Literature DB >> 33454188

Feeding difficulties in children and adolescents with spinal muscular atrophy type 2.

Renske I Wadman1, Ramona De Amicis2, Chiara Brusa3, Alberto Battezzati2, Simona Bertoli4, Tracey Davis5, Marion Main5, Adnan Manzur5, Chiara Mastella6, Pinki Munot5, Nadia Imbrigiotta3, Lucia Schottlaender7, Anna Sarkozy5, Federica Trucco3, Giovanni Baranello8, Mariacristina Scoto9, Francesco Muntoni10.   

Abstract

Disease course of feeding difficulties in spinal muscular atrophy type 2 is not well documented. Disease-modifying therapies rapidly change the trajectory of motor function and survival in spinal muscular atrophy, but effects on co-morbidities like bulbar function are unknown. We analysed data concerning feeding problems and their standard of care treatment in 146 patients with spinal muscular atrophy type 2. Data were collected from two separate cohorts: one single-centre retrospective chart review study from the United Kingdom (London), and one prospective questionnaire-based multicentre study from Italy. Cumulatively feeding difficulties were present in 88 patients (60%) in these 2 cohorts. Median age at onset of problems was 6.5years (range 0-16.5 years). Eighty-two patients (60%) showed periods of underweight according to age adjusted body mass index, and thirty-six patients (25%) showed malnourishment with a significant drop on their weight curves. Enteral feeding was indicated in 23 out of 72 patients in the UK cohort (32%) because of weight loss, oropharyngeal dysphagia or aspiration. Gastrostomy and its placement was generally well tolerated, uncomplicated in 96%, never reversed and performed without Nissen fundoplication in 66% of patients. After gastrostomy chest infections improved in 80% and nutritional status (e.g., Body Mass Index) in 84% of patients. These results show that feeding difficulties are a common problem in spinal muscular atrophy type 2. Treatment strategies should be tailor-made on the symptoms and needs of the individual patient.
Copyright © 2020. Published by Elsevier B.V.

Entities:  

Keywords:  Feeding difficulties; Nutrition; SMA; Spinal muscular atrophy; Underweight

Year:  2020        PMID: 33454188     DOI: 10.1016/j.nmd.2020.12.007

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  3 in total

Review 1.  Congenital or Early Developing Neuromuscular Diseases Affecting Feeding, Swallowing and Speech - A Review of the Literature from January 1998 to August 2021.

Authors:  Lotta Sjögreen; Lisa Bengtsson
Journal:  J Neuromuscul Dis       Date:  2022

2.  Distribution of weight, stature, and growth status in children and adolescents with spinal muscular atrophy: An observational retrospective study in the United States.

Authors:  Basil T Darras; Sabrina Guye; Janine Hoffart; Sophie Schneider; Isaac Gravestock; Ksenija Gorni; Sabine Fuerst-Recktenwald; Renata S Scalco; Richard S Finkel; Darryl C De Vivo
Journal:  Muscle Nerve       Date:  2022-05-11       Impact factor: 3.852

3.  Body mass index in type 2 spinal muscular atrophy: a longitudinal study.

Authors:  Gloria Ferrantini; Giorgia Coratti; Roberta Onesimo; Simona Lucibello; Sarah Bompard; Ida Turrini; Graziamaria Cicala; Michela Caprarelli; Maria Carmela Pera; Chiara Bravetti; Beatrice Berti; Valentina Giorgio; Claudio Bruno; Noemi Brolatti; Chiara Panicucci; Adele D'Amico; Antonella Longo; Chiara Leoni; Valeria A Sansone; Emilio Albamonte; Sonia Messina; Maria Sframeli; Enrico Bertini; Marika Pane; Eugenio Mercuri
Journal:  Eur J Pediatr       Date:  2022-01-19       Impact factor: 3.860

  3 in total

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