βIV-Spectrin Autoantibodies in 2 Individuals With Neuropathy of Possible Paraneoplastic Origin: A Case Series.

Humans Autoantibodies Hypesthesia Paraneoplastic Polyneuropathy Paresthesia Spectrin Animals Mice

Journal

Neurology(R) neuroimmunology & neuroinflammation
ISSN: 2332-7812
Titre abrégé: Neurol Neuroimmunol Neuroinflamm
Pays: United States
ID NLM: 101636388

Informations de publication

Date de publication:
07 2022
Historique:
received: 16 01 2022
accepted: 18 04 2022
entrez: 17 5 2022
pubmed: 18 5 2022
medline: 21 5 2022
Statut: epublish

Résumé

To identify the autoantigen in 2 individuals with possible seronegative paraneoplastic neuropathy. Serum and CSF were screened by tissue-based assay and panned for candidate autoantibodies by phage display immunoprecipitation sequencing (PhIP-Seq). The candidate antigen was validated by immunostaining knockout tissue and HEK 293T cell-based assay. Case 1 presented with gait instability, distal lower extremity numbness, and paresthesias after a recent diagnosis of serous uterine and fallopian carcinoma. Case 2 had a remote history of breast adenocarcinoma and presented with gait instability, distal lower extremity numbness, and paresthesias that progressed to generalized weakness. CSF and serum from both patients immunostained the axon initial segment (AIS) and node of Ranvier (NoR) of mice and enriched βIV-spectrin by PhIP-Seq. Patient CSF and serum failed to immunostain NoRs in dorsal root sensory neurons from βI/βIV-deficient mice. βIV-spectrin autoantibodies were confirmed by overexpression of AIS and nodal βIV-spectrin isoforms Σ1 and Σ6 by a cell-based assay. βIV-spectrin was not enriched in a combined 4,815 PhIP-Seq screens of healthy and other neurologic disease patients. Therefore, βIV-spectrin autoantibodies may be a marker of paraneoplastic neuropathy. This study provides Class IV evidence that βIV-spectrin antibodies are specific autoantibody biomarkers for paraneoplastic neuropathy.

Identifiants

DOI: 10.1212/NXI.0000000000001188 PMID: 35581007 PMC: PMC9128026
pubmed: 35581007
pii: 9/4/e1188
doi: 10.1212/NXI.0000000000001188
pmc: PMC9128026
pii:
doi:

Substances chimiques

Autoantibodies 0
Spectrin 12634-43-4

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : Howard Hughes Medical Institute
Pays : United States
Organisme : NIH HHS
ID : S10 OD017993
Pays : United States
Organisme : NIMH NIH HHS
ID : R01 MH122471
Pays : United States
Organisme : NIMH NIH HHS
ID : R25 MH060482
Pays : United States
Organisme : NINDS NIH HHS
ID : K08 NS096117
Pays : United States

Informations de copyright

Copyright © 2022 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.

Références

Nat Neurosci. 2014 Dec;17(12):1664-72
pubmed: 25362473
J Neurol Neurosurg Psychiatry. 2022 Feb;93(2):196-200
pubmed: 34921120
Proc Natl Acad Sci U S A. 2001 Jun 5;98(12):6945-50
pubmed: 11391009
J Cell Biol. 2002 Jan 21;156(2):337-48
pubmed: 11807096
Elife. 2020 Feb 13;9:
pubmed: 32052742
J Cell Biol. 2004 Sep 27;166(7):983-90
pubmed: 15381686

Auteurs

Christopher M Bartley (CM)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0002-3525-7653

Thomas T Ngo (TT)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0003-2504-1872

Bonny D Alvarenga (BD)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0001-7922-9454

Andrew F Kung (AF)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.

Lindsay H Teliska (LH)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0003-1733-6910

Michael Sy (M)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0003-4826-0066

Joseph L DeRisi (JL)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0002-4611-9205

Matthew N Rasband (MN)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0001-8184-2477

Sean J Pittock (SJ)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0002-6140-5584

Divyanshu Dubey (D)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.

Michael R Wilson (MR)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN.
ORCID: 0000-0002-8705-5084

Samuel J Pleasure (SJ)

From the Weill Institute for Neurosciences (C.M.B., T.T.N., B.D.A., M.R.W., S.J. Pleasure), Department of Psychiatry and Behavioral Sciences (C.M.B., T.T.N.), Department of Neurology (B.D.A., M.R.W., S.J. Pleasure), UCSF School of Medicine (A.F.K.), University of California, San Francisco; Department of Neuroscience (L.H.T., M.N.R.), Baylor College of Medicine, Houston, TX; Department of Neurology (M.S.), University of California, Irvine; Chan Zuckerberg Biohub (J.L.D.), San Francisco, CA; Department of Biochemistry and Biophysics (J.L.D.), University of California, San Francisco; Department of Laboratory Medicine and Pathology (S.J. Pittock, D.D.), Department of Neurology (S.J. Pittock, D.D.), andCenter MS and Autoimmune Neurology (S.J. Pittock, D.D.), Mayo Clinic, Rochester, MN. samuel.pleasure@ucsf.edu.
ORCID: 0000-0001-8599-1613

Articles similaires

[Redispensing of expensive oral anticancer medicines: a practical application].

Lisanne N van Merendonk, Kübra Akgöl, Bastiaan Nuijen
1.00
Humans Antineoplastic Agents Administration, Oral Drug Costs Counterfeit Drugs

Smoking Cessation and Incident Cardiovascular Disease.

Jun Hwan Cho, Seung Yong Shin, Hoseob Kim et al.
1.00
Humans Male Smoking Cessation Cardiovascular Diseases Female

Evaluation of Low-Value Services Across Major Medicare Advantage Insurers and Traditional Medicare.

Ciara Duggan, Adam L Beckman, Ishani Ganguli et al.
1.00
Humans United States Aged Cross-Sectional Studies Medicare Part C

Effectiveness of Virtual Yoga for Chronic Low Back Pain: A Randomized Clinical Trial.

Hallie Tankha, Devyn Gaskins, Amanda Shallcross et al.
1.00
Humans Yoga Low Back Pain Female Male

Classifications MeSH

questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Primates › Haplorhini › Catarrhini › Hominidae › Humains › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Globulines › Sérum-globulines › Immunoglobulines › Anticorps › Autoanticorps › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › États, signes et symptômes pathologiques › Signes et symptômes › Manifestations neurologiques › Troubles sensitifs › Troubles somatosensoriels › Hypoesthésie › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › Maladies du système nerveux › Maladies neuromusculaires › Neuropathies périphériques › Polyneuropathies › Neuropathie paranéoplasique › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › États, signes et symptômes pathologiques › Signes et symptômes › Manifestations neurologiques › Troubles sensitifs › Troubles somatosensoriels › Paresthésie › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › Acides aminés, peptides et protéines › Protéines › Protéines membranaires › Spectrine › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › Eucaryotes › Animaux › βIV-Spectrin Autoantibodies in 2 Individuals...
questionsmedicales.fr › Eucaryotes › Animaux › Chordés › Vertébrés › Mammifères › Eutheria › Rodentia › Muridae › Murinae › Souris › βIV-Spectrin Autoantibodies in 2 Individuals...