnosis and treatment. About etiology, it is important to rule out eosinophilic esophagitis (EoE), throught routine hystopathology performed along all the esophagus. Another aspects of this series are: the low incidence of caustic strictures, atypical in North Africa and the important role of the esophageal follow to know the length of stricture. About the endoscopic treatment, the Authors do not report the use of radiological control to verify the correct position of the guide wire (GW) through the stricture in to the stomach. The authors however reported an incidence of perforation 2/247 procedures, not higher than the literature. In our experience the Savary-Gillard bougies are more sure than balloon dilators in consolidated, ‘old’ and fibrotic strictures, like congenital stenosis and old caustic stenosis. We use the hydrostatic dilators only in Epidermolysis Bullosum strictures or in ‘recent’ caustic ingestion.
PMID: 22270041 [PubMed - as supplied by publisher]
Determination of IgG and IgA Antibodies against Native Gliadin is not Helpful for the Diagnosis of Coeliac Disease in Children Up to 2 Years of Age.
J Pediatr Gastroenterol Nutr. 2012 Jan 13;
Authors: Richter T, Bossuyt X, Vermeersch P, Uhlig HH, Stern M, Hauer A, Zimmer KP, Mearin L, Roo JH, Dähnrich C, Mothes T
Abstract
OBJECTIVE:: Assays for antibodies against native gliadin (anti-nGli) are still often assumed to perform better in the diagnosis of coeliac disease (CD) in young children than tests for antibodies to deamidated gliadin (anti-dGli), tissue transglutaminase (anti-tTG) and endomysium (EmA). We compared the performance of assays for anti-nGli, anti-dGli, anti-tTG and EmA in this age group. METHODS:: We investigated retrospectively 184 children (42 coeliacs under normal diet and 142 controls) up to 2 years of age. IgA and IgG-anti-dGli, IgA and IgG-anti-nGli, IgA and IgG-anti-tTG, and IgA-EmA were measured in serum. Areas under receiver-operating characteristics curves (AUCs), sensitivities, specificities, positive (PPV) and negative predictive values (NPV), positive and negative likelihood ratios (PLR and NLR), as well as diagnostic odds ratios (DOR) were calculated. RESULTS:: From all tests investigated, only assays for IgG-anti-dGli, IgA-anti-tTG and IgA-EmA had high specificity (≥ 0.96) connected with high sensitivity (≥ 0.86), with high positive predictive values (≥ 0.52 and ≥ 0.69 at pre-test probabilities of 0.05 and 0.1, respectively), and with high negative predictive values (≥ 0.99 and ≥ 0.98 at pre-test probabilities of 0.05 and 0.1, respectively). These assays also showed high PLR (≥ 24) at low NLR (≤ 0.15) and high DOR (≥ 136). CONCLUSIONS:: Our results do not support the use of assays of anti-nGli to diagnose CD in young children. IgA-anti-tTG, IgA-EmA and IgG-anti-dGli perform better than anti-nGli.
PMID: 22249805 [PubMed - as supplied by publisher]
Evaluation of risk factors for bleeding after liver biopsy in children.
J Pediatr Gastroenterol Nutr. 2012 Jan 13;
Authors: Westheim BH, Ostensen AB, Aagenæs I, Sanengen T, Almaas R
Abstract
OBJECTIVES:: Acetylsalicylic acid is used in liver transplanted children to prevent thrombosis of the hepatic artery. We evaluated whether acetylsalicylic acid and other risk factors are associated with bleeding after percutaneous liver biopsy. MATERIALS AND METHODS:: Medical charts, laboratory results, imaging studies, and anesthesia charts of 275 ultrasound guided liver biopsy procedures in 190 children were reviewed. 178 of the biopsies were performed on native livers and 97 on transplanted livers. RESULTS:: Three major and 28 minor bleedings were found. Mortality rate was 0. Acetylsalicylic acid had been given the last 5 days before 55 of the biopsy procedures and no increased risk of bleeding was found (odds ratio 0.96 (0.37-2.26), p = 1.00). Low-molecular-weight heparin and biopsies from focal lesions were risk factors for bleeding complications. Acute liver failure was associated with increased risk for major complications (odds ratio 26.1 (3.3-205, p = 0.01)) and was a risk factor for major bleedings. Postbiopsy ultrasound the day after the procedure (n = 266 (96% of 275 biopsies)) revealed minor bleedings after 7.1% of the biopsies and after 2.6% ultrasound revealed unsuspected bleedings, but none of these required intervention. CONCLUSIONS:: Ultrasound-guided liver biopsy in children is a procedure with a low rate of major complications and a high rate of minor bleedings not requiring intervention. Treatment with low-dose acetylsalicylic acid did not increase bleeding incidence or total complication rate. Low-molecular-weight heparin and biopsies from focal lesions were risk factors for bleeding complications. Routine ultrasound the day after the procedure did not change handling of the patients.
PMID: 22249806 [PubMed - as supplied by publisher]
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