Immunoglobulin G4-related Disease Accompanied by Peripheral Neuropathy: A Report of Two Cases.
IgG4-related disease
case report
peripheral neuropathy
systemic autoimmune disease
vasculitic neuropathy
Journal
Internal medicine (Tokyo, Japan)
ISSN: 1349-7235
Titre abrégé: Intern Med
Pays: Japan
ID NLM: 9204241
Informations de publication
Date de publication:
15 Jun 2021
15 Jun 2021
Historique:
pubmed:
19
1
2021
medline:
22
6
2021
entrez:
18
1
2021
Statut:
ppublish
Résumé
Due to its rarity and the limited literature, the clinicopathological characteristics of peripheral nerve involvement in immunoglobulin G4 (IgG4)-related disease are unknown. We present two cases of IgG4-related disease, accompanied by peripheral neuropathy, presenting as unilateral ptosis (case 1) and sclerosing cholangitis (case 2), respectively. In both cases, sural nerve biopsy indicated vasculitis as the underlying pathophysiology; the peripheral neuropathy was refractory to corticosteroid therapy. In contrast to the previously proposed pathomechanism of IgG4-related neuropathy (direct lymphoplasmacytic infiltration), the pathological findings in our cases suggest that vasculitis occurs secondary to systemic autoimmune conditions.
Identifiants
pubmed: 33456044
doi: 10.2169/internalmedicine.6461-20
pmc: PMC8263177
doi:
Substances chimiques
Immunoglobulin G
0
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
1941-1947Références
Rinsho Shinkeigaku. 2016 May 31;56(5):323-7
pubmed: 27098901
Hum Pathol. 2014 Feb;45(2):427-30
pubmed: 24289971
Arthritis Rheumatol. 2015 Jul;67(7):1688-99
pubmed: 25809420
Int J Rheumatol. 2012;2012:401890
pubmed: 22523496
JAMA Neurol. 2013 Apr;70(4):502-5
pubmed: 23440288
Mod Rheumatol. 2012 Feb;22(1):1-14
pubmed: 21881964
Ophthalmic Plast Reconstr Surg. 2020 Mar/Apr;36(2):109-117
pubmed: 31261163
Hum Pathol. 1991 Apr;22(4):387-95
pubmed: 2050373
Lancet Neurol. 2018 Feb;17(2):183-192
pubmed: 29413316
J Peripher Nerv Syst. 2010 Sep;15(3):176-84
pubmed: 21040139
Lancet. 2020 Mar 7;395(10226):816
pubmed: 32145795
N Engl J Med. 2001 Mar 8;344(10):732-8
pubmed: 11236777
Neurology. 2015 Oct 20;85(16):1400-7
pubmed: 26408495
Curr Treat Options Neurol. 2017 Apr;19(4):14
pubmed: 28374231
Jpn J Ophthalmol. 2015 Jan;59(1):1-7
pubmed: 25392273