Respiratory muscle tests in neuromuscular children

  • Research type

    Research Study

  • Full title

    Respiratory muscle strength tests in neuromuscular paediatric patients

  • IRAS ID

    236895

  • Contact name

    Francesco Muntoni

  • Contact email

    francesco.muntoni@gosh.nhs.uk

  • Sponsor organisation

    Head of Governance, Clinical Trials and Contracts Division of Research & Innovation | Joint R&D Offi

  • Duration of Study in the UK

    1 years, 9 months, 1 days

  • Research summary

    Respiratory muscle involvement in neuromuscular disorders (NMD) is a common feature.
    The progressive reduction in lung volumes (expressed as Vital Capacity, VC) due to the combination of respiratory muscle impairment with the rib cage stiffness correlates with the onset of sleep disordered breathing (SDB) ranging from obstructive sleep apnoea to carbon dioxide (CO2) retention (defined nocturnal hypoventilation).
    Therefore, it is important that both daytime and overnight respiratory assessment are carried out regularly in neuromuscular patients as per British Thoracic Society current guidelines on the respiratory management in NMD children [Hull J et al. Thorax. 2012 Jul;67 Suppl 1:i1-40].

    Sniff inspiratory pressure (SNIP) is a test of inspiratory muscle strength detected through a nasal probe. SNIP has been used in monitoring the respiratory decline over time in Duchenne muscular Dystrophy patients and other adult NMD.
    When it comes to children, SNIP has been proven to be feasible and reproducible.The most significant advantage of SNIP is its feasibility in young children/those with developmental delay or in patients with bulbar dysfunction/ facial weakness.
    Two works have tried to correlate SNIP not only with lung volumes, but also with the presence/severity of sleep disordered breathing in children with neuromuscular disorders, with contrasting results mostly caused by the heterogeneity of the NMD diagnostic groups [Bersanini C. et al Eur Respir J 2012; 39: 1206–1212, Anderson VB. et al Neuromuscular Disorders 22 (2012) 528–533].

    Specific differences of SNIP values among specific NMD diagnostic groups, both in terms of repeatability, absolute numbers compared to standard values or prognostic usefulness have not been specifically addressed yet.

    We hypothesized that SNIP measurements are feasible and repeatable in a paediatric NMD population and that vary significantly across specific diagnostic NMD subgroups.

  • REC name

    West Midlands - Black Country Research Ethics Committee

  • REC reference

    19/WM/0042

  • Date of REC Opinion

    5 Mar 2019

  • REC opinion

    Further Information Favourable Opinion