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Plasma exchange in pediatric anti-NMDAR encephalitis: A systematic review.
Brain Dev. 2016 Aug; 38(7):613-22.BD

Abstract

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.

Authors+Show Affiliations

Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy. Electronic address: suppiej@pediatria.unipd.it.Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.Department of Neurology, Ospedale Ca' Foncello, Treviso, Italy.Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.Immunotransfusion Section, University Hospital of Padua, Italy.Department of Neurology, Ospedale Ca' Foncello, Treviso, Italy.Immunotransfusion Section, University Hospital of Padua, Italy.Department of Neurology, Ospedale Ca' Foncello, Treviso, Italy.Neuroimmunology group, Institute for Neuroscience and Muscle Research, Kids Research Institute at the Children's Hospital at Westmead, University of Sydney, Australia.Pediatric Neurology Unit, Department of Woman's and Child's Health, University Hospital of Padua, Italy.

Pub Type(s)

Journal Article
Review
Systematic Review

Language

eng

PubMed ID

26926399

Citation

Suppiej, Agnese, et al. "Plasma Exchange in Pediatric anti-NMDAR Encephalitis: a Systematic Review." Brain & Development, vol. 38, no. 7, 2016, pp. 613-22.
Suppiej A, Nosadini M, Zuliani L, et al. Plasma exchange in pediatric anti-NMDAR encephalitis: A systematic review. Brain Dev. 2016;38(7):613-22.
Suppiej, A., Nosadini, M., Zuliani, L., Pelizza, M. F., Toldo, I., Bertossi, C., Tison, T., Zoccarato, M., Marson, P., Giometto, B., Dale, R. C., & Sartori, S. (2016). Plasma exchange in pediatric anti-NMDAR encephalitis: A systematic review. Brain & Development, 38(7), 613-22. https://doi.org/10.1016/j.braindev.2016.01.009
Suppiej A, et al. Plasma Exchange in Pediatric anti-NMDAR Encephalitis: a Systematic Review. Brain Dev. 2016;38(7):613-22. PubMed PMID: 26926399.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Plasma exchange in pediatric anti-NMDAR encephalitis: A systematic review. AU - Suppiej,Agnese, AU - Nosadini,Margherita, AU - Zuliani,Luigi, AU - Pelizza,Maria Federica, AU - Toldo,Irene, AU - Bertossi,Chiara, AU - Tison,Tiziana, AU - Zoccarato,Marco, AU - Marson,Piero, AU - Giometto,Bruno, AU - Dale,Russell C, AU - Sartori,Stefano, Y1 - 2016/02/28/ PY - 2015/10/16/received PY - 2016/01/19/revised PY - 2016/01/25/accepted PY - 2016/3/2/entrez PY - 2016/3/2/pubmed PY - 2017/1/14/medline KW - Anti-NMDAR KW - Apheresis KW - Children KW - Encephalitis KW - Immune therapy KW - Plasma exchange KW - Plasmapheresis SP - 613 EP - 22 JF - Brain & development JO - Brain Dev VL - 38 IS - 7 N2 - 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. SN - 1872-7131 UR - https://www.unboundmedicine.com/medline/citation/26926399/Plasma_exchange_in_pediatric_anti_NMDAR_encephalitis:_A_systematic_review_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S0387-7604(16)00026-7 DB - PRIME DP - Unbound Medicine ER -