You are here

Article Text

Article menu

Download PDFPDF

Multiple sclerosis
Research paper
How common is truly benign MS in a UK population?
  1. Emma Clare Tallantyre1,2,
  2. Paula C Major2,
  3. Michael J Atherton2,
  4. W Adam Davies2,
  5. Fady Joseph3,
  6. Valentina Tomassini1,2,
  7. Trevor P Pickersgill2,
  8. Katharine Elizabeth Harding1,2,
  9. Mark Douglas Willis1,2,
  10. Mia Winter4,
  11. Neil P Robertson1,2
  1. 1 Division of Psychological Medicine and Clinical Neurosciences, Cardiff University School of Medicine, Cardiff, UK
  2. 2 Helen Durham Centre for Neuroinflammation, University Hospital of Wales, Cardiff, UK
  3. 3 Department of Neurology, Royal Gwent Hospital, Newport, UK
  4. 4 Department of Clinical Neuropsychology, University Hospital of Wales, Cardiff, UK
  1. Correspondence to Dr Emma Clare Tallantyre, Division of Psychological Medicine and Clinical Neurosciences, Cardiff University School of Medicine, Cardiff, UK; TallantyreEC{at}cardiff.ac.uk

Abstract

Objectives The prevalence and definition of benign multiple sclerosis (BMS) remain controversial. Most definitions are based on the Expanded Disability Status Scale (EDSS), not encompassing the wider impact of disease. The explanation for favourable outcomes remains unclear. We aim to provide a detailed characterisation of patients with low EDSS scores at long disease durations.

Methods We screened a population-based registry containing 3062 people with MS to identify individuals with unlimited walking ability at disease durations >15 years. A representative cohort underwent detailed clinical assessment and classified as having BMS according to EDSS score <3, no significant fatigue, mood disturbance, cognitive impairment or disrupted employment, and had not received a disease-modifying therapy. We determined patient-reported perceptions of MS status and made comparisons with EDSS-based definitions.

Results Of 1049 patients with disease duration of >15 years, 200 (19.1%) had most recent EDSS score <4.0. Detailed contemporary clinical assessment of a representative sample of 60 of these patients revealed 48 (80%) had an EDSS score of <4.0, 35 (58%) <3.0 and 16 (27%) <2.0. Only nine (15%) fulfilled our criteria for BMS; impaired cognition (57%) and effects on employment (52%) the most common causes for exclusion. Meanwhile, 33/60 (69%) patients considered their disease benign. Population frequency for BMS was estimated at 2.9% (95% CI 2.0 to 4.1).

Conclusions Comprehensive assessment reveals a small minority of people with MS who appear genuinely benign after 15 years. Study of such individuals may uncover insights about disease pathogenesis. However, discrepancy between patient perception and clinician perception of BMS undermines use of the term ‘benign’ in clinical settings.

This is an Open Access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0

https://doi.org/10.1136/jnnp-2018-318802

Statistics from Altmetric.com

    Request Permissions

    If you wish to reuse any or all of this article please use the link below which will take you to the Copyright Clearance Center’s RightsLink service. You will be able to get a quick price and instant permission to reuse the content in many different ways.

    View Full Text

    Footnotes

    • Contributor ECT contributed to the conception or design of the work, the acquisition, analysis and interpretation of the data, drafting of the manuscript and provided final approval of the version published. PCM contributed to the acquisition of the data and drafting of the manuscript. MJA contributed to the acquisition of the data and drafting of the manuscript. WAD contributed to the acquisition of the data and drafting of the manuscript. FJ contributed to the acquisition of the data and drafting of the manuscript. VT contributed to the acquisition of the data and drafting of the manuscript. TPP contributed to the acquisition of the data and drafting of the manuscript. KEH contributed to the conception or design of the work, the acquisition of the data and drafting of the manuscript. MDW contributed to the acquisition of the data and drafting of the manuscript. MW contributed to the conception or design of the work, the acquisition, analysis and interpretation of the data, and drafting of the manuscript. NPR contributed to the conception or design of the work, the acquisition and interpretation of data, and drafting of the manuscript.

    • Funding This research was conducted while ECT was a Biogen Idec Postdoctoral Fellow.

    • Competing interests None declared.

    • Patient consent Obtained.

    • Ethics approval This study has been approved by the South East Wales Ethics Committee (ref no. 05/WSE03/111).

    • Provenance and peer review Not commissioned; externally peer reviewed.