Stridor-related gray matter alterations in multiple system atrophy: A pilot study

Claudia Testa, Giovanna Calandra-Buonaura, Stefania Evangelisti, Giulia Giannini, Federica Provini, Stefano Ratti, Annagrazia Cecere, Lia Talozzi, David Neil Manners, Raffaele Lodi, Caterina Tonon, Pietro Cortelli

Research output: Contribution to journalArticle

Abstract

Introduction: The neuroanatomical substrate of stridor associated with Multiple System Atrophy (MSA) remains unclear. We evaluated stridor-related gray matter (GM) changes in MSA. Methods: 36 MSA patients underwent standardized nocturnal video-polysomnography and brain MRI. Differences in GM density between MSA patients with and without stridor and a sample of 22 matched healthy controls were evaluated with Voxel Based Morphometry protocol supplemented by a specific tool (SUIT) for analysing infratentorial structures. Results: Stridor was confirmed in 14 patients (10 MSA-cerebellar variant; 10 M; mean ± SD age = 61.6 ± 8.9years; disease duration = 5.2 ± 2.9years) and absent in 22 (11 MSA-cerebellar variant; 18 M; age = 61.4 ± 9.9years; disease duration = 4.8 ± 3.4years). Compared to MSA without stridor, patients with stridor showed higher GM density in the cerebellum (p < 0.05, corrected for the MSA-cerebellar variant and uncorrected when considering both MSA-variants) and lower in the striatum (p < 0.05, uncorrected). Conclusions: This preliminary study has demonstrated for the first time in MSA stridor-related GM changes in striatal and cerebellar regions. Abnormalities in these regions were previously reported in dystonic disorders affecting laryngeal muscles, suggesting the hypothesis that stridor pathophysiology is dystonia-related. These results need however to be confirmed in a larger sample of patients.

Original languageEnglish
Pages (from-to)226-230
Number of pages5
JournalParkinsonism and Related Disorders
Volume62
DOIs
Publication statusPublished - May 1 2019

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Multiple System Atrophy
Respiratory Sounds
Gray Matter
Laryngeal Muscles
Dystonic Disorders
Corpus Striatum
Dystonia
Polysomnography
Cerebellum

Keywords

  • Multiple system atrophy
  • Stridor
  • Voxel based morphometry

ASJC Scopus subject areas

  • Neurology
  • Geriatrics and Gerontology
  • Clinical Neurology

Cite this

Stridor-related gray matter alterations in multiple system atrophy : A pilot study. / Testa, Claudia; Calandra-Buonaura, Giovanna; Evangelisti, Stefania; Giannini, Giulia; Provini, Federica; Ratti, Stefano; Cecere, Annagrazia; Talozzi, Lia; Manners, David Neil; Lodi, Raffaele; Tonon, Caterina; Cortelli, Pietro.

In: Parkinsonism and Related Disorders, Vol. 62, 01.05.2019, p. 226-230.

Research output: Contribution to journalArticle

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abstract = "Introduction: The neuroanatomical substrate of stridor associated with Multiple System Atrophy (MSA) remains unclear. We evaluated stridor-related gray matter (GM) changes in MSA. Methods: 36 MSA patients underwent standardized nocturnal video-polysomnography and brain MRI. Differences in GM density between MSA patients with and without stridor and a sample of 22 matched healthy controls were evaluated with Voxel Based Morphometry protocol supplemented by a specific tool (SUIT) for analysing infratentorial structures. Results: Stridor was confirmed in 14 patients (10 MSA-cerebellar variant; 10 M; mean ± SD age = 61.6 ± 8.9years; disease duration = 5.2 ± 2.9years) and absent in 22 (11 MSA-cerebellar variant; 18 M; age = 61.4 ± 9.9years; disease duration = 4.8 ± 3.4years). Compared to MSA without stridor, patients with stridor showed higher GM density in the cerebellum (p < 0.05, corrected for the MSA-cerebellar variant and uncorrected when considering both MSA-variants) and lower in the striatum (p < 0.05, uncorrected). Conclusions: This preliminary study has demonstrated for the first time in MSA stridor-related GM changes in striatal and cerebellar regions. Abnormalities in these regions were previously reported in dystonic disorders affecting laryngeal muscles, suggesting the hypothesis that stridor pathophysiology is dystonia-related. These results need however to be confirmed in a larger sample of patients.",
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T1 - Stridor-related gray matter alterations in multiple system atrophy

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AU - Testa, Claudia

AU - Calandra-Buonaura, Giovanna

AU - Evangelisti, Stefania

AU - Giannini, Giulia

AU - Provini, Federica

AU - Ratti, Stefano

AU - Cecere, Annagrazia

AU - Talozzi, Lia

AU - Manners, David Neil

AU - Lodi, Raffaele

AU - Tonon, Caterina

AU - Cortelli, Pietro

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N2 - Introduction: The neuroanatomical substrate of stridor associated with Multiple System Atrophy (MSA) remains unclear. We evaluated stridor-related gray matter (GM) changes in MSA. Methods: 36 MSA patients underwent standardized nocturnal video-polysomnography and brain MRI. Differences in GM density between MSA patients with and without stridor and a sample of 22 matched healthy controls were evaluated with Voxel Based Morphometry protocol supplemented by a specific tool (SUIT) for analysing infratentorial structures. Results: Stridor was confirmed in 14 patients (10 MSA-cerebellar variant; 10 M; mean ± SD age = 61.6 ± 8.9years; disease duration = 5.2 ± 2.9years) and absent in 22 (11 MSA-cerebellar variant; 18 M; age = 61.4 ± 9.9years; disease duration = 4.8 ± 3.4years). Compared to MSA without stridor, patients with stridor showed higher GM density in the cerebellum (p < 0.05, corrected for the MSA-cerebellar variant and uncorrected when considering both MSA-variants) and lower in the striatum (p < 0.05, uncorrected). Conclusions: This preliminary study has demonstrated for the first time in MSA stridor-related GM changes in striatal and cerebellar regions. Abnormalities in these regions were previously reported in dystonic disorders affecting laryngeal muscles, suggesting the hypothesis that stridor pathophysiology is dystonia-related. These results need however to be confirmed in a larger sample of patients.

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