Literature DB >> 35851655

Autoimmune parkinsonism with faciobrachiocrural dystonic seizures: a new phenotype of leucine-rich glioma-inactivated 1 (LGI1) autoimmunity.

Sruthi Sasikumar1, Jino Vincent1, Siby Gopinath1, Vivek Nambiar1, Saraf Udit Umesh1, Sudheeran Kannoth2,3, Dayana Antony1,4, Annamma Mathai1,4, Anandkumar Anandakuttan1.   

Abstract

Extrapyramidal symptoms are seen in patients with leucine-rich glioma-inactivated 1 (LGI1) antibody-positive patients infrequently and this can be successfully treated with immunotherapy. This is a retrospective hospital-based study from 2013 to 2021 at a tertiary care referral hospital in South India. LGI1 antibody-positive cases with Faciobrachio-crural dystonic seizures [FBCDS] were identified by reviewing electronic medical records and Neuroimmunology laboratory register. Clinical and laboratory details and treatment outcomes were analysed. There was a total of 23 patients who were positive for LGI1 antibody. Of these, three cases had FBCDS (2 males, age range 30-76 years). Upon reviewing the records they had additional asymmetric parkinsonian features. All had similar presentations with progressive slowness of activities and gait and later went on to have paroxysmal events of sudden falls with vocalization. Prolonged VEEG monitoring captured the habitual event, which were confirmed to be FBCDS. MRI did not show significant structural abnormalities, CSF showed elevated proteins and normal cell in two and lymphocytic pleocytosis in one, PET scans ruled out malignancy. Of the three patients, two were completely relieved of FBCDS with immunosuppression and there was complete resolution of extrapyramidal features in all. Thus, the patients in our series of FBCDS showed additional features of parkinsonism which responded well to immunotherapy. Involvement of basal ganglia can explain all the manifestations of this phenotype. This series reveals a unique phenotype of the LGI1 antibody.
© 2022. The Author(s) under exclusive licence to Belgian Neurological Society.

Entities:  

Keywords:  Autoimmune parkinsonism; Extrapyramidal syndrome; Faciobrachial crural dystonic seizures; Faciobrachial crural dystonic seizures with parkinsonism; Leucine-rich glioma-inactivated 1 (LGI1) antibodies.

Year:  2022        PMID: 35851655     DOI: 10.1007/s13760-022-02018-8

Source DB:  PubMed          Journal:  Acta Neurol Belg        ISSN: 0300-9009            Impact factor:   2.471


  1 in total

1.  Autoimmune Encephalitis versus Creutzfeldt-Jakob disease in a patient with typical Facio-brachial dystonic seizures: A case report with Diagnostic challenges.

Authors:  Vaibhav Seth; Suman Kushwaha; Ritu Verma; Priyankkumar Mukeshbhai Patel; R Kiran Gowda; Prateek Bapat
Journal:  Brain Behav Immun Health       Date:  2021-03-13
  1 in total

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