Evidence map›Paper›PMID 35514529›Full record

ArticleTherapeutic advances in neurological disorders2022

Improving pediatric multiple sclerosis interventional phase III study design: a meta-analysis.

Jennifer S Graves, Marius Thomas, Jun Li, Anuja R Shah, Alexandra Goodyear, Markus R Lange, Heinz Schmidli, Dieter A Häring, Tim Friede, Jutta Gärtner

Abstract read
In one paragraph

Article in Therapeutic advances in neurological disorders, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed, 1 pooled it
–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

3 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Therapeutic Advances in Pediatric Multiple Sclerosis.Children (Basel, Switzerland) · 2025
    Review
  3. 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

10 authors.

Jennifer S GravesDepartment of Neurosciences, University of California, San Diego, Box 0662 ACTRI, 9452 Medical Center Drive, Suite 4W-222, San Diego, CA 92037, USA.
Marius ThomasNovartis Pharma AG, Basel, Switzerland.ORCID https://orcid.org/0000-0003-2790-829X
Jun LiNovartis Pharma AG, Basel, Switzerland.
Anuja R ShahNovartis Healthcare Pvt. Ltd., Hyderabad, India.
Alexandra GoodyearNovartis Pharmaceuticals Corporation, East Hanover, NJ, USA at the time of article development.
Markus R LangeNovartis Pharma AG, Basel, Switzerland.
Heinz SchmidliNovartis Pharma AG, Basel, Switzerland.
Dieter A HäringNovartis Pharma AG, Basel, Switzerland.
Tim FriedeDepartment of Medical Statistics, University Medical Center Göttingen, Göttingen, Germany.ORCID https://orcid.org/0000-0001-5347-7441
Jutta GärtnerDepartment of Pediatrics and Adolescent Medicine, German Center for Multiple Sclerosis in Childhood and Adolescence, University Medical Center Göttingen, Göttingen, Germany.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Background: To support innovative trial designs in a regulatory setting for pediatric-onset multiple sclerosis (MS), the study aimed to perform a systematic literature review and meta-analysis of relapse rates with interferon β (IFN β), fingolimod, and natalizumab and thereby demonstrate potential benefits of Bayesian and non-inferiority designs in this population. Methods: We conducted a literature search in MEDLINE and EMBASE from inception until 17 June 2020 of all studies reporting annualized relapse rates (ARR) in IFN β-, fingolimod-, or natalizumab-treated patients with pediatric-onset relapsing-remitting MS. These interventions were chosen because the literature was mainly available for these treatments, and they are currently used for the treatment of pediatric MS. Two researchers independently extracted data and assessed study quality using the Cochrane Effective Practice and Organization of Care - Quality Assessment Tool. The meta-analysis estimates were obtained by Bayesian random effects model. Data were summarized as ARR point estimates and 95% credible intervals. Results: We found 19 articles, including 2 randomized controlled trials. The baseline ARR reported was between 1.4 and 3.7. The meta-analysis-based ARR was significantly higher in IFN β-treated patients (0.69, 95% credible interval: 0.51-0.91) versus fingolimod (0.11, 0.04-0.27) and natalizumab (0.17, 0.09-0.31). Based on the meta-analysis results, an appropriate non-inferiority margin versus fingolimod could be in the range of 2.29-2.67 and for natalizumab 1.72-2.29 on the ARR ratio scale. A Bayesian design, which uses historical information for a fingolimod or natalizumab control arm, could reduce the sample size of a new trial by 18 or 14 patients, respectively. Conclusion: This meta-analysis provides evidence that relapse rates are considerably higher with IFNs versus fingolimod or natalizumab. The results support the use of innovative Bayesian or non-inferiority designs to avoid exposing patients to less effective comparators in trials and bringing new medications to patients more efficiently.

Indexed as

annualized relapse rateclinical trial designfingolimodinterferonnatalizumabpediatric-onset multiple sclerosissystematic review

Identifiers

PMID35514529
PMCPMC9066624

What Socratic holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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 Socratic graph.