LGI1 and CASPR2 autoimmunity in children: Systematic literature review and report of a young girl with Morvan syndrome.

Autor: Nosadini M; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy; Neuroimmunology Group, Paediatric Research Institute 'Città della Speranza', Padova, Italy. Electronic address: margherita.nosadini@gmail.com., Toldo I; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy., Tascini B; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy., Bien CG; Epilepsy Center Bethel, Krankenhaus Mara, Bielefeld, Germany; Laboratory Krone, Bad Salzuflen, Germany., Parmeggiani L; Department of Neuropediatrics, Regional Hospital of Bolzano, Bolzano, Italy., De Gaspari P; Neuroimmunology Group, Paediatric Research Institute 'Città della Speranza', Padova, Italy., Zuliani L; Department of Neurology, Ospedale San Bortolo, AULSS8 Berica, Vicenza, Italy; Neuroimmunology Group, Paediatric Research Institute 'Città della Speranza', Padova, Italy., Sartori S; Paediatric Neurology and Neurophysiology Unit, Department of Women's and Children's Health, University Hospital of Padua, Italy; Neuroimmunology Group, Paediatric Research Institute 'Città della Speranza', Padova, Italy.
Jazyk: angličtina
Zdroj: Journal of neuroimmunology [J Neuroimmunol] 2019 Oct 15; Vol. 335, pp. 577008. Date of Electronic Publication: 2019 Jul 18.
DOI: 10.1016/j.jneuroim.2019.577008
Abstrakt: Leucine-rich glioma-inactivated protein 1 (LGI1) and contactin-associated protein-like 2 (CASPR2) neurological autoimmunity in adults has been associated with various clinical syndromes involving central, peripheral and autonomic nervous system, while data in children is limited. We perform the first systematic literature review on paediatric LGI1 and CASPR2 autoimmunity, with focus on clinical data, in order to contribute to the definition of clinical features of LGI1 and CASPR2 autoimmunity in paediatric age and favour early diagnosis. Additionally, we report the youngest-to-date case of Morvan syndrome. We identified 37 published paediatric cases of LGI1 and/or CASPR2 autoimmunity. Most frequent syndromes were encephalitis in LGI1-positive and isolated epilepsy in CASPR2-positive children, while syndromes with predominant peripheral symptoms were most frequent in double-positive children. With the limitations imposed by the low number of cases, differences to published adult cohorts included: absence of faciobrachial dystonic seizures and hyponatremia in patients with LGI1-positive encephalitis; slightly higher proportion of isolated epilepsy syndromes in CASPR2-positive patients; absence of tumour in the whole cohort.
(Copyright © 2019 Elsevier B.V. All rights reserved.)
Databáze: MEDLINE
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