Evidence map›Paper›PMID 41645108›Full record

ReviewBMC pediatrics2026

CASPR2 antibody-related neurological syndromes in children: three cases report and literature review.

Long-Ying Peng, Shu-Jing Pan, Ren-Ke Li, Juan Li, Jing Chen, Xiao-Hua Yu, Chang-Jian Yang, Xiao-Mei Shu, Mao-Qiang Tian

Abstract readReviewCase Reports
In one paragraph

Review in BMC pediatrics, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Long-Ying Peng *Department of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Shu-Jing Pan *Department of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Ren-Ke LiDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Juan LiDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Jing ChenDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Xiao-Hua YuDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Chang-Jian YangDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Xiao-Mei ShuDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China.
Mao-Qiang TianDepartment of Pediatrics, Affiliated Hospital of Zunyi Medical University, Zunyi, China. drmaoqiang@126.com.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundContactin-associated protein-like 2 (CASPR2) antibody-related neurological syndrome is well defined in adults, while data in children is rare. We perform this case series and literature review to ascertain the clinical features of this disorder in children. CASE PRESENTATIONS: We describe three cases who were diagnosed with CASPR2 antibody-related neurological syndrome, and then systematically characterized clinical features, therapeutic responses, and outcomes in 64 patients in literature. Among the 67 patients, the median age was 8.4 years (range 0.5–18), with a male predominance (65.7%). The most characteristic symptoms of CASPR2 antibody-related neurological syndrome include psychiatric abnormalities (68.7%), sleep disorders (55.2%), dysautonomia (44.8%), movement disorders (43.3%), seizures (41.8%), neuropathic pain (34.3%), and cognitive disturbances (31.3%). Autoimmune encephalitis (AE, 62.7%) was most frequent, followed by Morvan syndrome (MoS, 26.9%) and peripheral nerve hyperexcitability (PNH, 7.5%). CASPR2 antibodies are typically detected in serum (85.1%), but rarely in CSF exclusively (3.0%). Investigations revealed abnormalities in CSF analysis (22.9%; 13 AE, 5 MoS), brain MRI (24.1%; 11 AE, 3 MoS), and EEG (67.3%; 26 AE, 7 MoS). Neoplasm was not found in our case series and literature review. Regarding treatment, 46 patients received first-line immunotherapy alone, while 5 required second-line treatment. Immunotherapy demonstrated highly effective, with 95.8% achieving a favorable outcome (mRS score ≤ 2) and a low recurrence rate of 5.1%.

conclusionsWe therefore recommend that all children presenting with new-onset psychiatric abnormalities, sleep disorders, dysautonomia, movement disorders, seizures, neuropathic pain, and cognitive disturbances should be suspected of having CASPR2 antibody-related neurological syndrome. Neoplastic processes are rare in pediatric patients. Early recognition of the disease is important because it responds well to immunotherapy.

Indexed as

AutoantibodiesMembrane ProteinsNerve Tissue ProteinsNervous System DiseasesAdolescentChildChild, PreschoolFemaleHumansInfantMaleSyndromeAutoantibodiesCNTNAP2 protein, humanMembrane ProteinsNerve Tissue ProteinsAutoimmune encephalitisCASPR2ChildrenMorvan syndromePeripheral nerve hyperexcitability

Identifiers

PMID41645108
PMCPMC12990615

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.