Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders

Thomas G. Beach, Charles H. Adler, Geidy Serrano, Lucia I. Sue, D. G. Walker, Brittany Dugger, Holly A. Shill, Erika Driver-Dunckley, John N. Caviness, Anthony Intorcia, Jessica Filon, Sarah Scott, Angelica Garcia, Brittany Hoffman, Christine M. Belden, Kathryn J. Davis, Marwan N. Sabbagh

Research output: Contribution to journalArticle

28 Citations (Scopus)

Abstract

Background: Clinical misdiagnosis, particularly at early disease stages, is a roadblock to finding new therapies for Lewy body disorders. Biopsy of a peripheral site might provide improved diagnostic accuracy. Previously, we reported, from both autopsy and needle biopsy, a high prevalence of submandibular gland synucleinopathy in Parkinson's disease (PD). Here, we report on an extension of these studies to subjects with dementia with Lewy bodies (DLB) and other Lewy body disorders in 228 autopsied subjects from the Arizona Study of Aging and Neurodegenerative Disorders. Objective: To provide an estimate of the prevalence of histological synucleinopathy in the submandibular glands of subjects with PD and other Lewy body disorders. Methods: Submandibular gland sections from autopsied subjects were stained with an immunohistochemical method for β-synuclein phosphorylated at serine 129. Included were 146 cases with CNS Lewy-type synucleinopathy (LTS), composed of 46 PD, 28 DLB, 14 incidental Lewy body disease (ILBD), 33 Alzheimer's disease with Lewy bodies (ADLB) and 2 with progressive supranuclear palsy and Lewy bodies (PSPLB). Control subjects included 79 normal elderly, 15 AD, 12 PSP, 2 conticobasal degeneration (CBD) and 2 multiple system atrophy (MSA). Results: Submandibular gland LTS was found in 42/47 (89%) of the PD subjects, 20/28 (71%) DLB, 4/33 (12%) ADLB and 1/9 (11%) ILBD subjects but none of the 110 control subjects. Conclusions: These results provide support for further clinical trials of in vivo submandibular gland diagnostic biopsy for PD and DLB. An accurate peripheral biopsy diagnosis would assist subject selection for clinical trials and could also be used to verify other biomarkers.

Original languageEnglish (US)
Pages (from-to)153-163
Number of pages11
JournalJournal of Parkinson's Disease
Volume6
Issue number1
DOIs
StatePublished - Mar 30 2016

Fingerprint

Lewy Bodies
Lewy Body Disease
Submandibular Gland
Parkinson Disease
Biopsy
Synucleins
Clinical Trials
Multiple System Atrophy
Needle Biopsy
Diagnostic Errors
Neurodegenerative Diseases
Serine
Patient Selection
Autopsy
Alzheimer Disease
Biomarkers

Keywords

  • biomarker
  • Biopsy
  • clinical trial
  • diagnosis
  • pathology
  • therapy

ASJC Scopus subject areas

  • Clinical Neurology
  • Cellular and Molecular Neuroscience

Cite this

Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders. / Beach, Thomas G.; Adler, Charles H.; Serrano, Geidy; Sue, Lucia I.; Walker, D. G.; Dugger, Brittany; Shill, Holly A.; Driver-Dunckley, Erika; Caviness, John N.; Intorcia, Anthony; Filon, Jessica; Scott, Sarah; Garcia, Angelica; Hoffman, Brittany; Belden, Christine M.; Davis, Kathryn J.; Sabbagh, Marwan N.

In: Journal of Parkinson's Disease, Vol. 6, No. 1, 30.03.2016, p. 153-163.

Research output: Contribution to journalArticle

Beach, TG, Adler, CH, Serrano, G, Sue, LI, Walker, DG, Dugger, B, Shill, HA, Driver-Dunckley, E, Caviness, JN, Intorcia, A, Filon, J, Scott, S, Garcia, A, Hoffman, B, Belden, CM, Davis, KJ & Sabbagh, MN 2016, 'Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders', Journal of Parkinson's Disease, vol. 6, no. 1, pp. 153-163. https://doi.org/10.3233/JPD-150680
Beach, Thomas G. ; Adler, Charles H. ; Serrano, Geidy ; Sue, Lucia I. ; Walker, D. G. ; Dugger, Brittany ; Shill, Holly A. ; Driver-Dunckley, Erika ; Caviness, John N. ; Intorcia, Anthony ; Filon, Jessica ; Scott, Sarah ; Garcia, Angelica ; Hoffman, Brittany ; Belden, Christine M. ; Davis, Kathryn J. ; Sabbagh, Marwan N. / Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders. In: Journal of Parkinson's Disease. 2016 ; Vol. 6, No. 1. pp. 153-163.
@article{f4e3e139fd4a4908a6fba334f8c02e6c,
title = "Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders",
abstract = "Background: Clinical misdiagnosis, particularly at early disease stages, is a roadblock to finding new therapies for Lewy body disorders. Biopsy of a peripheral site might provide improved diagnostic accuracy. Previously, we reported, from both autopsy and needle biopsy, a high prevalence of submandibular gland synucleinopathy in Parkinson's disease (PD). Here, we report on an extension of these studies to subjects with dementia with Lewy bodies (DLB) and other Lewy body disorders in 228 autopsied subjects from the Arizona Study of Aging and Neurodegenerative Disorders. Objective: To provide an estimate of the prevalence of histological synucleinopathy in the submandibular glands of subjects with PD and other Lewy body disorders. Methods: Submandibular gland sections from autopsied subjects were stained with an immunohistochemical method for β-synuclein phosphorylated at serine 129. Included were 146 cases with CNS Lewy-type synucleinopathy (LTS), composed of 46 PD, 28 DLB, 14 incidental Lewy body disease (ILBD), 33 Alzheimer's disease with Lewy bodies (ADLB) and 2 with progressive supranuclear palsy and Lewy bodies (PSPLB). Control subjects included 79 normal elderly, 15 AD, 12 PSP, 2 conticobasal degeneration (CBD) and 2 multiple system atrophy (MSA). Results: Submandibular gland LTS was found in 42/47 (89{\%}) of the PD subjects, 20/28 (71{\%}) DLB, 4/33 (12{\%}) ADLB and 1/9 (11{\%}) ILBD subjects but none of the 110 control subjects. Conclusions: These results provide support for further clinical trials of in vivo submandibular gland diagnostic biopsy for PD and DLB. An accurate peripheral biopsy diagnosis would assist subject selection for clinical trials and could also be used to verify other biomarkers.",
keywords = "biomarker, Biopsy, clinical trial, diagnosis, pathology, therapy",
author = "Beach, {Thomas G.} and Adler, {Charles H.} and Geidy Serrano and Sue, {Lucia I.} and Walker, {D. G.} and Brittany Dugger and Shill, {Holly A.} and Erika Driver-Dunckley and Caviness, {John N.} and Anthony Intorcia and Jessica Filon and Sarah Scott and Angelica Garcia and Brittany Hoffman and Belden, {Christine M.} and Davis, {Kathryn J.} and Sabbagh, {Marwan N.}",
year = "2016",
month = "3",
day = "30",
doi = "10.3233/JPD-150680",
language = "English (US)",
volume = "6",
pages = "153--163",
journal = "Journal of Parkinson's Disease",
issn = "1877-7171",
publisher = "IOS Press",
number = "1",

}

TY - JOUR

T1 - Prevalence of Submandibular Gland Synucleinopathy in Parkinson's Disease, Dementia with Lewy Bodies and other Lewy Body Disorders

AU - Beach, Thomas G.

AU - Adler, Charles H.

AU - Serrano, Geidy

AU - Sue, Lucia I.

AU - Walker, D. G.

AU - Dugger, Brittany

AU - Shill, Holly A.

AU - Driver-Dunckley, Erika

AU - Caviness, John N.

AU - Intorcia, Anthony

AU - Filon, Jessica

AU - Scott, Sarah

AU - Garcia, Angelica

AU - Hoffman, Brittany

AU - Belden, Christine M.

AU - Davis, Kathryn J.

AU - Sabbagh, Marwan N.

PY - 2016/3/30

Y1 - 2016/3/30

N2 - Background: Clinical misdiagnosis, particularly at early disease stages, is a roadblock to finding new therapies for Lewy body disorders. Biopsy of a peripheral site might provide improved diagnostic accuracy. Previously, we reported, from both autopsy and needle biopsy, a high prevalence of submandibular gland synucleinopathy in Parkinson's disease (PD). Here, we report on an extension of these studies to subjects with dementia with Lewy bodies (DLB) and other Lewy body disorders in 228 autopsied subjects from the Arizona Study of Aging and Neurodegenerative Disorders. Objective: To provide an estimate of the prevalence of histological synucleinopathy in the submandibular glands of subjects with PD and other Lewy body disorders. Methods: Submandibular gland sections from autopsied subjects were stained with an immunohistochemical method for β-synuclein phosphorylated at serine 129. Included were 146 cases with CNS Lewy-type synucleinopathy (LTS), composed of 46 PD, 28 DLB, 14 incidental Lewy body disease (ILBD), 33 Alzheimer's disease with Lewy bodies (ADLB) and 2 with progressive supranuclear palsy and Lewy bodies (PSPLB). Control subjects included 79 normal elderly, 15 AD, 12 PSP, 2 conticobasal degeneration (CBD) and 2 multiple system atrophy (MSA). Results: Submandibular gland LTS was found in 42/47 (89%) of the PD subjects, 20/28 (71%) DLB, 4/33 (12%) ADLB and 1/9 (11%) ILBD subjects but none of the 110 control subjects. Conclusions: These results provide support for further clinical trials of in vivo submandibular gland diagnostic biopsy for PD and DLB. An accurate peripheral biopsy diagnosis would assist subject selection for clinical trials and could also be used to verify other biomarkers.

AB - Background: Clinical misdiagnosis, particularly at early disease stages, is a roadblock to finding new therapies for Lewy body disorders. Biopsy of a peripheral site might provide improved diagnostic accuracy. Previously, we reported, from both autopsy and needle biopsy, a high prevalence of submandibular gland synucleinopathy in Parkinson's disease (PD). Here, we report on an extension of these studies to subjects with dementia with Lewy bodies (DLB) and other Lewy body disorders in 228 autopsied subjects from the Arizona Study of Aging and Neurodegenerative Disorders. Objective: To provide an estimate of the prevalence of histological synucleinopathy in the submandibular glands of subjects with PD and other Lewy body disorders. Methods: Submandibular gland sections from autopsied subjects were stained with an immunohistochemical method for β-synuclein phosphorylated at serine 129. Included were 146 cases with CNS Lewy-type synucleinopathy (LTS), composed of 46 PD, 28 DLB, 14 incidental Lewy body disease (ILBD), 33 Alzheimer's disease with Lewy bodies (ADLB) and 2 with progressive supranuclear palsy and Lewy bodies (PSPLB). Control subjects included 79 normal elderly, 15 AD, 12 PSP, 2 conticobasal degeneration (CBD) and 2 multiple system atrophy (MSA). Results: Submandibular gland LTS was found in 42/47 (89%) of the PD subjects, 20/28 (71%) DLB, 4/33 (12%) ADLB and 1/9 (11%) ILBD subjects but none of the 110 control subjects. Conclusions: These results provide support for further clinical trials of in vivo submandibular gland diagnostic biopsy for PD and DLB. An accurate peripheral biopsy diagnosis would assist subject selection for clinical trials and could also be used to verify other biomarkers.

KW - biomarker

KW - Biopsy

KW - clinical trial

KW - diagnosis

KW - pathology

KW - therapy

UR - http://www.scopus.com/inward/record.url?scp=84963732467&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84963732467&partnerID=8YFLogxK

U2 - 10.3233/JPD-150680

DO - 10.3233/JPD-150680

M3 - Article

C2 - 26756744

AN - SCOPUS:84963732467

VL - 6

SP - 153

EP - 163

JO - Journal of Parkinson's Disease

JF - Journal of Parkinson's Disease

SN - 1877-7171

IS - 1

ER -