Validation of robust tools to measure sialorrhea in amyotrophic lateral sclerosis: A study in a large French cohort

Abstract

There is an unmet need for validated tools to measure sialorrhea in amyotrophic lateral sclerosis, especially to evaluate treatments. We assessed the inter-/intra-rate reviewer reliability of two scales: the Oral Secretion Scale (OSS), specifically developed for ALS patients, and the Sialorrhea Scoring Scale (SSS), initially developed for Parkinson's disease patients. Sialorrhea was rated in 69 ALS consecutive patients by four evaluators: two neurologists, one nurse and one speech therapist. Inter-rater reliability was evaluated by the light kappa coefficient and intra-rater reliability by the weighted kappa coefficient. We also compared patients’ and caregivers’ answers. Results demonstrated that the two scales present a high inter-/intra-rater reliability: weighted kappas were 0.85 for both scales and light kappas 0.89 for the OSS and 0.88 for the SSS. Both scales also showed a good intra-profession reliability (OSS kappa = 0.84; SSS kappa = 0.79) and agreement between patients’ and caregivers’ answers. The SSS showed a higher responsiveness compared to OSS. In conclusion, both Oral Secretion Scale and Sialorrhea Scoring Scale are reliable tools to measure sialorrhea in ALS patients. Because of the wide range of salivation degrees, SSS may be more sensitive as a tool to evaluate treatments in patients with severe hypersialorrhea.

Key words::

• Risk
• ventilation
• therapy

Acknowledgements

Pamela A. Cazzolli received research grants from ResMed Inc, Bella Vista NSW, Australia. Gaelle Bruneteau received research grants from ARSla (Association pour la Recherche sur la SLA) and AFM (Association Francaise contre les Myopathies). Jesus Gonzalez enjoyed consultancies, with personal honorarium for technical expertise, from Boerhinger Ingelheim, Resmed, Philips Respironics, and Covidien; and also research grants from ARSla (French Association for Research in ALS), and Fondation Latran. François Salachas received travel grants from Euromotor consortium.

Benjamin Rix Brooks served on various advisory boards, e.g. Scientific Advisory Board membership for Avanir Pharmaceuticals, Teva Pharmaceuticals, Biogen Idec Pharmaceuticals, Cephalon Pharmaceuticals, Amgen Pharmaceuticals, Regeneron Pharmaceuticals, Sanofi-Aventis Pharmaceuticals, Rhone-Poulenc Rorer Pharmaceuticals, Center for Disease Control ALS Registry, American Academy of Neurology Task Force on ALS performance Measures, World Federation of Neurology, ALS Research Group. He was also a member of the NorthEast ALS Consortium, SouthEast ALS Alliance. He was supported in part by Carolinas ALS Research Fund, Pinstripes Fund at the Carolinas Healthcare Foundation and Muscular Dystrophy Association − ALS Division, and was a Site Investigator or Principal Investigator and/or Steering Committee Member on clinical trials in ALS including Ceftriaxone, Dexpramipexole, Cytokinetics CK2017357, Neuraltus, High-Fat, Tamoxifen-Creatine Selection, GSK anti-NOGO monoclonal antibody, Nuedexta, TCH346, xaliproden, BNDF, CNTF, GDNF.riluzole, IGF-1, albuterol, tamoxifen. Pierre François Pradat received research grants from ARSla (French Association for Research in ALS), AFM (French Association againt Myopathies), IRME (Institute for Research on Spinal Cord), INSERM (French National Institute for Health and Medical Research) and DHOS (Direction for Hospitalization and Organization of Healthcare). He received travel grants from GlaskoSmithKline, Sanofi-Aventis, Euromotor consortium and Thierry Latran's foundation.

Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.