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Gastrointestinal bleeding in children with familial dysautonomia: a case-control study.
Ramprasad, Chethan; Palma, Jose-Alberto; Norcliffe-Kaufmann, Lucy; Levy, Joseph; Chen, Lea Ann; Kaufmann, Horacio.
Afiliación
  • Ramprasad C; Division of Gastroenterology and Hepatology, Department of Medicine, New York University Grossman School of Medicine, New York City, NY, USA.
  • Palma JA; Division of Gastroenterology, University of Pennsylvania, Philadelphia, PA, USA.
  • Norcliffe-Kaufmann L; Department of Neurology, Dysautonomia Center, New York University Grossman School of Medicine, 530 First Av, Suite 9Q, New York City, NY, 10016, USA.
  • Levy J; Department of Neurology, Dysautonomia Center, New York University Grossman School of Medicine, 530 First Av, Suite 9Q, New York City, NY, 10016, USA.
  • Chen LA; Division of Pediatric Gastroenterology, New York University Grossman School of Medicine, New York City, NY, USA.
  • Kaufmann H; Division of Gastroenterology and Hepatology, Department of Medicine, New York University Grossman School of Medicine, New York City, NY, USA.
Clin Auton Res ; 33(2): 87-92, 2023 04.
Article en En | MEDLINE | ID: mdl-36735101
OBJECTIVE: Familial dysautonomia (FD) is a rare inherited autosomal recessive disorder with abnormal somatosensory, enteric, and afferent autonomic neurons. We aimed to define the incidence of gastrointestinal bleeding and its associated risk factors in patients with FD. METHODS: In this retrospective case-control study, we identified all episodes of gastrointestinal bleeding in patients with FD, occurring over four decades (January 1980-December 2017), using the New York University FD registry. RESULTS: We identified 104 episodes of gastrointestinal bleeding occurring in 60 patients with FD. The estimated incidence rate of gastrointestinal bleeds in the FD population rate was 4.20 episodes per 1000 person-years. We compared the 60 cases with 94 age-matched controls. Bleeding in the upper gastrointestinal tract from gastric and duodenal ulcers occurred most frequently (64 bleeds, 75.6%). Patients were more likely to have a gastrostomy (G)-tube and a Nissen fundoplication [odds ratio (OR) 3.73, 95% confidence interval (CI) 1.303-13.565] than controls. The mean time from G-tube placement to first gastrointestinal bleed was 7.01 years. The mean time from Nissen fundoplication to bleed was 7.01 years. Cases and controls had similar frequency of intake of nonsteroidal antiinflammatory drugs (NSAID) and selective serotonin reuptake inhibitors (SSRI). CONCLUSION: The incidence of gastrointestinal bleeding in the pediatric FD population was estimated to be 4.20 per 1000 person-years, 21 times higher than in the general pediatric population (0.2 per 1000 person-years). Patients with FD with a G-tube and a Nissen fundoplication had a higher risk of a subsequent gastrointestinal bleeding.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Disautonomía Familiar Tipo de estudio: Etiology_studies / Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Child / Humans Idioma: En Revista: Clin Auton Res Asunto de la revista: NEUROLOGIA Año: 2023 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Alemania
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Disautonomía Familiar Tipo de estudio: Etiology_studies / Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Child / Humans Idioma: En Revista: Clin Auton Res Asunto de la revista: NEUROLOGIA Año: 2023 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Alemania