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Published in: European Journal of Medical Research 1/2023

Open Access 01-12-2023 | Polyneuropathy | Research

Analysis of clinical features and risk factors of peripheral neuropathy in patients with primary Sjögren's syndrome

Authors: Zhihong Wu, Dong Wang, Lirong Chen, Kaipu Xianyu, Huiqing Yang

Published in: European Journal of Medical Research | Issue 1/2023

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Abstract

Objective

To observe the clinical features and efficacy of immunosuppressive therapy in patients with primary Sjögren's syndrome (PSS) combined with peripheral neuropathy (PN) syndrome and to explore the risk factors for PN in patients with PSS.

Methods

Sixty consecutive patients with PSS admitted to the Department of Rheumatology and Immunology, Wuhan No. 1 Hospital, from January 2014 to June 2020 were analysed retrospectively. Patients were divided into a PN group (N = 15) and a non-PN group (N = 45). The clinical characteristics of the two groups were compared, and the independent risk factors for PN combined with PSS were analysed by multivariate logistic regression. The patients with PSS combined with PN were followed up to observe the effect of immunosuppressive therapy.

Results

The patients with PN had a longer course of disease than those without PN (z = − 3.225, P = 0.001), and the incidence of Raynaud's phenomenon, anti-SSB antibody, rheumatoid factor and hyperglobulinaemia was higher (all P < 0.05) in patients with PN than in those without PN. Multivariate logistic regression analysis showed that hyperglobulinaemia, RF and anti-SSB antibodies were independent risk factors for PN with PSS (P < 0.05). Fourteen patients with PSS-PN were treated with immunosuppressants. The clinical symptoms of 10 patients were relieved, and mRS scores of 10 patients were decreased.

Conclusion

PN is a common complication in PSS patients. Patients with PSS combined with PN have a longer course of disease and a significantly higher percentage of Raynaud's phenomenon, positive anti-SSB antibody, positive RF and hyperglobulinaemia. Immunosuppressive therapy was effective for partial remission of PN with PSS.
Literature
1.
go back to reference Qin B, Wang J, Yang Z, Yang M, Ma N, Huang F, et al. Epidemiology of primary Sjogren’s syndrome: a systematic review and meta-analysis. Ann Rheum Dis. 2015;74(11):1983–9.CrossRef Qin B, Wang J, Yang Z, Yang M, Ma N, Huang F, et al. Epidemiology of primary Sjogren’s syndrome: a systematic review and meta-analysis. Ann Rheum Dis. 2015;74(11):1983–9.CrossRef
2.
go back to reference Pavlakis PP, Alexopoulos H, Kosmidis ML, Stamboulis E, Routsias JG, Tzartos SJ, et al. Peripheral neuropathies in Sjogren syndrome: a new reappraisal. J Neurol Neurosurg Psychiatry. 2011;82(7):798–802.CrossRef Pavlakis PP, Alexopoulos H, Kosmidis ML, Stamboulis E, Routsias JG, Tzartos SJ, et al. Peripheral neuropathies in Sjogren syndrome: a new reappraisal. J Neurol Neurosurg Psychiatry. 2011;82(7):798–802.CrossRef
3.
go back to reference Lafitte C, Amoura Z, Cacoub P, Pradat-Diehl P, Picq C, Salachas F, et al. Neurological complications of primary Sjogren’s syndrome. J Neurol. 2001;248(7):577–84.CrossRef Lafitte C, Amoura Z, Cacoub P, Pradat-Diehl P, Picq C, Salachas F, et al. Neurological complications of primary Sjogren’s syndrome. J Neurol. 2001;248(7):577–84.CrossRef
4.
go back to reference Mori K, Iijima M, Koike H, Hattori N, Tanaka F, Watanabe H, et al. The wide spectrum of clinical manifestations in Sjogren’s syndrome-associated neuropathy. Brain. 2005;128(Pt 11):2518–34.CrossRef Mori K, Iijima M, Koike H, Hattori N, Tanaka F, Watanabe H, et al. The wide spectrum of clinical manifestations in Sjogren’s syndrome-associated neuropathy. Brain. 2005;128(Pt 11):2518–34.CrossRef
5.
go back to reference Sopacua M, Hoeijmakers JGJ, Merkies ISJ, Lauria G, Waxman SG, Faber CG. Small-fiber neuropathy: expanding the clinical pain universe. J Peripher Nerv Syst. 2019;24(1):19–33.CrossRef Sopacua M, Hoeijmakers JGJ, Merkies ISJ, Lauria G, Waxman SG, Faber CG. Small-fiber neuropathy: expanding the clinical pain universe. J Peripher Nerv Syst. 2019;24(1):19–33.CrossRef
6.
go back to reference Vitali C. Classification criteria for Sjogren’s syndrome. Ann Rheum Dis. 2003;62(1):94–5 (author reply 5).CrossRef Vitali C. Classification criteria for Sjogren’s syndrome. Ann Rheum Dis. 2003;62(1):94–5 (author reply 5).CrossRef
7.
go back to reference Shiboski SC, Shiboski CH, Criswell L, Baer A, Challacombe S, Lanfranchi H, et al. American College of Rheumatology classification criteria for Sjogren’s syndrome: a data-driven, expert consensus approach in the Sjogren’s International Collaborative Clinical Alliance cohort. Arthritis Care Res. 2012;64(4):475–87.CrossRef Shiboski SC, Shiboski CH, Criswell L, Baer A, Challacombe S, Lanfranchi H, et al. American College of Rheumatology classification criteria for Sjogren’s syndrome: a data-driven, expert consensus approach in the Sjogren’s International Collaborative Clinical Alliance cohort. Arthritis Care Res. 2012;64(4):475–87.CrossRef
8.
go back to reference Dillingham T, Chen S, Andary M, Buschbacher R, Del Toro D, Smith B, et al. Establishing high-quality reference values for nerve conduction studies: a report from the normative data task force of the American Association of Neuromuscular & Electrodiagnostic Medicine. Muscle Nerve. 2016;54(3):366–70.CrossRef Dillingham T, Chen S, Andary M, Buschbacher R, Del Toro D, Smith B, et al. Establishing high-quality reference values for nerve conduction studies: a report from the normative data task force of the American Association of Neuromuscular & Electrodiagnostic Medicine. Muscle Nerve. 2016;54(3):366–70.CrossRef
9.
go back to reference Tankisi H, Pugdahl K, Fuglsang-Frederiksen A, Johnsen B, de Carvalho M, Fawcett PR, et al. Pathophysiology inferred from electrodiagnostic nerve tests and classification of polyneuropathies. Suggested guidelines. Clin Neurophysiol. 2005;116(7):1571–80.CrossRef Tankisi H, Pugdahl K, Fuglsang-Frederiksen A, Johnsen B, de Carvalho M, Fawcett PR, et al. Pathophysiology inferred from electrodiagnostic nerve tests and classification of polyneuropathies. Suggested guidelines. Clin Neurophysiol. 2005;116(7):1571–80.CrossRef
10.
go back to reference Seror R, Ravaud P, Bowman SJ, Baron G, Tzioufas A, Theander E, et al. EULAR Sjogren’s syndrome disease activity index: development of a consensus systemic disease activity index for primary Sjogren’s syndrome. Ann Rheum Dis. 2010;69(6):1103–9.CrossRef Seror R, Ravaud P, Bowman SJ, Baron G, Tzioufas A, Theander E, et al. EULAR Sjogren’s syndrome disease activity index: development of a consensus systemic disease activity index for primary Sjogren’s syndrome. Ann Rheum Dis. 2010;69(6):1103–9.CrossRef
11.
go back to reference Vitali C. Measurement of disease activity and damage in Sjogren’s syndrome. Rheum Dis Clin North Am. 2008;34(4):963–71.CrossRef Vitali C. Measurement of disease activity and damage in Sjogren’s syndrome. Rheum Dis Clin North Am. 2008;34(4):963–71.CrossRef
12.
go back to reference Jaskolska M, Chylinska M, Masiak A, Nowicka-Sauer K, Sieminski M, Zietkiewicz M, et al. Peripheral neuropathy and health-related quality of life in patients with primary Sjogren’s syndrome: a preliminary report. Rheumatol Int. 2020;40(8):1267–74.CrossRef Jaskolska M, Chylinska M, Masiak A, Nowicka-Sauer K, Sieminski M, Zietkiewicz M, et al. Peripheral neuropathy and health-related quality of life in patients with primary Sjogren’s syndrome: a preliminary report. Rheumatol Int. 2020;40(8):1267–74.CrossRef
13.
go back to reference Jaskolska M, Chylinska M, Masiak A, Sieminski M, Zietkiewicz M, Czuszynska Z, et al. Neuro-Sjogren: uncommon or underestimated problem? Brain Behav. 2020;10(8): e01665.CrossRef Jaskolska M, Chylinska M, Masiak A, Sieminski M, Zietkiewicz M, Czuszynska Z, et al. Neuro-Sjogren: uncommon or underestimated problem? Brain Behav. 2020;10(8): e01665.CrossRef
14.
go back to reference Gemignani F, Marbini A, Pavesi G, Di Vittorio S, Manganelli P, Cenacchi G, Mancia D. Peripheral neuropathy associated with primary. J Neurol Neurosurg Psychiatry. 1994;57(8):983–6.CrossRef Gemignani F, Marbini A, Pavesi G, Di Vittorio S, Manganelli P, Cenacchi G, Mancia D. Peripheral neuropathy associated with primary. J Neurol Neurosurg Psychiatry. 1994;57(8):983–6.CrossRef
15.
go back to reference Liampas A, Parperis K, Erotocritou MF, Nteveros A, Papadopoulou M, Moschovos C, et al. Primary Sjogren syndrome-related peripheral neuropathy: a systematic review and meta-analysis. Eur J Neurol. 2023;30(1):255–65.CrossRef Liampas A, Parperis K, Erotocritou MF, Nteveros A, Papadopoulou M, Moschovos C, et al. Primary Sjogren syndrome-related peripheral neuropathy: a systematic review and meta-analysis. Eur J Neurol. 2023;30(1):255–65.CrossRef
16.
go back to reference Font J, Ramos-Casals M, de la Red G, Pou A, Casanova A, Garcia-Carrasco M, et al. Pure sensory neuropathy in primary Sjogren’s syndrome. Long term prospective follow up and review of the literature. J Rheumatol. 2003;30(7):1552–7. Font J, Ramos-Casals M, de la Red G, Pou A, Casanova A, Garcia-Carrasco M, et al. Pure sensory neuropathy in primary Sjogren’s syndrome. Long term prospective follow up and review of the literature. J Rheumatol. 2003;30(7):1552–7.
17.
go back to reference Tani J, Liao HT, Hsu HC, Chen LF, Chang TS, Shin-Yi Lin C, et al. Immune-mediated axonal dysfunction in seropositive and seronegative primary Sjogren’s syndrome. Ann Clin Transl Neurol. 2020;7(5):819–28.CrossRef Tani J, Liao HT, Hsu HC, Chen LF, Chang TS, Shin-Yi Lin C, et al. Immune-mediated axonal dysfunction in seropositive and seronegative primary Sjogren’s syndrome. Ann Clin Transl Neurol. 2020;7(5):819–28.CrossRef
18.
go back to reference Sene D, Jallouli M, Lefaucheur JP, Saadoun D, Costedoat-Chalumeau N, Maisonobe T, et al. Peripheral neuropathies associated with primary Sjogren syndrome: immunologic profiles of nonataxic sensory neuropathy and sensorimotor neuropathy. Medicine. 2011;90(2):133–8.CrossRef Sene D, Jallouli M, Lefaucheur JP, Saadoun D, Costedoat-Chalumeau N, Maisonobe T, et al. Peripheral neuropathies associated with primary Sjogren syndrome: immunologic profiles of nonataxic sensory neuropathy and sensorimotor neuropathy. Medicine. 2011;90(2):133–8.CrossRef
19.
go back to reference Sene D, Cacoub P, Authier FJ, Haroche J, Creange A, Saadoun D, et al. Sjogren syndrome-associated small fiber neuropathy: characterization from a prospective series of 40 cases. Medicine. 2013;92(5):e10–8.CrossRef Sene D, Cacoub P, Authier FJ, Haroche J, Creange A, Saadoun D, et al. Sjogren syndrome-associated small fiber neuropathy: characterization from a prospective series of 40 cases. Medicine. 2013;92(5):e10–8.CrossRef
20.
go back to reference Hsu CW, Su YJ, Chang WN, Tsai NW, Chiu WC, Cheng BC, et al. The association between serological biomarkers and primary Sjogren’s syndrome associated with peripheral polyneuropathy. Biomed Res Int. 2014;2014: 902492.CrossRef Hsu CW, Su YJ, Chang WN, Tsai NW, Chiu WC, Cheng BC, et al. The association between serological biomarkers and primary Sjogren’s syndrome associated with peripheral polyneuropathy. Biomed Res Int. 2014;2014: 902492.CrossRef
21.
go back to reference Jamilloux Y, Magy L, Hurtevent JF, Gondran G, de Seze J, Launay D, et al. Immunological profiles determine neurological involvement in Sjogren’s syndrome. Eur J Intern Med. 2014;25(2):177–81.CrossRef Jamilloux Y, Magy L, Hurtevent JF, Gondran G, de Seze J, Launay D, et al. Immunological profiles determine neurological involvement in Sjogren’s syndrome. Eur J Intern Med. 2014;25(2):177–81.CrossRef
22.
go back to reference Ye W, Chen S, Huang X, Qin W, Zhang T, Zhu X, et al. Clinical features and risk factors of neurological involvement in Sjogren’s syndrome. BMC Neurosci. 2018;19(1):26.CrossRef Ye W, Chen S, Huang X, Qin W, Zhang T, Zhu X, et al. Clinical features and risk factors of neurological involvement in Sjogren’s syndrome. BMC Neurosci. 2018;19(1):26.CrossRef
23.
go back to reference Cafaro G, Perricone C, Carubbi F, Baldini C, Quartuccio L, Priori R, et al. Peripheral nervous system involvement in Sjogren’s syndrome: analysis of a cohort from the Italian research group on Sjogren’s syndrome. Front Immunol. 2021;12: 615656.CrossRef Cafaro G, Perricone C, Carubbi F, Baldini C, Quartuccio L, Priori R, et al. Peripheral nervous system involvement in Sjogren’s syndrome: analysis of a cohort from the Italian research group on Sjogren’s syndrome. Front Immunol. 2021;12: 615656.CrossRef
24.
go back to reference Ramos-Casals M, Brito-Zeron P, Bombardieri S, Bootsma H, De Vita S, Dorner T, et al. EULAR recommendations for the management of Sjogren’s syndrome with topical and systemic therapies. Ann Rheum Dis. 2020;79(1):3–18.CrossRef Ramos-Casals M, Brito-Zeron P, Bombardieri S, Bootsma H, De Vita S, Dorner T, et al. EULAR recommendations for the management of Sjogren’s syndrome with topical and systemic therapies. Ann Rheum Dis. 2020;79(1):3–18.CrossRef
25.
go back to reference Pindi Sala T, Villedieu M, Damian L, Crave JC, Pautot V, Stojanovich L, et al. Long-term efficacy of immunoglobulins in small fiber neuropathy related to Sjogren’s syndrome. J Neurol. 2020;267(12):3499–507.CrossRef Pindi Sala T, Villedieu M, Damian L, Crave JC, Pautot V, Stojanovich L, et al. Long-term efficacy of immunoglobulins in small fiber neuropathy related to Sjogren’s syndrome. J Neurol. 2020;267(12):3499–507.CrossRef
26.
go back to reference Mekinian A, Ravaud P, Hatron PY, Larroche C, Leone J, Gombert B, et al. Efficacy of rituximab in primary Sjogren’s syndrome with peripheral nervous system involvement: results from the AIR registry. Ann Rheum Dis. 2012;71(1):84–7.CrossRef Mekinian A, Ravaud P, Hatron PY, Larroche C, Leone J, Gombert B, et al. Efficacy of rituximab in primary Sjogren’s syndrome with peripheral nervous system involvement: results from the AIR registry. Ann Rheum Dis. 2012;71(1):84–7.CrossRef
27.
go back to reference Hirsch S, Skripuletz T, Seeliger T, Witte T, Thiele T. Selenium deficiency is associated with polyneuropathy in primary Sjogren’s syndrome. Clin Nutr ESPEN. 2022;50:212–7.CrossRef Hirsch S, Skripuletz T, Seeliger T, Witte T, Thiele T. Selenium deficiency is associated with polyneuropathy in primary Sjogren’s syndrome. Clin Nutr ESPEN. 2022;50:212–7.CrossRef
Metadata
Title
Analysis of clinical features and risk factors of peripheral neuropathy in patients with primary Sjögren's syndrome
Authors
Zhihong Wu
Dong Wang
Lirong Chen
Kaipu Xianyu
Huiqing Yang
Publication date
01-12-2023
Publisher
BioMed Central
Keyword
Polyneuropathy
Published in
European Journal of Medical Research / Issue 1/2023
Electronic ISSN: 2047-783X
DOI
https://doi.org/10.1186/s40001-023-01013-w

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