Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma

Objectives. Sjögren’s syndrome (SS) may be complicated by some neurological manifestations, generally sensory polyneuropathy. Furthermore, involvement of cranial nerves was described as rare complications of SS. Methods. We reported 2 cases: the first one was a 40-year-old woman who developed neurit...

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Main Authors: Michele Colaci, Giulia Cassone, Andreina Manfredi, Marco Sebastiani, Dilia Giuggioli, Clodoveo Ferri
Format: Article
Language:English
Published: Wiley 2014-01-01
Series:Case Reports in Neurological Medicine
Online Access:http://dx.doi.org/10.1155/2014/590292
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author Michele Colaci
Giulia Cassone
Andreina Manfredi
Marco Sebastiani
Dilia Giuggioli
Clodoveo Ferri
author_facet Michele Colaci
Giulia Cassone
Andreina Manfredi
Marco Sebastiani
Dilia Giuggioli
Clodoveo Ferri
author_sort Michele Colaci
collection DOAJ
description Objectives. Sjögren’s syndrome (SS) may be complicated by some neurological manifestations, generally sensory polyneuropathy. Furthermore, involvement of cranial nerves was described as rare complications of SS. Methods. We reported 2 cases: the first one was a 40-year-old woman who developed neuritis of the left optic nerve as presenting symptom few years before the diagnosis of SS; the second was a 54-year-old woman who presented a paralysis of the right phrenic nerve 7 years after the SS onset. An exhaustive review of the literature on patients with cranial or phrenic nerve involvements was also carried out. Results. To the best of our knowledge, our second case represents the first observation of SS-associated phrenic nerve mononeuritis, while optic neuritis represents the most frequent cranial nerve involvement detectable in this connective tissue disease. Trigeminal neuropathy is also frequently reported, whereas neuritis involving the other cranial nerves is quite rare. Conclusions. Cranial nerve injury is a harmful complication of SS, even if less commonly recorded compared to peripheral neuropathy. Neurological manifestations may precede the clinical onset of SS; therefore, in patients with apparently isolated cranial nerve involvement, a correct diagnosis of the underlying SS is often delayed or overlooked entirely; in these instances, standard clinicoserological assessment is recommendable.
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spelling doaj-art-197c7eb0a8e04558aff012ebda3f8c622025-02-03T06:01:18ZengWileyCase Reports in Neurological Medicine2090-66682090-66762014-01-01201410.1155/2014/590292590292Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic DilemmaMichele Colaci0Giulia Cassone1Andreina Manfredi2Marco Sebastiani3Dilia Giuggioli4Clodoveo Ferri5Chair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyChair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyChair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyChair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyChair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyChair and Rheumatology Unit, Medical School, Azienda Ospedaliero-Universitaria, University of Modena and Reggio Emilia, Policlinico di Modena, Via del Pozzo 71, 41100 Modena, ItalyObjectives. Sjögren’s syndrome (SS) may be complicated by some neurological manifestations, generally sensory polyneuropathy. Furthermore, involvement of cranial nerves was described as rare complications of SS. Methods. We reported 2 cases: the first one was a 40-year-old woman who developed neuritis of the left optic nerve as presenting symptom few years before the diagnosis of SS; the second was a 54-year-old woman who presented a paralysis of the right phrenic nerve 7 years after the SS onset. An exhaustive review of the literature on patients with cranial or phrenic nerve involvements was also carried out. Results. To the best of our knowledge, our second case represents the first observation of SS-associated phrenic nerve mononeuritis, while optic neuritis represents the most frequent cranial nerve involvement detectable in this connective tissue disease. Trigeminal neuropathy is also frequently reported, whereas neuritis involving the other cranial nerves is quite rare. Conclusions. Cranial nerve injury is a harmful complication of SS, even if less commonly recorded compared to peripheral neuropathy. Neurological manifestations may precede the clinical onset of SS; therefore, in patients with apparently isolated cranial nerve involvement, a correct diagnosis of the underlying SS is often delayed or overlooked entirely; in these instances, standard clinicoserological assessment is recommendable.http://dx.doi.org/10.1155/2014/590292
spellingShingle Michele Colaci
Giulia Cassone
Andreina Manfredi
Marco Sebastiani
Dilia Giuggioli
Clodoveo Ferri
Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
Case Reports in Neurological Medicine
title Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
title_full Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
title_fullStr Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
title_full_unstemmed Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
title_short Neurologic Complications Associated with Sjögren’s Disease: Case Reports and Modern Pathogenic Dilemma
title_sort neurologic complications associated with sjogren s disease case reports and modern pathogenic dilemma
url http://dx.doi.org/10.1155/2014/590292
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