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Adult-onset Still's disease with concurrent acute necrotizing encephalopathy: a case report
Xue Yang1, Meiling Wei2, Shuguang Chu3
1Department of Rheumatology, Huashan Hospital, Fudan University, Shanghai, China.
BMC Neurology
|September 1, 2022
Summary
This case report details a rare instance of acute necrotizing encephalopathy (ANE) following adult-onset Still's disease (AOSD). Early diagnosis of these conditions is challenging but crucial for patient outcomes.
Area of Science:
- Neurology
- Rheumatology
- Pathology
Background:
- Acute necrotizing encephalopathy (ANE) presents with symmetrical brain lesions, primarily in the thalami.
- Adult-onset Still's disease (AOSD) is a systemic inflammatory disorder with fever, rash, and joint pain.
- Both ANE and AOSD may be triggered by infections and involve hypercytokinemia.
Observation:
- A 46-year-old male diagnosed with AOSD developed sudden unconsciousness.
- Brain imaging revealed symmetrical lesions in the thalami, striatum, and brainstem, indicative of ANE.
- Hemorrhage occurred within the lesions shortly after admission, leading to the patient's death.
Findings:
- This report describes a rare case of ANE occurring after AOSD.
- The exact pathological mechanism linking AOSD and ANE remains undetermined.
- Diagnostic challenges exist in recognizing these conditions early.
Implications:
- Highlights the potential neurological complications of AOSD.
- Emphasizes the need for heightened clinical suspicion for ANE in AOSD patients.
- Underscores the prognostic significance of early diagnosis in managing these rare conditions.
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