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Paediatric antibody prevalence in seizure score to predict autoimmune aetiology in seizure disorders
Takayuki Mori1, Shimpei Matsuda1, Hiroya Nishida1
1Department of Brain & Neurosciences, Tokyo metropolitan Institute of Medical Science, Tokyo, Japan.
Insights
A new Paediatric Antibody Prevalence in Seizure (PAPS) score improves diagnosis for children with autoimmune central nervous system disease. This score offers better specificity than the Antibody Prevalence in Epilepsy (APE) score for identifying antibody needs.
Area of Science:
- Neurology
- Immunology
- Pediatrics
Background:
- Autoimmune central nervous system diseases can present with seizures in children.
- Accurate identification of antineuronal antibodies is crucial for diagnosis and treatment.
- Existing scoring systems, like the Antibody Prevalence in Epilepsy (APE) score, may have limitations in pediatric populations.
Purpose of the Study:
- To modify the Antibody Prevalence in Epilepsy (APE) score for children with suspected autoimmune central nervous system disease and seizures.
- To develop and validate a new scoring system, the Paediatric Antibody Prevalence in Seizure (PAPS) score, to aid in antineuronal antibody testing decisions.
Main Methods:
- Retrospective analysis of cerebrospinal fluid from 157 children (0-18 years) with suspected autoimmune CNS disease.
- Development of the PAPS score using a development cohort (n=79) and validation in a separate cohort (n=78).
- Selection of seven variables through univariate and multivariate analysis to construct the PAPS score.
Main Results:
- The PAPS score demonstrated a sensitivity of 83.3% and specificity of 77.8% for detecting antineuronal antibodies.
- The APE score showed high sensitivity (92%) but low specificity (22.2%).
- The PAPS score achieved a significantly better area under the curve (0.832) compared to the APE score (p < 0.001).
Conclusions:
- The existing APE score has high sensitivity but limited specificity in pediatric epilepsy cases.
- The newly developed PAPS score shows improved diagnostic performance and may be a valuable tool for guiding antineuronal antibody testing in children.
- The PAPS score offers a more reliable method for determining the necessity of antibody testing in pediatric autoimmune CNS disorders.
Aim:
To modify the antibody prevalence in epilepsy (APE) score of children with suspected autoimmune central nervous system disease with seizures.
Methods:
We retrospectively analysed the cerebrospinal fluid of 157 children (aged 0-18 years) with suspected autoimmune central nervous system disease for antineuronal antibodies in our laboratory from 2016 to 2023. Participants were randomly divided into the development cohort (n = 79, 35 females; median 7 years, SD 4 years 7 months, range 4-11 years) and validation cohort (n = 78, 28 females; median 7 years, SD 4 years 5 months, range 4-12 years). A paediatric antibody prevalence in seizure (PAPS) score was created for one cohort and evaluated in the other. Seven variables were selected through univariate and multivariate analysis to create a PAPS score.
Results:
One hundred and fifty-seven children who fulfilled the inclusion criteria were enrolled; 49 tested positive for antineuronal antibodies. The sensitivity and specificity of an APE score of 4 and greater were 92% and 22.2% respectively; the sensitivity and specificity of a PAPS score of 2.5 and greater were 83.3% and 77.8% respectively. The area under the curve was 0.832 (95% confidence interval = 0.743-0.921), which was significantly better than that for the APE score (p < 0.001).
Interpretation:
The APE score had high sensitivity but low specificity in children. The PAPS score may be useful for determining the need for antineuronal antibody testing.
What This Paper Adds:
The antibody prevalence in epilepsy (APE) score had high sensitivity but low specificity in children. The sensitivity and specificity of the paediatric antibody prevalence in seizure (PAPS) score were 83.3% and 77.8% respectively. There were significant differences in the area under the curve for the APE and PAPS scores.
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