A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome

A 68-year-old Caucasian female was admitted to the emergency department with a progressive history of behavioural symptoms and anxiety followed by visual and auditory hallucinations, forgetfulness, and impaired gait in the previous 3 months. On examination she was psychotic and had a postural and re...

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Main Authors: João Peres, Simão Cruz, Rita Oliveira, Luís Santos, Ana Valverde
Format: Article
Language:English
Published: Wiley 2017-01-01
Series:Case Reports in Neurological Medicine
Online Access:http://dx.doi.org/10.1155/2017/5696512
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author João Peres
Simão Cruz
Rita Oliveira
Luís Santos
Ana Valverde
author_facet João Peres
Simão Cruz
Rita Oliveira
Luís Santos
Ana Valverde
author_sort João Peres
collection DOAJ
description A 68-year-old Caucasian female was admitted to the emergency department with a progressive history of behavioural symptoms and anxiety followed by visual and auditory hallucinations, forgetfulness, and impaired gait in the previous 3 months. On examination she was psychotic and had a postural and rest tremor of the upper limbs, cogwheel rigidity of the four limbs, retropulsion on standing position, and inability to walk. During the following 2 weeks she developed xerostomia and unilateral parotiditis that improved with steroids. A simultaneous improvement of the cognitive abilities allowed for the detection of sensory ataxia of the lower limbs. Sensory ganglionopathy was then detected with electrophysiological studies. A diagnosis of Sjögren syndrome was suspected and confirmed by salivary gland scintigraphy, Schirmer’s test, and submaxillary gland biopsy. We report a case of Sjögren syndrome associated with central and peripheral nervous system involvement, without sicca symptoms preceding the neurological clinical picture. The coexistence of ganglionopathy and a favourable response to immunosuppression are key features that can lead to the correct diagnosis in cases with atypical CNS symptoms, mimicking a rapidly progressive dementia.
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spelling doaj-art-c6cf2cf55fb549f38745f276eb29ce1c2025-08-20T02:19:41ZengWileyCase Reports in Neurological Medicine2090-66682090-66762017-01-01201710.1155/2017/56965125696512A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren SyndromeJoão Peres0Simão Cruz1Rita Oliveira2Luís Santos3Ana Valverde4Neurology Department, Hospital Professor Doutor Fernando Fonseca, Amadora, PortugalNeurology Department, Hospital Professor Doutor Fernando Fonseca, Amadora, PortugalAnatomic Pathology Department, Hospital Professor Doutor Fernando Fonseca, Amadora, PortugalNeurology Department, Hospital de Egas Moniz-Centro Hospitalar Lisboa Oeste, Lisbon, PortugalNeurology Department, Hospital Professor Doutor Fernando Fonseca, Amadora, PortugalA 68-year-old Caucasian female was admitted to the emergency department with a progressive history of behavioural symptoms and anxiety followed by visual and auditory hallucinations, forgetfulness, and impaired gait in the previous 3 months. On examination she was psychotic and had a postural and rest tremor of the upper limbs, cogwheel rigidity of the four limbs, retropulsion on standing position, and inability to walk. During the following 2 weeks she developed xerostomia and unilateral parotiditis that improved with steroids. A simultaneous improvement of the cognitive abilities allowed for the detection of sensory ataxia of the lower limbs. Sensory ganglionopathy was then detected with electrophysiological studies. A diagnosis of Sjögren syndrome was suspected and confirmed by salivary gland scintigraphy, Schirmer’s test, and submaxillary gland biopsy. We report a case of Sjögren syndrome associated with central and peripheral nervous system involvement, without sicca symptoms preceding the neurological clinical picture. The coexistence of ganglionopathy and a favourable response to immunosuppression are key features that can lead to the correct diagnosis in cases with atypical CNS symptoms, mimicking a rapidly progressive dementia.http://dx.doi.org/10.1155/2017/5696512
spellingShingle João Peres
Simão Cruz
Rita Oliveira
Luís Santos
Ana Valverde
A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
Case Reports in Neurological Medicine
title A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
title_full A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
title_fullStr A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
title_full_unstemmed A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
title_short A Case Report of Nonvasculitic Autoimmune Inflammatory Meningoencephalitis with Sensory Ganglionopathy: A Rare Presentation of Sjögren Syndrome
title_sort case report of nonvasculitic autoimmune inflammatory meningoencephalitis with sensory ganglionopathy a rare presentation of sjogren syndrome
url http://dx.doi.org/10.1155/2017/5696512
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