Echographic Assessment of Diaphragmatic Function in Duchenne Muscular Dystrophy from Childhood to Adulthood.


Journal

Journal of neuromuscular diseases
ISSN: 2214-3599
Titre abrégé: J Neuromuscul Dis
Pays: Netherlands
ID NLM: 101649948

Informations de publication

Date de publication:
Historique:
pubmed: 20 12 2018
medline: 3 1 2020
entrez: 20 12 2018
Statut: ppublish

Résumé

Duchenne muscular dystrophy (DMD) is an X-linked recessive genetic muscle disorder. Respiratory muscle function is classically affected in this disease. Ultrasound recently emerged as a non-invasive tool to assess diaphragm function. However, there are only a few studies using diaphragm ultrasound (US) in DMD. We aimed to assess diaphragm ultrasound patterns in DMD, their relationship with age and their association with home mechanical ventilation (HMV). We included DMD patients followed at Raymond Poincaré Hospital who benefited from diaphragm ultrasound and pulmonary function tests. There were 110 DMD patients and 17 male sex-matched healthy subjects included. In all, 94% of patients were permanent wheelchair users. Median body mass index (BMI) was 18 kg/m2. DMD patients disclosed a reduced forced vital capacity (VC) (12% of predicted value), and 78% of patients were on HMV. In patients, right and left diaphragmatic motions on deep inspiration were reduced and end expiratory diaphragm thickness was borderline normal. In patients, right and left diaphragmatic thickening fractions (TF) were reduced 12.7% and 15.5%, respectively. Age and end expiratory thickness were significantly inversely associated (p = 0.005 for the right diaphragm, p = 0.018 for the left diaphragm). Diaphragm TF was significantly inversely associated with age (p = 0.001 for the right side, p < 0.0001 for the left side). Right and left inspiratory diaphragm motions were significantly inversely associated with age (p < 0.0001). This study describes the severity of diaphragm dysfunction in patients with DMD. Diaphragm US may be a non-invasive outcome measure for DMD.

Sections du résumé

BACKGROUND BACKGROUND
Duchenne muscular dystrophy (DMD) is an X-linked recessive genetic muscle disorder. Respiratory muscle function is classically affected in this disease. Ultrasound recently emerged as a non-invasive tool to assess diaphragm function. However, there are only a few studies using diaphragm ultrasound (US) in DMD.
PURPOSE OBJECTIVE
We aimed to assess diaphragm ultrasound patterns in DMD, their relationship with age and their association with home mechanical ventilation (HMV).
METHODS METHODS
We included DMD patients followed at Raymond Poincaré Hospital who benefited from diaphragm ultrasound and pulmonary function tests.
RESULTS RESULTS
There were 110 DMD patients and 17 male sex-matched healthy subjects included. In all, 94% of patients were permanent wheelchair users. Median body mass index (BMI) was 18 kg/m2. DMD patients disclosed a reduced forced vital capacity (VC) (12% of predicted value), and 78% of patients were on HMV. In patients, right and left diaphragmatic motions on deep inspiration were reduced and end expiratory diaphragm thickness was borderline normal. In patients, right and left diaphragmatic thickening fractions (TF) were reduced 12.7% and 15.5%, respectively. Age and end expiratory thickness were significantly inversely associated (p = 0.005 for the right diaphragm, p = 0.018 for the left diaphragm). Diaphragm TF was significantly inversely associated with age (p = 0.001 for the right side, p < 0.0001 for the left side). Right and left inspiratory diaphragm motions were significantly inversely associated with age (p < 0.0001).
CONCLUSION CONCLUSIONS
This study describes the severity of diaphragm dysfunction in patients with DMD. Diaphragm US may be a non-invasive outcome measure for DMD.

Identifiants

pubmed: 30562904
pii: JND180326
doi: 10.3233/JND-180326
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

55-64

Auteurs

Abdallah Fayssoil (A)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.
Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Cendrine Chaffaut (C)

SBIM, CHU Saint Louis, APHP, Université Paris Diderot, Paris, France.

Adam Ogna (A)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Tanya Stojkovic (T)

Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Laure Lamothe (L)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Dominique Mompoint (D)

Service de Radiologie, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Paris Meng (P)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Helene Prigent (H)

Service de Physiologie - Explorations fonctionnelles, CHU Raymond Poincaré, APHP, Université de Versailles saint Quentin en Yvelines, Garches, France.

Bernard Clair (B)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Anthony Behin (A)

Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Pascal Laforet (P)

Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Guillaume Bassez (G)

Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Robert Carlier (R)

Service de Radiologie, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

David Orlikowski (D)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.
Centre d'Investigation clinique et Innovation technologique CIC 14.29, INSERM, Garches, France.

Helge Amthor (H)

Service de Pédiatrie, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Susana Quijano Roy (S)

Service de Pédiatrie, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Pascal Crenn (P)

Service de nutrition, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

Sylvie Chevret (S)

SBIM, CHU Saint Louis, APHP, Université Paris Diderot, Paris, France.

Bruno Eymard (B)

Institut de Myologie, CHU Pitié Salpetrière, Paris, France.

Frederic Lofaso (F)

Service de Physiologie - Explorations fonctionnelles, CHU Raymond Poincaré, APHP, Université de Versailles saint Quentin en Yvelines, Garches, France.

Djillali Annane (D)

Service de Réanimation msédicale et unité de ventilation à domicile, CHU Raymond Poincaré, APHP, Université de Versailles Saint Quentin en Yvelines, Garches, France.

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