Dystonia; A central nervous system presentation of Sj�gren's syndrome

Berg, Jan S.P. van den; Horstink, M.W.I.M.; Hoogen, F.H.J. van den; Oyen, Wim J.G.

doi:10.1002/1531-8257(199903)14:2<374::aid-mds1032>3.0.co;2-w

Cited by 24 publications

(18 citation statements)

References 15 publications

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“…Autoimmune-mediated movement disorders are thought to occur in a number of autoimmune disorders, such as Sjögren’s Syndrome (van den Berg et al 1999; Venegas et al 2005; Papageorgiou et al 2007; Min and Youn 2009), Sydenham's chorea (Vincent et al 2011), and certain cases of Tourette’s Syndrome (Morer et al 2008; Dehning et al 2009). Methodologies currently exist that may provide clinically useful means to image neuroinflammation in these and other disorders.…”

Section: Introductionmentioning

confidence: 99%

Neuroinflammation, Neuroautoimmunity, and the Co-Morbidities of Complex Regional Pain Syndrome

Cooper

Clark

2012

J Neuroimmune Pharmacol

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Complex Regional Pain Syndrome (CRPS) is associated with non-dermatomal patterns of pain, unusual movement disorders, and somatovisceral dysfunctions. These symptoms are viewed by some neurologists and psychiatrists as being psychogenic in origin. Recent evidence, however, suggests that an autoimmune attack on self-antigens found in the peripheral and central nervous system may underlie a number of CRPS symptoms. From both animal and human studies, evidence is accumulating that neuroinflammation can spread, either anterograde or retrograde, via axonal projections in the CNS, thereby establishing neuroinflammatory tracks and secondary neuroinflammatory foci within the neuraxis. These findings suggest that neuroinflammatory lesions, as well as their associated functional consequences, should be evaluated during the differential diagnosis of non-dermatomal pain presentations, atypical movement disorders, as well as other “medically unexplained symptoms”, which are often attributed to psychogenic illness.

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Section: Introductionmentioning

confidence: 99%

Neuroinflammation, Neuroautoimmunity, and the Co-Morbidities of Complex Regional Pain Syndrome

Cooper

Clark

2012

J Neuroimmune Pharmacol

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“…Additionally, the associated collective central and peripheral involvement, such as neurogenic bladder, autonomic dysfunction and sensory neuropathy, which is more frequently reported, was chronologically matching the presence of dystonia, and preceding the appearance of the SS manifestations7 This is the first reported case that describes the development of neurological symptoms involving primary dystonia prior to sicca syndrome and positive serology. van der Berg et al 8 reported the first patient with SS and dystonia. This patient presented with painless fixed dystonic posture of the left hand with hyperflexion of the left third and fifth fingers and wrist.…”

Section: Discussionmentioning

confidence: 99%

Case of primary Sjogren’s syndrome preceded by dystonia

Ararat

Berrios²,

Hannoun

et al. 2018

BMJ Case Reports

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There are only six cases in literature that describe development of dystonia with Sjogren's syndrome (SS). We describe a case of a 43-year-old woman who presented with symptoms including movement disorder, sensory neurogenic bladder, sensory loss and neuropathic pain, migraine like headaches, musculoskeletal pain, Raynaud's phenomenon and dysautonomia. Symptoms started in 2000, with weakness that progressed to dystonia in 2003. Diagnostic work-up was inconclusive with negative inflammatory serologies, cerebrospinal fluid and MRI for many years. After patient developed sicca syndrome with dry eyes and mouth in 2009, her rheumatoid factor titre was elevated (550 IU/mL), erythrocyte sedimentation rate, anti-Sjogrens syndrome-related antigen A (anti-Ro/SSA) and anti-SSB/La: anti-Sjogrens syndrome-related antigen B (anti-La/SSB) became positive. Lip biopsy confirmed diagnosis of SS. She was diagnosed with primary SS with neurological involvement. Her symptoms responded well to intravenous methylprednisolone. Symptoms stabilised with trials of immune-suppressive therapy. This is a case that demonstrates the delay of diagnosing SS with preceding unique neurological association.

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“…Further neurological examination showed no abnormalities by electromyography (EMG) or magnetic resonance imaging (MRI) of the brain. However, single photon emission computed tomography (SPECT) with iodobenzamide (IBZM) labelled with iodine-123 revealed decreased uptake in the right striatum (striatum:occipital cortex ratio 1.53 on the left, 1.41 on the right, mean normal (2SD) value 1.66 (0.16)), indicating a loss of D2 receptors in the right striatum (neurological details have been published previously) 12. A diagnosis of neurovasculitis located in the right striatum was made, and was considered to be an extraglandular complication of SS.…”

Section: Case Reportmentioning

confidence: 90%

Reversibility of histological and immunohistological abnormalities in sublabial salivary gland biopsy specimens following treatment with corticosteroids in Sjogren's syndrome

Zandbelt¹,

Hoogen²,

Wilde³

et al. 2001

Annals of the Rheumatic Diseases

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Sjögren's syndrome (SS) is a chronic autoimmune disease characterised by specific lesions in exocrine glands, so sublabial minor salivary gland biopsy (SLGB) plays an important part in its diagnosis. The extent and composition of the lymphocytic infiltrate in SLGB specimens can be considered as target organ specific parameters. They are quantified after histological and immunohistological examination by a focus score (describing the extent of the infiltrate) and IgA% score (describing the composition of the infiltrate), respectively. However, little is known about the factors that contribute to the extent and composition of the infiltrate and whether these features are reversible as repeated SLGBs are rarely performed. A patient with SS is described who underwent SLGBs before and after treatment with high dose corticosteroids. After treatment there was not only clinical improvement, but also improvement in the histological and immunohistological parameters. Although these findings need to be confirmed in further studies, this suggests that histopathological changes may be reversible in SS. Furthermore, it shows that the potential eVects of corticosteroid use should be taken into account when interpreting SLGB specimens. When clinical changes do parallel histological changes, repeated SLGBs might oVer a marker for disease activity in patients with SS.

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Dystonia; A central nervous system presentation of Sj�gren's syndrome

Cited by 24 publications

References 15 publications

Neuroinflammation, Neuroautoimmunity, and the Co-Morbidities of Complex Regional Pain Syndrome

Neuroinflammation, Neuroautoimmunity, and the Co-Morbidities of Complex Regional Pain Syndrome

Case of primary Sjogren’s syndrome preceded by dystonia

Reversibility of histological and immunohistological abnormalities in sublabial salivary gland biopsy specimens following treatment with corticosteroids in Sjogren's syndrome

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