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Biomedical subjects

J F Mougenot

Publications and source records attributed to J F Mougenot.

At least 19 recordsLinked to original sources

Ultrasonographic assessment of inflammatory bowel disease in children: comparison with ileocolonoscopy.

OBJECTIVES: To determine the feasibility and value of transabdominal ultrasonography of the terminal ileum and colon of children with inflammatory bowel disease (IBD) and to compare the findings with those of ileocolonoscopy. STUDY DESIGN: Thirty-eight patients ranging in age from 4 to 18 years who underwent ileocolonoscopy for management of IBD or for diagnosis were studied prospectively. Twenty-one patients had Crohn disease, nine had ulcerative colitis, and eight served as control subjects. Transabdominal ultrasonography was performed on the day before ileocolonoscopy. Ultrasonographic findings were compared with the results of ileocolonoscopy, used as the reference method. RESULTS: Peristalsis was recorded in all segments of the control subjects; the thickness of the terminal ileum was always less than 2.5 mm, and that of the large bowel, 2 mm or less. In the two patient subgroups, the thickness range of affected ileal and colonic segments was similar, but values were significantly different from those of the control subjects (chi-square test, p <0.0001). The overall sensitivity of the method was 88%, and the specificity, 93%. CONCLUSION: Transabdominal ultrasonography should prove to be a useful clinical and investigational technique, although further studies are needed to assess its value in the treatment of children with IBD.

Adolescent

Histopathology and microbiology of isolated rectal bleeding in neonates: the so-called 'ecchymotic colitis'.

Rectal bleeding in neonates is an alarming event which suggests a possible necrotizing enterocolitis (NEC) but is usually the only symptom of an unexplained colitis characterized endoscopically by ecchymotic mucosal lesions, the so-called 'ecchymotic colitis' (EC). We studied histologically and bacteriologically 18 infants (mean age: 18 days) presenting with rectal bleeding by systematic rectosigmoidoscopy and intestinal biopsies. The 18 infants were hospitalized. Prematurity was found in seven cases and an underlying condition in 14 cases (respiratory distress: six cases; infection: six cases; surgery: two cases). Histology showed a mild to moderate inflammation (10/12) of the mucosa with a prevalence of polymorphonuclear cells (8/10), frequent focal haemorrhages (11/12) and foci of pneumatosis (4/12). Numerous bacteria were seen in the mucus layer focally forming large clusters. Cultures of intestinal biopsies yielded exclusively Enterobacteriaceae species: Escherichia coli (seven cases), Klebsiella spp. (seven cases), and Enterobacter cloacae (three cases); four cases were sterile. Our study demonstrates that neonatal bleeding is associated with endoscopic and histological 'ecchymotic colitis' lesions and with a peculiar microbial flora of EBC strains. EC and necrotizing enterocolitis share similar features raising the question of the link between the two syndromes.

Biopsy

Increased TIA1-expressing intraepithelial lymphocytes in cow's milk protein intolerance.

BACKGROUND: Although a high level of intraepithelial lymphocytes (IELs) has been demonstrated in intestinal biopsies from children with cow's milk protein intolerance (CMPI), the properties of IELs in food-sensitive enteropathies remain unclear. In the present study, we analyzed the cytotoxic potential of IELs in CMPI, using a monoclonal antibody directed against the cytotoxic granule-associated protein TIA1. METHODS: The study included 18 duodenal biopsies from 10 children previously diagnosed with CMPI and on a cow's milk-free diet of various duration. Six normal duodenal biopsies from children free of food intolerance served as controls. Immunostaining of formalin-fixed tissues was used to determine in the intraepithelial compartment (1) the number of TIA1-expressing cells per 100 epithelial cells, (2) the number of IELs per 100 epithelial cells, (3) the ratio of TIA1-expressing IELs (TIA1/IEL ratio). RESULTS: In CMPI, the number of IELs and TIA1-positive cells, as well as the TIA1/IEL ratio was significantly increased compared with controls. Moreover, a negative correlation between the TIA1/IEL ratio and the duration of the diet was observed. CONCLUSIONS: These results suggest that the recruitment of IELs with cytotoxic potential is increased in CMPI, and that IEL-mediated cytotoxicity could be involved in the pathogenesis of the disease.

Antibodies, Monoclonal

[Contribution of endosonography to the diagnosis and follow-up of pediatric gastric neurofibroma revealing von Recklinghausen's disease].

BACKGROUND: Gastrointestinal involvement in von Recklinghausen's disease (RD) is rare during childhood; its symptoms are late and its prognosis is poor, related to local recurrence and risk of malignant transformation. CASE REPORT: A 13 year-old boy was admitted for hematemesis revealing gastric ulcer. A second episode of hematemesis led to identify a sessile gastric tumor in this patient having numerous skin café-au-lait spots. Recurrent bleeding required laparotomy that showed diffuse infiltration into the anterior gastric wall: histological examination of the excised piece showed characteristic features of neurofibromatosis. The patient was not compliant to the endosonographic survey so that a symptomatic relapse led to total gastrectomy: histological examination did not show malignant transformation. CONCLUSION: Endoscopy is a major tool for identifying gastrointestinal localization of RD but endosonography is necessary to precise the extent of the tumor.

Adolescent

Reevaluation of the relative risk for susceptibility to celiac disease of HLA-DRB1, -DQA1, -DQB1, -DPB1, and -TAP2 alleles in a French population.

In a population of 46 children with CD recruited in the Paris area of France, an excess of DRB1*03 and DRB1*07 alleles and of DR3/DR7, DR3/DR3 and DR11(or 12)/DR7 phenotypes was found (RRs of 6.3, 9.3, 24.6, 15, and 15.1, respectively), which is reminiscent of the markers of susceptibility observed in southern rather than in northern European celiac patients. More importantly, the highest association with CD was not found in individuals expressing the DQA1*0501-DQB1*0201 heterodimer in single dosage (RR = 24.9) or in homozygous state, but in people co-expressing one copy of DQA1*0501-DQB1*0201 on one haplotype and a second copy of DQB1*0201 on the second haplotype (RR = 35.7). This suggests that in our population either DQB1*0201 or a gene closely linked to DQB1*0201 influences the susceptibility to CD conferred by the DQA1*0501-DQB1*0201 heterodimer. Significant positive or negative RRs conferred by some TAP2 or DPB1 alleles were found. However, they were moderate compared to the RR conferred by the expression of a second copy of DQB1*0201. Moreover, they were no longer significant when patients were compared with HLA-DR matched controls. This suggests that associations of CD with TAP2 and DPB1 alleles are secondary to linkage disequilibria and argues against the contribution of these alleles in resistance and/or susceptibility to CD. Thus the "raison d'être" of a "DQB1*0201 second haplotype effect" in susceptibility to CD remains to be elucidated.

ATP Binding Cassette Transporter, Subfamily B, Mem

[Esophageal dilatation in pediatrics: study of 33 patients].

BACKGROUND: Esophageal dilatation is usually regarded as an effective therapy in a majority of esophageal stenosis in childhood. However, the limited number of pediatric data does not allow definite conclusions on indications and complications of such a procedure. PATIENTS AND METHODS: The files of 33 children whose esophageal stenosis had been treated by dilatation by the same operator between 1983 and 1992 were retrospectively reviewed. The structure mechanisms were: group 1: repair of esophageal atresia (n = 9), group 2: caustic esophagitis (n = 6), group 3: peptic esophagitis (n = 12), group 4: unclassified structures (congenital esophageal stenosis, achalasia) (n = 6). The dilatations were performed under general anesthesia, and the dilatator guide was introduced under endoscopic control. Two methods were used: Savary esophageal bougies and balloon dilatation. A thoracic X-ray was systematically performed after each dilatation. RESULTS: One hundred and fourteen dilatations (3.5 dilatations/child) were performed (range: 1-32 dilatations). Twenty-five of the 33 children (76%) were dramatically improved after mechanical dilatation. Esophageal dilatation was unsuccessful in the eight other patients, seven of them requiring a surgical repair. Complications occurred in 3.4% of the dilatations: one esophageal perforation, one pneumomediastinum and two cardiac arrests (one of vagal origin and 1 after accidental extubation). All patients survived. Efficacy, duration of dilatation and complication rates were not similar in the four groups. CONCLUSIONS: Esophageal dilatation should be considered as a simple and effective procedure when strict security rules are respected by a trained operator.

Adolescent

[Adenocarcinoma on villous tumor of the rectum in an adolescent].

CASE REPORT--A 15-year 6-month-old boy suffered from isolated recurrent rectal bleeding. Rectoscopy and colonoscopy allowed to identify and to excise one large size villous polyp, which was the site of dysplasia and liberkühn adenocarcinoma. Transrectal ultrasonography showed thickened mucosa and submucosa and suspect adenopathy. Proctectomy, ganglionic curage and coloanal anastomosis were then performed; one of the excised adenopathies was metastatic. The patient died twelve hours after surgery from an unexplained cardiovascular collapse. CONCLUSIONS--Unspecific symptoms of colorectal cancer explain that its diagnosis is often delayed. Prognosis is poor related to the advanced stages at diagnosis and mucinous adenocarcinomas. Better knowledge of the clinical presentations and of high risk situations as polyposis, ulcerative colitis, hereditary non polyposis colorectal cancer could improve the prognosis.

Adenocarcinoma

Intractable diarrhea of infancy with epithelial and basement membrane abnormalities.

We describe a form of intractable diarrhea in six children (four girls) with similar clinical histories and identical histopathologic features. The children had watery diarrhea of neonatal onset requiring total parenteral nutrition. Two had siblings who had died of diarrhea in the first year of life; two others are sisters. Repeated duodenal or jejunal biopsies revealed villous atrophy with normal or hyperplastic and regenerative cryptae, normal cellularity of the lamina mesenterii propria, and no signs of T-cell activation. The main histologic features are epithelial dysplasia with focal crowding and disorganization of the surface enterocytes, pseudocystic formation of the glands, and abnormal regenerative cryptae. The basement membrane components were studied with polyclonal antibodies on frozen specimens, and were compared with biopsy specimens from patients with celiac disease or autoimmune enteropathy. Relative to the control subjects, there was faint and irregular deposition of laminin at the epithelium-lamina mesenterii propria interface, whereas deposits of heparan sulfate proteoglycan were large and lamellar. The primary or secondary nature of these modifications of the basement membrane remains to be determined, but the modifications might be related to epithelial abnormalities and to the severity of this neonatal diarrhea, which resisted all treatment and necessitated permanent total parenteral nutrition.

Basement Membrane

Detection of Mycobacterium paratuberculosis by polymerase chain reaction in children with Crohn's disease.

Mycobacterium paratuberculosis, the causative agent of Johne's disease (a chronic enteritis in ruminants), has been suspected to be involved in Crohn's disease. In this study, polymerase chain reaction was used to detect the presence of IS900 DNA sequences specific to M. paratuberculosis genomes in biopsies and surgical resections from 53 children with various gastrointestinal diseases and disorders. IS900 sequences were found in 13 of 18 samples from patients with Crohn's disease (72%; P < .01 vs. samples from patients without Crohn's disease), in 1 of 5 with ulcerative colitis, in 2 of 6 with severe unclassified colitis, and in 7 of 24 with other gastrointestinal illnesses. These results appear to support the hypothesis that M. paratuberculosis is involved in the pathogenesis of Crohn's disease.

Child, Preschool

[A rare cause of rectal hemorrhage in children: rectal angiodysplasia].

BACKGROUND: Parietal vascular malformations of the intestinal tract are rare and their diagnosis is often difficult. CASE REPORT: A 2 year-old boy had suffered from intermittent rectal bleeding since the age of one year. Endoscopic examination showed unspecific congestive changes and biopsy showed features of chronic inflammatory changes in the rectum. Barium enema was normal. The patient was operated on, but surgery failed to find any cause for these hemorrhages. Rectal bleedings recurred, some of them resulting in severe anemia. Two further endoscopic examination were grossly negative as was inferior mesenteric arteriography. Spontaneous rectal prolapsus occurred when the boy was 4 years old. This allowed resection of all the submucosal veins of the anal canal; it was followed by complete cessation of rectal bleeding with a follow-up of 36 months. Histological examination showed several ectasias of the capillaries and veins in the submucosa and muscular layers. CONCLUSION: Angiectases can occur in a small part of the intestinal tract, and can escape detection by repeated specialized investigation.

Arteriovenous Malformations

[Contribution of genetic typing for the diagnosis of familial adenomatous polyposis in pediatrics].

BACKGROUND: The gene responsible for familial adenomatous polyposis, (APC), has been recently cloned and genetic map with several polymorphic markers has been established. POPULATION AND METHODS: Blood samples (20 ml) were taken from 34 subjects belonging to four families at risk for familial adenomatous polyposis. Nineteen of these 34, less than 20 years old, had one parent having polyposis or dead because of it. Polyposis was diagnosed, in ten of these 19 by endoscopy. Genomic DNA was extracted from peripheral leukocytes and Southern blot analyses were performed in each family, using RFLPs on both sides of the APC locus. RESULTS: DNA analysis identified normal and mutant haplotypes at the APC locus in each family. It was thus possible to follow the segregation of mutant alleles. These results were compared with the anamnestic and endoscopic data. Bearing in mind the risk of recombination when using extragenic markers, RFLPs allowed early diagnosis of APC in pre and/or asymptomatic patients. CONCLUSIONS: Genetic analysis can be used to diagnose APC in affected families, provided the risk of recombination is taken into account. Intragenic microsatellites markers will soon be available. These will provide more information on the APC gene, and hence direct molecular diagnosis of APC.

Adenomatous Polyposis Coli

[Multicenter study of sodium alginate in the treatment of regurgitation in infants].

An open multicenter study was performed to assess the efficacy and safety of alginic acid in two different dosages in 76 pediatric patients with gastroesophageal reflux confirmed by pH monitoring. Among the 69 patients in whom endoscopy was carried out before treatment, 18 had erythematous esophagitis and 5 had erosive esophagitis. Irrespective of the dosage used, the frequency of regurgitation and vomiting decreased significantly (p < 0.00001 and p = 0.01, respectively). Clinical and biochemical tolerance were outstanding and no adverse effects were recorded. On the basis of these data, the recommended dosage is 1 to 2 ml/kg/day in divided doses after meals.

Alginates

[Severe Crohn's disease in children. Retrospective study of 38 cases].

Thirty-eight children presenting with severe Crohn's disease (CD) were studied retrospectively over a mean period of 6 years. The severity of CD was estimated according to an activity score. This was initially 66 +/- 19 reaching 100 in 16% of cases. Initial involvement included ileon and large intestine only (n = 23), large intestine (n = 11) and ileon only (n = 4), with upper GI tract or perineal involvement in 32 and 75% of cases respectively. The treatments consisted of corticosteroids (n = 34), azathioprine (n = 11), continuous enteral feeding (CEF) (n = 25), parenteral nutrition (PN) (n = 30). Mean recurrence rates per patient and per year were 0.6 in the groups including ileon and large intestine or the large intestine only and 0.3 in the ileal group. A surgical resection was performed in 20 cases with a mean rate of reoperation of 40% at 5 years. Reoperations were necessary only in the group involving the ileon and large intestine. Mean weight catch-up in 3 months and height growth speed were 6 kg and 1.7 to 3.1 cm/year under CED and/or PN. After a 6 year follow-up the activity index was decreased by 70% with a quality of life considered to be good in 60% of cases.

Child