ArticleNeurology2022
Diagnosing Premotor Multiple System Atrophy: Natural History and Autonomic Testing in an Autopsy-Confirmed Cohort.
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10 citing papers in PubMed, 16 citations in OpenAlex.
- Article
- Heart rate variability provides prognostic value in multiple system atrophy.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2026Article
- Locus coeruleus degeneration is associated with orthostatic hypotension in Parkinson's disease and multiple system atrophy.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2026Article
- A case report of syndrome of inappropriate antidiuretic hormone secretion unveiling hypothalamic involvement in multiple system atrophy.Frontiers in endocrinology · 2026Article
- Rapid Progression and Shorter Survival in Indians Affected with Probable MSA: A Prospective Cohort Study.Movement disorders clinical practice · 2025Article
- Multimodal Autonomic Biomarkers Predict Phenoconversion in Pure Autonomic Failure.Annals of clinical and translational neurology · 2025Observational
- Autonomic nervous system dysfunction in idiopathic REM sleep behavior disorder as a short-term risk for a synucleinopathy.Journal of neurology · 2024Article
- The comorbidity profiles and medication issues of patients with multiple system atrophy: a systematic cross-sectional analysis.Journal of neurology · 2024Article
- Deterioration of Cough, Respiratory, and Vocal Cord Functions in Patients with Multiple System Atrophy.Neurology international · 2023Article
- Autonomic function testing in multiple system atrophy: a prognostic biomarker? and other updates on recent autonomic research.Clinical autonomic research : official journal of the Clinical Autonomic Research Society · 2022Article
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16 authors at 1 institution in 2 countries.
Ekawat VichayanratFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Fernanda ValerioFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0001-5248-7314
Shiwen KoayFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0002-0860-9211
Eduardo De Pablo-FernandezFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0003-2834-2515
Jalesh PanickerFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Huw MorrisFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0002-5473-3774
Kailash BhatiaFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0001-8185-286X
Viorica ChelbanFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Henry HouldenFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.ORCID 0000-0002-2866-7777
Niall QuinnFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Judith Navarro-OtanoFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Yasuo MikiFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Janice HoltonFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Thomas WarnerFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Christopher MathiasFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom.
Valeria IodiceFrom the Autonomic Unit (E.V., F.V., S.K., J.N.-O., V.I.), National Hospital for Neurology and Neurosurgery, Queen Square; Department of Brain Repair and Rehabilitation (E.V., S.K., J.P., C.M., V.I.), Reta Lila Weston Institute for Neurological Studies (E.D.P.-F., N.Q., Y.M., J.H., T.W.), and Queen Square Brain Bank for Neurological Disorders (E.D.P.-F., N.Q., Y.M., J.H., T.W.), UCL Queen Square Institute of Neurology; Department of Uro Neurology (J.P.), National Hospital for Neurology and Neurosurgery; Department of Clinical and Movement Neuroscience (V.C., H.H.), and Department of Neuromuscular Diseases (Y.M.), UCL Institute of Neurology, Queen Square, London, United Kingdom; Service of Neurology (H.M., K.B.), Hospital Clinic, Barcelona, Spain and Institut d'Investigacions Biomèdiques August Pi i Sunyer (IDIBAPS); Department of Neuropathology (J.N.-O.), Institute of Brain Science, Hirosaki University Graduate School of Medicine, Japan; and The Lindo Wing (C.M.), Imperial College Healthcare NHS Trust, St Mary's Hospital, London, United Kingdom. v.iodice@ucl.ac.uk.
Hirosaki University · JP
Funding
Department of HealthMedical Research Council N013255/1
Abstract
BACKGROUND AND
objectivesNonmotor features precede motor symptoms in many patients with multiple system atrophy (MSA). However, little is known about differences between the natural history, progression, and prognostic factors for survival in patients with MSA with nonmotor vs motor presentations. We aimed to compare initial symptoms, disease progression, and clinical features at final evaluation and investigate differences in survival and natural history between patients with MSA with motor and nonmotor presentations.
methodsMedical records of autopsy-confirmed MSA cases at Queen Square Brain Bank who underwent both clinical examination and cardiovascular autonomic testing were identified. Clinical features, age at onset, sex, time from onset to diagnosis, disease duration, autonomic function tests, and plasma noradrenaline levels were evaluated.
resultsForty-seven patients with autopsy-confirmed MSA (age 60 ± 8 years; 28 men) were identified. Time from symptom onset to first autonomic evaluation was 4 ± 2 years, and the disease duration was 7.7 ± 2.2 years. Fifteen (32%) patients presented with nonmotor features including genitourinary dysfunction, orthostatic hypotension, or REM sleep behavior disorder before developing motor involvement (median delay 1-6 years). A third (5/15) were initially diagnosed with pure autonomic failure (PAF) before evolving into MSA. All these patients had normal supine plasma noradrenaline levels (332.0 ± 120.3 pg/mL) with no rise on head-up tilt (0.1 ± 0.3 pg/mL). Patients with MSA with early cardiovascular autonomic dysfunction (within 3 years of symptom onset) had shorter survival compared with those with later onset of cardiovascular autonomic impairment (6.8 years [5.6-7.9] vs 8.5 years [7.9-9.2]; DISCUSSION: Almost one-third of patients with MSA presented with nonmotor features, which could predate motor symptoms by up to 6 years. Cardiovascular autonomic failure and early urinary catheterization were predictors of poorer outcomes. A normal supine plasma noradrenaline level in patients presenting with PAF phenotype is a possible autonomic biomarker indicating later conversion to MSA.
Indexed as
Autonomic Nervous System DiseasesMultiple System AtrophyPure Autonomic FailureAutonomic Nervous SystemAutopsyDisease ProgressionHumansNorepinephrineNorepinephrine
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