Sjogren’s syndrome related sensory motor neuropathy and autonomic neuropathy: A Case report
DOI:
https://doi.org/10.17161/rrnmf.v1i5.14526Keywords:
sjogrens, neuropathy, NCS, EMG, biopsyAbstract
Neurologic involvement has been reported in primary Sjogren’s syndrome (SS) in approximately 10–25% of cases 1, 2, 3. Peripheral neuropathy is a major neurological manifestation of Sjögren's syndrome 4 and its etiology has been considered to be vasculitis in the peripheral nerves 2. While neuropathic symptoms of SS can be varied, it is unusual to have two different types of neuropathic presentations simultaneously in a patient. We describe a case of Sjogren’s syndrome presenting with autonomic symptoms who was noted to have large fiber neuropathy on EMG and inflammatory changes on nerve biopsy.
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2. Mori K, Iijima M, Koike H, et al. The wide spectrum of clinical manifestations in Sjögren's syndrome-associated neuropathy. Brain. 2005;128(Pt 11):2518‐2534. doi:10.1093/brain/awh605
3. Griffin JW, Cornblath DR, Alexander E, et al. Ataxic sensory neuropathy and dorsal root ganglionitis associated with Sjögren's syndrome. Ann Neurol 1990; 27: 304–15.
4. Noguchi Y, Tsuchiyama T, Matsumoto T, et al. Two distinct types of neuropathy associated with Sjögren's syndrome developed in one patient. The importance of the selection of an appropriate therapeutic regimen. Rinsho Shinkeigaku 2003; 43: 539–43.
5. Koike H, Sobue G. Brain Nerve. 2013;65(11):1333-1342.
6. Margaretten M. Neurologic Manifestations of Primary Sjögren Syndrome. Rheum Dis Clin North Am. 2017;43(4):519-529. doi:10.1016/j.rdc.2017.06.002
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8. Tobón GJ, Pers JO, Devauchelle-Pensec V, et al. Neurological Disorders in Primary Sjögren's Syndrome. Autoimmune Dis. 2012;2012:645967. doi:10.1155/2012/645967
9. Park K, Haberberger RV, Gordon TP, et al. Antibodies interfering with the type 3 muscarinic receptor pathway inhibit gastrointestinal motility and cholinergic neurotransmission in Sjögren's syndrome. Arthritis Rheum. 2011 May; 63(5):1426-34.
10. Gøransson LG, Herigstad A, Tjensvoll AB, et al. Peripheral neuropathy in primary Sjögren’s syndrome: a population-based study. Archives of Neurology. 2006;63(11):1612–1615.
11. Sivadasan A, Muthusamy K, Patel B, et al. Clinical spectrum, therapeutic outcomes, and prognostic predictors in sjogren's syndrome-associated neuropathy. Ann Indian Acad Neurol 2017;20:278-83
12. McCoy SS, Baer AN. Neurological Complications of Sjögren's Syndrome: Diagnosis and Management. Curr Treatm Opt Rheumatol. 2017;3(4):275-288. doi:10.1007/s40674-017-0076-9
13. Chen WH, Yeh JH, Chiu HC. Plasmapheresis in the treatment of ataxic sensory neuropathy associated with Sjögren's syndrome. Eur Neurol 2001; 45: 270–4.
14. Molina JA, Benito-Leon J, Bermejo F, et al. Intravenous immunoglobulin therapy in sensory neuropathy associated with Sjögren's syndrome. J Neurol Neurosurg Psychiatry 1996; 60: 699.
15. Pascual J, Cid C, Berciano J. High-dose IV immunoglobulin for peripheral neuropathy associated with Sjögren's syndrome. Neurology 1998; 51: 650–1.
16. Takahashi Y, Takata T, Hoshino M, et al. Benefit of IVIG for long-standing ataxic sensory neuronopathy with Sjögren's syndrome. IV immunoglobulin. Neurology 2003; 60: 503–5.
17. Alix JJ, Hadjivassiliou M, Ali R, et al. Sensory ganglionopathy with livedoid vascu- lopathy controlled by immunotherapy. Muscle Nerve. 2015;51(2):296–301. 10.1002/mus.24452.
18. Gorson KC, Natarajan N, Ropper AH, et al. Rituximab treatment in patients with IVIg-dependent immune polyneuropathy: a prospective pilot trial. Muscle Nerve. 2007;35(1):66–9. 10.1002/mus.20664.
2. Mori K, Iijima M, Koike H, et al. The wide spectrum of clinical manifestations in Sjögren's syndrome-associated neuropathy. Brain. 2005;128(Pt 11):2518‐2534. doi:10.1093/brain/awh605
3. Griffin JW, Cornblath DR, Alexander E, et al. Ataxic sensory neuropathy and dorsal root ganglionitis associated with Sjögren's syndrome. Ann Neurol 1990; 27: 304–15.
4. Noguchi Y, Tsuchiyama T, Matsumoto T, et al. Two distinct types of neuropathy associated with Sjögren's syndrome developed in one patient. The importance of the selection of an appropriate therapeutic regimen. Rinsho Shinkeigaku 2003; 43: 539–43.
5. Koike H, Sobue G. Brain Nerve. 2013;65(11):1333-1342.
6. Margaretten M. Neurologic Manifestations of Primary Sjögren Syndrome. Rheum Dis Clin North Am. 2017;43(4):519-529. doi:10.1016/j.rdc.2017.06.002
7. Perzyńska-Mazan J, Maślińska M, Gasik R. Neurological manifestations of primary Sjögren's syndrome. Reumatologia. 2018;56(2):99-105. doi:10.5114/reum.2018.75521
8. Tobón GJ, Pers JO, Devauchelle-Pensec V, et al. Neurological Disorders in Primary Sjögren's Syndrome. Autoimmune Dis. 2012;2012:645967. doi:10.1155/2012/645967
9. Park K, Haberberger RV, Gordon TP, et al. Antibodies interfering with the type 3 muscarinic receptor pathway inhibit gastrointestinal motility and cholinergic neurotransmission in Sjögren's syndrome. Arthritis Rheum. 2011 May; 63(5):1426-34.
10. Gøransson LG, Herigstad A, Tjensvoll AB, et al. Peripheral neuropathy in primary Sjögren’s syndrome: a population-based study. Archives of Neurology. 2006;63(11):1612–1615.
11. Sivadasan A, Muthusamy K, Patel B, et al. Clinical spectrum, therapeutic outcomes, and prognostic predictors in sjogren's syndrome-associated neuropathy. Ann Indian Acad Neurol 2017;20:278-83
12. McCoy SS, Baer AN. Neurological Complications of Sjögren's Syndrome: Diagnosis and Management. Curr Treatm Opt Rheumatol. 2017;3(4):275-288. doi:10.1007/s40674-017-0076-9
13. Chen WH, Yeh JH, Chiu HC. Plasmapheresis in the treatment of ataxic sensory neuropathy associated with Sjögren's syndrome. Eur Neurol 2001; 45: 270–4.
14. Molina JA, Benito-Leon J, Bermejo F, et al. Intravenous immunoglobulin therapy in sensory neuropathy associated with Sjögren's syndrome. J Neurol Neurosurg Psychiatry 1996; 60: 699.
15. Pascual J, Cid C, Berciano J. High-dose IV immunoglobulin for peripheral neuropathy associated with Sjögren's syndrome. Neurology 1998; 51: 650–1.
16. Takahashi Y, Takata T, Hoshino M, et al. Benefit of IVIG for long-standing ataxic sensory neuronopathy with Sjögren's syndrome. IV immunoglobulin. Neurology 2003; 60: 503–5.
17. Alix JJ, Hadjivassiliou M, Ali R, et al. Sensory ganglionopathy with livedoid vascu- lopathy controlled by immunotherapy. Muscle Nerve. 2015;51(2):296–301. 10.1002/mus.24452.
18. Gorson KC, Natarajan N, Ropper AH, et al. Rituximab treatment in patients with IVIg-dependent immune polyneuropathy: a prospective pilot trial. Muscle Nerve. 2007;35(1):66–9. 10.1002/mus.20664.
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Published
2020-11-30
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Clinic and Case Reports
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Copyright (c) 2020 Amtul Farheen, William Jens, Cunfeng Pu, Aiesha Ahmed
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How to Cite
Farheen, A., Jens, W., Pu, C., & Ahmed, A. (2020). Sjogren’s syndrome related sensory motor neuropathy and autonomic neuropathy: A Case report. RRNMF Neuromuscular Journal, 1(5), 25-28. https://doi.org/10.17161/rrnmf.v1i5.14526
