Cécile Talbotec

Université René Descartes - Paris 5, Lutetia Parisorum, Île-de-France, France

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Publications (54)162.47 Total impact

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    ABSTRACT: Objectives: To describe the indications for home parenteral nutrition (HPN) in children with primary digestive diseases (PDD) and to identify factors associated with weaning off. Methods: All children initially discharged on HPN between 1/1/2000 and 31/12/2009 for chronic intestinal failure (IF) were included. The associations between clinical factors and weaning off of HPN were assessed using a multivariable Cox regression model. Results: Among the 151 children (boys=58%) included in this study, 98 (65%) presented with short bowel syndrome (SBS), 17 (11%) with digestive neuromuscular disorders, 14 (9%) with mucosal diseases, 13 (9%) with inflammatory bowel disease (IBD) and 9 (6%) with other PDD. The probability of survival was approximately 100%. At the end of the follow-up, the probability for weaning off of HPN was 0.73 [95% confidence interval (CI): 0.54-0.84] but varied according to the underlying cause of IF (for example, SBS and IBD had a better prognosis). The median time until weaning off was 21 months [95% CI: 18-38 months]. Unfavourable prognostic factors for weaning off of HPN included a bowel remnant of <40 cm, the presence of <50% of the colon, and daily lipid intakes >1.5 (g/kg•d). Underlying disease was also associated with weaning off. Conclusion: HPN is a safe therapeutic option for children with chronic IF requiring long-term nutritional management. Prognostic factors for weaning off of HPN were identified and highlight the relevance of SBS anatomy and PN caloric intake. The outcome of children on HPN was primarily dependent on the underlying disease.
    Journal of pediatric gastroenterology and nutrition 09/2015; DOI:10.1097/MPG.0000000000000980 · 2.63 Impact Factor

  • Fabienne Charbit-Henrion · Cecile Talbotec · Virginie Colomb ·

    Journal of Pediatric Gastroenterology and Nutrition 03/2015; 60(3):e26. DOI:10.1097/MPG.0000000000000646 · 2.63 Impact Factor
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    ABSTRACT: Background Eosinophilic esophagitis (EoE) is a clinicopathologic disease that presents with a massive infiltration of the esophagus by eosinophils triggered by food antigen(s). Objective To determine the impact of dietary therapy on nutritional parameters in patients who present with EoE. Methods A convenience retrospective study analyzed patients with EoE after a 2-month dietary therapy (6-food elimination diet, avoidance of the 6 most common allergenic foods, plus avoidance of those eliciting positive skin testing, plus amino-acid formula as replacement for dairy products). Pre- and postdiet allergic and nutritional status were evaluated. Results Of 111 eligible patients, 59 patients, with a median age of 77.7 months (range, 9-189 months) were enrolled. Dietary therapy significantly increased the return to normal endoscopic appearance (47.4%, P < .0009) and led to complete remission (<5 eosinophils/esophageal HPF and disappearance of symptoms) in 59.3%. All symptoms improved, digestive (98.3%), cutaneous (80%), and respiratory (92.8%). The prediet median weight-for-height (WFH) z score was −0.75 (−3.00 to 5.69), and the postdiet WFH did not significantly differ, −0.51 (−3.09 to 5.00). The prediet WFH z score was less than −2 (moderate malnutrition) in 10.1%. Postdiet blood eosinophils counts decreased in absolute numbers and in counts ≥500 × 106/L (P < .0001). Evaluation after 1 year of progressive reintroduction of eliminated foods was available in 33 children: the median WFH z score did not significantly improve, from −0.89 (range, −3.00 to 0.67) at enrollment to −0.59 (range, −3.66 to 2.24). Conclusion The nutritional status of children with EoE was mildly affected and not worsened by the 2-month dietary therapy.
    10/2014; 2(5):587–593. DOI:10.1016/j.jaip.2014.05.012

  • Journal of Crohn s and Colitis 09/2014; 8:S398. DOI:10.1016/S1873-9946(14)50018-9 · 6.23 Impact Factor
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    ABSTRACT: BACKGROUND: Inflammatory bowel disease (IBD) is one of the most common chronic gastrointestinal diseases, but the underlying molecular mechanisms remain largely unknown. Studies of monogenic diseases can provide insight into the pathogenesis of IBD. OBJECTIVE:We thought to determine the underlying molecular causes of IBD occurring in 2 unrelated families in association with an immune deficiency. METHODS: We performed genetic linkage analysis and candidate gene sequencing on 13 patients from a large consanguineous family affected by early-onset IBD, progressive immune deficiency, and, in some cases, autoimmunity and alopecia, a condition we named enteropathy-lymphocytopenia-alopecia. The candidate gene was also sequenced in an unrelated patient with a similar phenotype. We performed histologic analysis of patients' intestinal biopsy specimens and carried out functional assays on PBMCs. Gut organoids derived from a patient's biopsy specimen were analyzed. RESULTS: We identified biallelic missense mutations in tetratricopeptide repeat domain 7A (TTC7A) in all patients from both families. The resulting TTC7A depletion modified the proliferation, adhesion, and migratory capacities of lymphocytes through inappropriate activation of the RhoA signaling pathway. Normal function was restored by wild-type TTC7A expression or addition of a RhoA kinase inhibitor. The growth and polarity of gut epithelial organoids were also found to be dependent on the RhoA signaling pathway. CONCLUSIONS: We show that TTC7A regulates the actin cytoskeleton dynamics in lymphocytes through the RhoA signaling pathway and is required in both lymphocytes and epithelial cells for maintaining equilibrium between cell proliferation, migration, polarization, and cell death. Our study highlights variability in the phenotypic expression resulting from TTC7A deficiency and outlines that impairment of both epithelial cells and lymphocytes cooperatively causes IBD.
    Journal of Allergy and Clinical Immunology 08/2014; DOI:10.1016/j.jaci.2014.07.019 · 11.48 Impact Factor
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    ABSTRACT: Objective: Anastomotic ulceration (AU) is a rare complication after intestinal resection and anastomosis, described mostly in children. The main symptom is occult bleeding, leading to iron-deficiency anemia, which is life threatening. Methods: The present survey reports a series of patients with AU after intestinal resection in infancy, focusing on predictive factors, medical and surgical treatment options, and long-term outcomes. Eleven patients (7 boys) born between 1983 and 2005 with AU after an intestinal resection and anastomosis in infancy were included in this retrospective review. Results: The diagnosis of AU was often delayed for several years. No predictive factor (including the primary disease, the length of the remnant bowel, and the loss of the ileocaecal valve) could be identified. Numerous treatment options, including antibiotics and anti-inflammatory drugs, proved to be ineffective to induce prolonged remission. Even after surgical resection, relapses were observed in 5/7 children. Conclusions: The mechanism leading to AU remains unknown. Contrary to previous reports with limited follow-up, no medical or surgical treatment could prevent recurrences. Because relapses may occur several years after treatment, long-term follow-up is needed.
    Journal of Pediatric Gastroenterology and Nutrition 06/2014; 59(4). DOI:10.1097/MPG.0000000000000472 · 2.63 Impact Factor
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    ABSTRACT: Background and aims Chronic intestinal failure (CIF) requires long term parenteral nutrition (PN) and, in some patients, intestinal transplantation (ITx). Indications and timing for ITx remain poorly defined. In the present study we aimed to analyse causes and outcome of children with CIF. Methods 118 consecutive patients referred to our institution were assessed by a multidisciplinary team and four different categories were defined retrospectively based on their clinical course: Group 1: patients with reversible intestinal failure; group 2: patients unsuitable for ITx, group 3: patients listed for ITx; group 4 : patients stable under PN. Analysis involved comparison between groups for nutritional status, central venous catheter (CVC) related complications, liver disease, and outcome after transplantation by using non parametric tests, Mann-Whitney tests, Kruskal-Wallis, Wilcoxon signed rank tests and chi square distribution for percentage. Results 118 children (72 boys) with a median age of 15 months at referral (2 months - 16 years) were assessed. Etiology of IF was short bowel syndrome [n=47], intractable diarrhea of infancy [n=37], total intestinal aganglionosis [n=18], and chronic intestinal pseudoobstruction [n=17]. Most patients (89.8%) were totally PN dependent, with 48 children (40.7%) on home-PN prior to admission. Nutritional status was poor with a median body weight at -1.5 z-score (ranges: -5 to +2,5) and median length at -2,0 z-score (ranges: -5.5 to +2.3). The mean number of CVC inserted per patient was 5.2 (range 1 - 20) and the mean number of CRS per patient was 5.5 (median: 5; range 0 – 12) Fifty-five patients (46.6%) had thrombosis of ≥ 2 main venous axis. At admission 34.7% of patients had elevated bilirubin (≥ 50 μmol/l), and 19.5% had platelets < 100 000/ml, and 15 % had both. Liver biopsy performed in 79 children was normal (n=4), or showed F1 or F2 fibrosis (n= 29), bridging fibrosis F3 (n=20), or cirrhosis (n=26). Group 1 included 10 children finally weaned from PN (7-years survival: 100%). Group 2 included 12 children with severe liver disease and associated disorders unsuitable for transplantation (7-years survival: 16.6%). Group 3 included 66 patients (56%) who were listed for small bowel or liver-small bowel transplantation, 62/66 have been transplanted (7years survival: 74.6%). Factors influencing outcome after liver-ITx were body weight (p<.004), length (p<.001), pre-Tx bilirubin plasma level (p<.001) and thrombosis (p<.01) for isolated ITx, Group 4 included 30 children (25.4%) with irreversible IF considered as potential candidates for isolated ITx. Four children were lost from follow up and 3 died within 2 years (survival 88,5%). Among potential candidates, the following parameters improved significantly during the first 12 months of follow up: Body weight (p.0001), length (p<.0001) and bilirubin (p<.0001). Conclusions many patients had a poor nutritional status with severe complications especially liver disease. PN related complications were the most relevant indication for ITx, but also a negative predictor for outcome. Early patient referral for Tx-assessment might help to identify and separate children with irreversible IF from children with transient IF or uncomplicated long-term PN, allowing to adapt a patient-based treatment strategy including or not ITx.
    Clinical Nutrition 04/2014; 34(3). DOI:10.1016/j.clnu.2014.04.015 · 4.48 Impact Factor

  • Cahiers de Nutrition et de Diététique 12/2013; 48:S37. DOI:10.1016/S0007-9960(13)70326-2
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    ABSTRACT: Early-onset inflammatory bowel disease starting within the first months of life could be due to a particular genetic defect. We set up the GENetically determined ImmUne-mediated enteropathieS (GENIUS) network and collected infants with a proven defect of the IL10 axis for accurate phenotyping of disease presentation and evolution. Ten patients with early-onset inflammatory bowel disease with confirmed mutations in IL10, IL10RA, or IL10RB genes were characterized on clinical, endoscopic-histological, immunobiological, and radiological findings. Functional assays to confirm defective responses to IL10 were performed on peripheral blood mononuclear cells. A functional defect in IL10 signaling was confirmed in all IL10R patients tested. Disease started with severe diarrhea within the first 12 weeks in all patients. All infants showed Crohn's disease-like ulcerations limited to the colon with marked perianal inflammation (fissures, abscess, and fistula); disease progression to the small bowel occurred in only 1 patient. Four of the 10 patients had granulomata on histology, and all patients showed Crohn's disease-like mesenteric infiltration on imaging. Disease pattern was indistinguishable between IL10R alpha or beta chain or IL10 defects; autoimmunity was not observed. Mutations in IL10 were more frequently associated with bacterial and viral infections. Patients responded partially to treatment with steroids or anti-tumor necrosis factor drugs, whereas hematopoietic stem cell transplantation proved efficacious. The importance of the IL10 pathway within the colonic mucosa is highlighted by the development of severe colitis within a few weeks in infants with mutations in IL10, IL10RA, or IL10RB. Immunosuppression failed to correct the defect in this pathway, which seems to be a key to controlling inflammation in the colon.
    Inflammatory Bowel Diseases 11/2013; 19(13). DOI:10.1097/01.MIB.0000435439.22484.d3 · 4.46 Impact Factor
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    ABSTRACT: Congenital tufting enteropathy (CTE) is a rare and severe enteropathy recently ascribed to mutations in the epcam gene. Here we establish SPINT2, previously ascribed to congenital sodium diarrhea, as a second gene associated with CTE and report molecular and immunohistochemistry data in 57 CTE patients. Inclusion criteria were early onset diarrhea and intestinal insufficiency with the typical histological CTE abnormalities. The clinical phenotype was registered, the entire coding regions of epcam and SPINT2 sequenced, and immunostaining of EpCAM and SPINT2 performed on intestinal biopsies. An epcam mutation was involved in 41 patients (73 %) who mainly displayed isolated digestive symptoms. Mutations severely affected gene expression since the EpCAM signal on intestinal tissues was either undetectable or low and irregular. Twelve other patients (21 %) carried mutations in SPINT2, and were phenotypically characterized by systematic association with keratitis (p < 10(-4)) and, for half of them, with choanal atresia (p < 10(-4)). Dependency on parenteral nutrition (PN) was comparable in patients with epcam or SPINT2 mutations, but the frequent epcam mutation c.556-14A>G (abnormal splicing) was significantly associated with a better outcome (p = 0.032) with milder PN dependency to weaning in some cases. Finally, four patients (7 %) with isolated digestive symptoms had no detectable epcam or SPINT2 mutation. Two candidate genes, Elf3 and Claudin7, were excluded from this population. Our study allows us to separate CTE patients into at least three genetic classes, each with specific phenotypes. The genetics approach raises the question of the distinction between two congenital enteropathies. Our findings should help improve the diagnosis of CTE, guide toward strategies of long-term PN management, and limit indications for intestinal transplantation to life-threatening PN complications.
    Human Genetics 10/2013; 133(3). DOI:10.1007/s00439-013-1380-6 · 4.82 Impact Factor
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    ABSTRACT: Mucormycosis, an emerging fungal infection in solid organ transplant patients, is mostly located in rhino-orbito-cerebral, pulmonary, and cutaneous areas, or disseminated with poor prognosis. A 4-year-old girl with chronic intestinal pseudo-obstruction syndrome underwent a modified multivisceral transplantation, including half of the stomach, the duodeno-pancreas, the small bowel, and the right colon. On postoperative day 5, a digestive perforation was suspected. Surgical exploration found a small necrotic area on the native stomach, which was externally drained. The next day, massive gastric bleeding occurred. During the emergency laparotomy, 2 hemorrhagic ulcers were found and resected from the transplanted stomach. Pathology and fungal culture showed mucormycosis caused by Lichtheimia (formerly Absidia) ramosa in both the transplanted and native stomach. High-dose intravenous liposomal amphotericin B was immediately started. No other site of fungal infection was found. The child recovered, and 3 years after transplantation, is alive and well, off parenteral nutrition. The originality of this case is the very early presentation after transplantation, the unusual site, and the complete recovery after rapid medico-surgical management. The origin of the fungus and treatment are discussed.
    Transplant Infectious Disease 09/2013; 15(6). DOI:10.1111/tid.12147 · 2.06 Impact Factor
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    ABSTRACT: BACKGROUND: Small bowel transplantation has now become a recognized treatment of irreversible, permanent, and subtotal intestinal failure. OBJECTIVE: The aim of this study was to assess intestinal absorption at the time of weaning from parenteral nutrition in a series of children after intestinal transplantation. DESIGN: Twenty-four children (age range: 14-115 mo) received intestinal transplantation, together with the liver in 6 children and the colon in 16 children. Parenteral nutrition was slowly tapered while increasing enteral tube feeding. The absorption rate was measured from a 3-d stool balance analysis performed a few days after the child had weaned from parenteral nutrition to exclusive enteral tube feeding. Results were analyzed according to the resting energy expenditure (REE; Schofield formula).Results: All children were weaned from parenteral nutrition between 31 and 85 d posttransplantation. Median intakes were as follows: energy, 107 kcal ⋅ kg(-1) ⋅ d(-1) (range: 79-168 kcal ⋅ kg(-1) ⋅ d(-1)); lipids, 39 kcal ⋅ kg(-1) ⋅ d(-1) (range: 20-70 kcal ⋅ kg(-1) ⋅ d(-1)); and nitrogen, 17 kcal ⋅ kg(-1) ⋅ d(-1) (range: 11-27 kcal ⋅ kg(-1) ⋅ d(-1)). Median daily stool output was 998 mL/d (range: 220-2025 mL/d). Median absorption rates were 88% (range: 75-96%) for energy, 82% (range: 55-98%) for lipids, and 77% (range: 61-88%) for nitrogen. The ratios for ingested energy to REE and absorbed energy to REE were 2.2 (range: 1.6-3.6) and 1.8 (range: 1.3-3.3), respectively.Conclusion: These data indicate a suboptimal intestinal graft absorption capacity with fat malabsorption, which necessitates energy intakes of at least twice the REE.
    American Journal of Clinical Nutrition 02/2013; 97(4). DOI:10.3945/ajcn.112.050799 · 6.77 Impact Factor
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    ABSTRACT: The pathogenesis of inflammatory bowel disease (IBD) is multifactorial, with some patients presenting additional autoimmune symptoms. Inflammatory colitis associated with autoimmune (AI) liver disease appears to have clinical features different from those of "classical" ulcerative colitis (CUC). The aim of this study was to describe these features, in order to differentiate a subgroup of colitis associated with autoimmunity (CAI) from CUC. Twenty-eight consecutive children with inflammatory colitis associated with primary sclerosing cholangitis (PSC), celiac disease, or AI hepatitis were compared with a matched control group of 27 children with isolated UC. Clinical course, histology, as well as inflammatory profile in the colonic mucosa based on real-time polymerase chain reaction (PCR) were analyzed. In CAI the main digestive symptoms at disease onset were abdominal pain (12/28) and bloody strings in the stool (12/28), along with a high prevalence of autoimmune diseases in relatives, as compared with bloody diarrhea in the CUC group (26/27). At diagnosis, pancolitis was seen in 18/28 CAI patients compared with 8/27 in UC. In CAI, the pathological findings were different from CUC: 1) major lesions predominantly located in the right colon; 2) pseudo-villous appearance of the mucosa, and strong infiltration with eosinophils; 3) mild glandular lesions; and 4) differing inflammatory infiltrate with reduced FOXP3, interleukin (IL)-2, and thymic stromal lymphopoietin (TSLP) levels. Evolution in CAI was less aggressive, requiring less corticosteroids/immunomodulators. Precise clinical, histological, and molecular analyses reveal marked differences between patients with CUC and those with associated AI phenomena, supporting the hypothesis of a distinct AI presentation of IBD. (Inflamm Bowel Dis 2012).
    Inflammatory Bowel Diseases 10/2012; 18(10):1809-17. DOI:10.1002/ibd.22864 · 4.46 Impact Factor

  • Journal of pediatric gastroenterology and nutrition 02/2012; 54(5):699. DOI:10.1097/MPG.0b013e31824f8790 · 2.63 Impact Factor
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    ABSTRACT: Thiopurines are considered first-line immunomodulators for the prevention of relapse in moderate to severe pediatric Crohn's disease (CD). Early introduction of thiopurines was shown in a pediatric trial to maintain steroid-free remission in 90% of patients for 18 months. In the present study we analyzed the tolerance and efficacy of azathioprine (AZA) to maintain remission in a homogenous single-center observational cohort of children with CD. In all, 105 pediatric CD patients (male/female 68/37) were retrospectively evaluated for the efficacy of AZA (doses 1.4-4 mg/kg) to maintain remission at 6, 12, 18, and 24 months of follow-up. Overall, 93 children were included with active disease (pediatric Crohn's disease activity index [PCDAI] >30), steroid/enteral-nutrition dependency, or postileocecal resection. Remission was defined as PCDAI ≤10 without steroids. Patients requiring antitumor necrosis factor (TNF) medication, other immunomodulators, or surgery were considered to experience a relapse. Based on PCDAI, steroid-free remission was achieved in 56/93 (60.2%), 37/93 (39.8%), 31/93 (33.3%), and 29/93 (31.2%) at visits month (M)6, M12, M18, and M24, respectively. Within the first 4 weeks, AZA was stopped in 10/93 patients due to adverse reactions (pancreatitis, nausea, vomiting, skin reactions, general weakness), or not introduced due to low thiopurine methyl transferase (TPMT) activity (n = 3). No neutropenia occurred in patients with normal TPMT activity. Three infectious episodes were documented requiring temporary AZA suspension. AZA is efficacious in maintaining remission in pediatric CD patients, but to a lesser extent than previously suggested. The majority of patients who are in steroid-free remission at 12 months remained in prolonged remission. Overall tolerance of AZA was excellent.
    Inflammatory Bowel Diseases 10/2011; 17(10):2138-43. DOI:10.1002/ibd.21612 · 4.46 Impact Factor
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    ABSTRACT: Nutritional therapy has an established role as induction therapy in paediatric Crohn's disease. However, compliance is the main difficulty and may be greatly influenced by the administration route. To analyse the efficiency of exclusive nutrition to induce remission in children with Crohn's disease comparing fractionated oral vs. continuous enteral feeding. The medical records of 106 patients treated by exclusive nutritional therapy [Modulen IBD (R)] by either oral or continuous enteral route were reviewed retrospectively. Comparative analyses of remission rates, changes in anthropometry, Paediatric Crohn's disease Activity Index (PCDAI), laboratory indices and compliance rates were performed. On exclusive enteral nutrition, at 8 weeks, 34/45 patients achieved remission in the oral group (75% on intention-to-treat analysis) and 52/61 (85%) in the enteral nutrition group (P = 0.157). All patients showed a significant decrease in disease severity assessed by PCDAI (P < 0.0001) and significant improvements in anthropometric measures and inflammatory indices. No difference was observed whether Modulen IBD was administered orally or by continuous enteral feeding, apart from weight gain, which was greater in the enteral group (P = 0.041). In a subgroup of patients, mucosal healing was evidenced on follow-up endoscopies showing a clear correlation to remission. Compliance rates (87% and 90%) were similar. Nevertheless, noncompliant patients had lower mucosal healing and remission rates. These retrospective data suggest that the use of fractionated oral nutritional therapy might be as efficacious as continuous enteral administration to induce remission and mucosal healing in children with Crohn's disease. However, appropriate prospective clinical trials are needed to confirm these findings.
    Alimentary Pharmacology & Therapeutics 06/2011; 33(12):1332-9. DOI:10.1111/j.1365-2036.2011.04662.x · 5.73 Impact Factor
  • Olivier Goulet · Cécile Talbotec ·

    La Revue du praticien 05/2011; 61(5):648-9.
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    ABSTRACT: Microvillous inclusion disease (MVID) is a rare congenital enterocyte disorder causing severe diarrhea and intestinal failure. The objective of this study was to analyze clinical evolution and the most frequent complications of MVID in children receiving parenteral nutrition (PN) and after small-bowel transplantation (SBTx) with the aim to improve treatment strategies and prognosis. From 1995 to 2009, 24 patients (16 boys, median follow-up 4.7 years, range: from birth to 23.5 years) with MVID were admitted to our unit. The recorded parameters included growth, neurological development, liver and renal functions, bone disease, and outcome. Almost half of the children were from consanguineous families from the Mediterranean area. All of the patients completely depended on PN. Four children died of PN complications before 4 years of age. Before or without SBTx, growth failure was common (mean height -2.5 standard deviations [SD]), as was developmental delay (12/24), liver (20/22 with fibrosis) or kidney disease (3/23 with moderate renal insufficiency), and osteoporosis (6/24). Thirteen children underwent SBTx (9 isolated, 4 combined with liver Tx) at a median age of 3.5 years. Follow-up after SBTx was 0.4 to 14 years. Patient survival rates were 63% without SBTx and 77% with SBTx. After SBTx, 4 children experienced catch-up growth. PN in MVID is difficult to manage and requires expertise. Despite improved results in expert centers, the risk of death or irreversible sequelae is higher with PN than after Tx. SBTx, despite being complicated, remains the only hope to improve the quality of life and long-term prognosis of these children.
    Journal of pediatric gastroenterology and nutrition 03/2011; 52(4):460-5. DOI:10.1097/MPG.0b013e3181fb4559 · 2.63 Impact Factor

Publication Stats

709 Citations
162.47 Total Impact Points


  • 2010-2014
    • Université René Descartes - Paris 5
      • Faculté de Médecine
      Lutetia Parisorum, Île-de-France, France
  • 2012
    • American Hospital of Paris
      Lutetia Parisorum, Île-de-France, France
  • 2011-2012
    • Assistance Publique – Hôpitaux de Paris
      Lutetia Parisorum, Île-de-France, France