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2000
Volume 21, Issue 1
  • ISSN: 1573-4056
  • E-ISSN: 1875-6603

Abstract

Introduction/Background:

Cytotoxic lesions of the corpus callosum (CLOCC) are a rare clinical-radiological syndrome with an unclear specific pathogenesis, and cases occurring consecutively in siblings are exceptionally uncommon. This study reports two pediatric siblings with CLOCC (one experiencing two episodes), highlighting the potential role of genetic susceptibility in its pathogenesis. The findings contribute to the limited literature on familial CLOCC and recurrent cases in children.

Case Presentation:

Two brothers (aged 9 and 12) presented with sudden-onset aphasia and unilateral limb weakness, preceded by rhinorrhea. Magnetic resonance imaging (MRI) revealed reversible lesions in the splenium of the corpus callosum and bilateral frontoparietal white matter, consistent with CLOCC. Both patients received immunomodulatory therapy (, corticosteroids, intravenous immunoglobulin) and symptomatic treatment, achieving full neurological recovery within approximately one week. The elder sibling had a recurrence two years later (when the patient was 14 years old) with similar imaging findings. Laboratory tests ruled out common infections, and cerebrospinal fluid analysis was unremarkable.

Conclusion:

This case underscores CLOCC as a heterogeneous condition with possible genetic predisposition, as evidenced by its occurrence in siblings and recurrence in one sibling. While prognosis is generally favorable, the observed sibling clustering and individual recurrence suggest the need for further research into underlying genetic or immunological mechanisms.

This is an open access article published under CC BY 4.0 https://creativecommons.org/licenses/by/4.0/legalcode
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2025-10-03
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  • Article Type:
    Research Article
Keyword(s): Brain diseases; Case reports; Corpus callosum; Encephalitis; Siblings
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