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Cerebral blood flow abnormalities with central sparing on arterial spin labeling in mild encephalopathy associated
Yuki Nakajima1, Shinya Kobayashi1, Hideki Tanoue1
1Department of Pediatrics, Showa General Hospital, Tokyo, Japan.
Insights
Arterial spin labeling (ASL) may aid in the early diagnosis of mild encephalopathy with excitotoxicity (MEEX) and acute encephalopathy with biphasic seizures and late reduced diffusion (AESD). This neuroimaging technique shows unique findings in MEEX, potentially facilitating timely intervention for better patient outcomes.
Area of Science:
- Pediatric neurology
- Neuroimaging
- Neuroinflammation
Background:
- Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) and mild encephalopathy with excitotoxicity (MEEX) are common in Japanese children.
- AESD is characterized by biphasic seizures, delayed diffusion abnormalities (bright tree appearance), and altered cerebral blood flow (CBF) on arterial spin labeling (ASL).
- MEEX presents with elevated glutamine (Gln) on magnetic resonance spectroscopy (MRS) but lacks the characteristic biphasic course and diffusion abnormalities of AESD.
Observation:
- A 4-year-old girl with febrile status epilepticus initially suspected of AESD.
- ASL-MRI on day 2 revealed increased CBF in specific brain regions, suggesting AESD with central sparing.
- Subsequent clinical course and MRS findings (elevated Gln) led to a revised diagnosis of MEEX.
Findings:
- The patient diagnosed with MEEX exhibited unique ASL findings not previously reported in MEEX.
- ASL demonstrated increased CBF in the frontal, temporal, and occipital regions with central sulcus sparing.
- While initial MRI suggested AESD, later imaging lacked characteristic AESD findings, and MRS confirmed elevated Gln.
Implications:
- ASL may be a valuable tool for the early diagnosis of MEEX, similar to its utility in AESD.
- Early ASL assessment in encephalopathic children could facilitate prompt diagnosis and intervention.
- This case highlights the potential of ASL in differentiating and diagnosing acute encephalopathies in pediatric patients.
Background:
Acute encephalopathy with biphasic seizures and late reduced diffusion (AESD) and mild encephalopathy associated with excitotoxicity (MEEX) are the most frequent acute encephalopathies in pediatric patients in Japan. AESD typically presents with biphasic seizures and delayed reduced diffusion in the subcortical area, called bright tree appearance (BTA), on radiological examination. In patients with AESD, arterial spin labeling (ASL) shows decreased cerebral blood flow (CBF) in the hyperacute stage and increased CBF in the acute stage, suggesting the usefulness of ASL for the early diagnosis of AESD. Additionally, proton magnetic resonance spectroscopy (MRS) shows elevated glutamate (Glu) and glutamine (Gln) in AESD. MEEX is a group of mild encephalopathies with transient elevation of Gln on MRS similar to that in AESD; however, MEEX does not include any clinical biphasic course or abnormalities, including BTA on diffusion-weighted imaging. Although the usefulness of ASL for AESD has been reported, there are no reports for patients with MEEX. In this study, we report our experience with a 4-year-old girl diagnosed with MEEX who showed unique findings on ASL.
Case Presentation:
The patient was a 4-year-old girl admitted to the emergency room with febrile status epilepticus. Considering the possibility of AESD, vitamin therapy was initiated. ASL-MR imaging (MRI) of the brain performed on the second day showed increased blood flow in the frontal, temporal, and occipital regions with spared central sulcus, which indicated AESD with central sparing. The patient was diagnosed with AESD, and the treatment included pulse steroid therapy and immunoglobulin therapy from day 3. The patient remained mildly unconscious but gradually became conscious by day 7 with no seizures. Brain MRI performed on day 8 did not show any characteristic AESD findings, such as BTA. Furthermore, MRS showed elevated Gln, which, along with the clinical course, led to the diagnosis of MEEX. The patient was discharged on day 16 without obvious sequelae.
Conclusions:
ASL may be useful in the early diagnosis of MEEX as well as AESD, facilitating early intervention.
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