Proportion and spectrum of movement disorders in adolescent and adult patients of autoimmune encephalitis of non-neoplastic aetiology

Dash, Deepa; Ihtisham, Kavish; Tripathi, Madhavi

doi:10.1016/j.jocn.2018.10.076

Cited by 11 publications

(10 citation statements)

References 12 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The population of South Asia accounts for about 40% of Asia's population and about one quarter of the world's population. However, research on AE from this region remains sparse and limited mostly to case reports or series amounting to less than 200 cases among a 2 billion population [6][7][8][9]. Sri Lanka is an island of 65,610 km 2 , situated just south of the Indian subcontinent between northern latitudes 5 o to 10 o with a population of approximately 22 million.…”

Section: Introductionmentioning

confidence: 99%

Autoimmune encephalitis in a South Asian population

et al. 2021

View full text Add to dashboard Cite

Background Autoimmune encephalitis (AE) is now considered a main, potentially curable cause of encephalitis, but remains conspicuously underreported from South Asia. We studied the clinical characteristics in relation to their antibody status and outcomes of patients presenting with AE in Sri Lanka. Methods Patients admitting to government hospitals who were clinically suspected of AE by an on-site neurologist were prospectively recruited over a period of 12 months. Sera and cerebrospinal fluid were tested for NMDAR, AMPAR1, AMPAR2, LGI1, CASPR2, GABARB1/B2 antibodies (Ab) using commercial cell-based assays. Demographic, clinical and laboratory data were compiled into an investigator-administered proforma. Patients were reviewed at 1 year follow up either in person or via telephone. Results One-hundred and forty-two patients from 21 of 25 districts in Sri Lanka (median age = 20.5 years; range 1–86 years; females = 61.3%) were recruited. Of them, 65 (45.8%; median age = 19 years; range 1–86 years; females = 64.6%) fulfilled diagnostic criteria for probable NMDAR-antibody encephalitis (NMDARE) and 6 (4.2%; median age = 44 years; range 28–71 years; females = 83.3%) limbic encephalitis (LE). Abnormal behaviour (95.3%), seizures (81.5%) and movement disorders (69.2%) were the most frequent clinical manifestations of probable NMDARE. NMDAR-antibodies were detectable in 29 (44.6%) and not detectable in 36 in CSF of probable-NMDARE patients. Abnormal EEG was more frequent (p = 0.003) while a worse outcome (OR = 2.78; 95% CI = 0.88–9.09) and deaths (OR = 2.38; 95% CI = 0.67–8.33) were more likely in antibody-negative than antibody-positive probable-NMDARE. Most patients with LE had amnesia (50%) and/or confusion (100%) with agitation (83.3%) and seizures (100%) but none had detectable antibodies to any of the antigens tested. Conclusions NMDARE is the commonest type of AE among South Asians as is the case worldwide. Clinical presentations of NMDARAb-positive and NMDARAb-negative AE patients do not significantly differ but EEG may be a useful marker of an autoimmune basis for psychiatric symptoms.

show abstract

Section: Introductionmentioning

confidence: 99%

Autoimmune encephalitis in a South Asian population

et al. 2021

View full text Add to dashboard Cite

show abstract

“…Aetiologies are likely to be multiple. Motor stereotypies have been reported in several neurological disorders, namely, frontotemporal dementia (Mendez, Shapira, & Miller, 2005; Prioni et al, 2018; Rascovsky et al, 2011), progressive supranuclear palsy and Parkinson’s disease with dementia (Prioni et al, 2012; Prioni et al, 2018), strokes involving basal ganglia (Kulisevsky, Berthier, Avila, & Roig, 1996; Maraganore, Lees, & Marsden, 1991; Yasuda, Akiguchi, Ino, Nabatabe, & Kameyama, 1990), and antibody NMDAR positive autoimmune encephalitis (Baizabal-Carvallo & Jankovic, 2018; Dash, Ihtisham, Tripathi, & Tripathi, 2019). Family history of motor stereotypies has been reported with a range from 17% to 39% (K.…”

Section: Introductionmentioning

confidence: 99%

Prevalence and determinants of motor stereotypies in autism spectrum disorder: A systematic review and meta-analysis

Melo

Ruano

Jorge

et al. 2019

Autism

View full text Add to dashboard Cite

Stereotypies are frequently reported in people with autism spectrum disorder (ASD) but remain one of the less explained phenomena. We aimed to describe, through a systematic review and a meta-analysis, the prevalence of motor stereotypies in ASD and study the factors that influence this prevalence. Our literature search included MEDLINE, Scopus, and PsycINFO databases. Quality and risk of bias were assessed. Thirty-seven studies were included and the median prevalence of motor stereotypies in ASD was 51.8%, ranging from 21.9% to 97.5%. The most frequent determinants associated with a higher number of stereotypies in ASD were a younger age, lower intelligence quotient, and a greater severity of ASD. Moreover, gender did not seem to influence the prevalence of stereotypies. Meta-analytic analysis showed that lower IQ and autism diagnosis (independent of IQ) are associated with a higher prevalence of motor stereotypies (odds ratio = 2.5 and 4.7, respectively). Limitations of the reviewed literature include the use of convenience samples, with small sizes and heterogeneous inclusion criteria, and the predominance of high-functioning autism individuals.

show abstract

“…The present case is unique given the rare etiology of chronic tremor syndrome. Most tremor seen in patients with infectious and autoimmune encephalitis 8,9,18) is transient, self-limiting, and subsides with treatment of the underlying disorders. To review cases of atypical chronic tremor after encephalitis, a literature search on MEDLINE was completed using the title/abstract-tagged keywords "tremor AND encephalitis."…”

Section: Discussionmentioning

confidence: 99%

Thalamic Deep Brain Stimulation for Refractory Atypical Tremor after Encephalitis of Unknown Etiology: A Case Report

Nakajima

Toda

Nishida

et al. 2021

NMC Case Report Journal

View full text Add to dashboard Cite

Tremor associated with encephalitis is usually transient and rarely becomes chronic and refractory. Treatment for such tremor using deep brain stimulation (DBS) has not yet been reported. We report an uncommon case of chronic tremor after encephalitis of unknown etiology and its outcome treated with thalamic DBS. A 47-year-old man presented with a 6-month history of medically refractory tremor after non-infectious and probable autoimmune encephalitis. The patient showed an atypical mixture of resting, postural, kinetic, and intention tremor. The tremor significantly disabled the patient's activities of daily life (ADL). The patient underwent bilateral thalamic DBS surgery. DBS leads were placed to cross the border between the ventralis oralis posterior (Vop) nucleus and ventralis intermedius (Vim) nucleus of the thalamus. Stimulation of both the Vop and Vim using the bipolar contacts controlled the mixed occurrence of tremor. The ADL and performance scores on The Essential Tremor Rating Assessment Scale (TETRAS) improved from 47 to 0 and from 44 to 9, respectively. The therapeutic effects have lasted for 24 months. Administration of combined Vop and Vim DBS may control uncommon tremor of atypical etiology and phenomenology.

show abstract

Proportion and spectrum of movement disorders in adolescent and adult patients of autoimmune encephalitis of non-neoplastic aetiology

Cited by 11 publications

References 12 publications

Autoimmune encephalitis in a South Asian population

Autoimmune encephalitis in a South Asian population

Prevalence and determinants of motor stereotypies in autism spectrum disorder: A systematic review and meta-analysis

Thalamic Deep Brain Stimulation for Refractory Atypical Tremor after Encephalitis of Unknown Etiology: A Case Report

Contact Info

Product

Resources

About