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Plasma exchange in pediatric anti-NMDAR encephalitis: A systematic review
Agnese Suppiej1, Margherita Nosadini1, Luigi Zuliani2
1Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.
Insights
Plasma exchange (PE) for pediatric anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis is increasingly used with steroids and IVIG. Early PE administration with steroids shows a trend toward better outcomes.
Area of Science:
- Neurology
- Immunology
- Pediatrics
Background:
- Pediatric anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis is a severe autoimmune neurological disorder.
- Plasma exchange (PE) is an extracorporeal treatment used to remove antibodies from the blood.
- Understanding the optimal use of PE in pediatric anti-NMDAR encephalitis is crucial for improving patient outcomes.
Purpose of the Study:
- To determine the common methods of using plasma exchange (PE) in pediatric anti-NMDAR encephalitis.
- To identify the most effective combinations of PE with other immunotherapies for treating this condition.
Main Methods:
- A systematic literature review was conducted on studies published between 2007 and 2015.
- The review focused on pediatric cases of anti-NMDAR encephalitis treated with PE.
- Data from 71 articles involving 242 pediatric patients were analyzed.
Main Results:
- Plasma exchange was frequently administered with corticosteroids and intravenous immunoglobulin (IVIG) (69.5%) or corticosteroids alone (18%).
- PE was often used as a second or third-line treatment, with only 14.3% of cases receiving it as first-line therapy.
- Outcomes showed a trend towards better recovery when PE was initiated within 30 days of symptom onset and when combined with corticosteroids.
Conclusions:
- There is a limited amount of high-quality data regarding the use of PE in pediatric anti-NMDAR encephalitis.
- The use of PE in this condition is increasing, commonly alongside steroids and IVIG.
- Evidence suggests that early PE administration, particularly with steroids, may be associated with improved patient outcomes.
Objective:
To clarify the most frequent modalities of use of plasma exchange (PE) in pediatric anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis and to establish the most effective association with other immunotherapies.
Methods:
Systematic literature review on PE in pediatric anti-NMDAR encephalitis (2007-2015).
Results:
Seventy-one articles were included (mostly retrospective), reporting a total of 242 subjects (73.2%, 93/127 females; median age at onset 12years, range 1-18). Median time to immunotherapy was 21days (range 0-190). In most cases, PE was given with steroids and IVIG (69.5%, 89/128), or steroids only (18%, 23/128); in a minority, it was associated with IVIG only (7%, 9/128), or was the only first-line treatment (5.5%, 7/128). In 54.5% (65/119), PE was the third treatment after steroids and IVIG, in 31.1% (37/119) the second after steroids or IVIG; only in 14.3% (17/119) was it the first treatment. Second-line immunotherapies were administered in 71.9% (100/139). Higher rates of full/substantial recovery at follow-up were observed with immunotherapy given ⩽30days from onset (69.4%, 25/36) compared to later (59.2%, 16/27), and when PE was associated with steroids (66.7%, 70/105) rather than not (46.7%, 7/15). Significant adverse reactions to PE were reported in 6 patients.
Conclusion:
Our review disclosed a paucity of quality data on PE in pediatric anti-NMDAR encephalitis. PE use in this condition has been increasingly reported, most often with steroids and IVIG. Despite the limited number of patients, our data seem to confirm the trend towards a better outcome when PE was administered early, and when given with steroids.
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