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Kikuchi-Fujimoto Disease With Encephalopathy in Children: Case Reports and Literature Review
Yu-Ting Pan1, Li-Ming Cao2, Yan Xu1
1Department of Rheumatology and Immunology, Children's Hospital of Nanjing Medical University, Nanjing, China.
Insights
Kikuchi-Fujimoto disease (KFD) can cause encephalopathy in children. Early diagnosis via CSF analysis and MRI, followed by immunomodulatory therapy, improves outcomes for pediatric KFD with encephalopathy.
Area of Science:
- Pediatric Neurology
- Infectious Diseases
- Immunology
Background:
- Kikuchi-Fujimoto disease (KFD) is a rare, self-limiting condition causing lymphadenitis and fever.
- Encephalopathy is an uncommon but serious neurological complication of KFD in children.
- This study reviews pediatric KFD with encephalopathy cases and literature.
Observation:
- Neurological symptoms in pediatric KFD with encephalopathy emerged 10 days to 3 months after KFD onset.
- Cerebrospinal fluid (CSF) analysis revealed elevated protein levels, with other labs typically normal.
- Brain MRI showed characteristic hyperintense T2/FLAIR signals in various brain regions.
Findings:
- Diagnosis relies on CSF analysis and brain MRI due to non-specific early symptoms.
- Brain MRI findings include supratentorial white matter, deep gray matter, and brainstem involvement.
- Glucocorticoids and immunoglobulin therapy showed potential efficacy.
Implications:
- Early diagnosis of KFD with encephalopathy is crucial for timely intervention.
- Immunomodulatory therapy can significantly improve prognosis in affected children.
- Further research into the pathophysiology and optimal treatment of KFD-associated encephalopathy is warranted.
Abstract:
Background: Kikuchi-Fujimoto disease (KFD) is a benign and self-limiting disease characterized by regional lymphadenitis and low-grade fever. Encephalopathy may present in children with KFD. We present three cases of KFD with encephalopathy in children and a literature review. Methods: Literature published between 2010 and 2020 was reviewed to understand the clinical features, laboratory findings, and treatments for encephalopathy occurring in children with KFD. Results: The interval between KFD and onset of neurological symptoms was 10 days to 3 months. Laboratory results were normal, except for high protein levels in cerebrospinal fluid findings. Brain magnetic resonance imaging (MRI) findings include hyperintense T2 and FLAIR signal in the supratentorial white matter, deep gray matter, brain stem, cerebellum, temporal lobes, pons, and basal ganglia. Glucocorticoids and immunoglobulin could be effective for treating KFD with encephalopathy. Conclusion: The early clinical manifestations of KFD with encephalopathy in children lack specificity, and the diagnosis is mainly based on CSF analysis and brain MRI findings. Early and timely immunomodulatory therapy is effective and can improve the prognosis of patients with KFD with encephalopathy.
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