Trial reportBlood advances2020
Young adult outcomes of childhood prophylaxis for severe hemophilia A: results of the Joint Outcome Continuation Study.
Trial report in Blood advances, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT01000844 (Continuation of Children Enrolled in Protocol #95-011, 'A Randomized Prospective Study for the Prevention of Joint Disease in Children With Severe Factor VIII Deficiency'), which is not on this map. Cited by 62 papers, 4 of them syntheses that pooled 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.
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.
Continuation of Children Enrolled in Protocol #95-011, 'A Randomized Prospective Study for the Prevention of Joint Disease in Children With Severe Factor VIII Deficiency'
Who cites it
62 citing papers in PubMed, 4 syntheses or guidelines pooled it, 118 citations in OpenAlex.
- Clotting factor concentrates for preventing bleeding and bleeding-related complications in previously untreated or minimally treated children with hemophilia A or B.The Cochrane database of systematic reviews · 2025Pooled it
- Global trends in hemophilic arthropathy research: a bibliometric and visualization analysis.Frontiers in medicine · 2025Pooled it
- Risk of low bone mineral density in patients with haemophilia: a systematic review and meta-analysis.Journal of orthopaedic surgery and research · 2024Pooled it
- Factor VIII replacement prophylaxis in patients with hemophilia A transitioning to adults: a systematic literature review.Orphanet journal of rare diseases · 2021Pooled it
- Pain-Related Quality of Life Outcomes in People With Haemophilia A Receiving Emicizumab: A Post Hoc Analysis of the HAVEN 1, 3 and 4 and STASEY Studies.Haemophilia : the official journal of the World Federation of Hemophilia · 2025Trial
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- Emicizumab prophylaxis in children with hemophilia A with factor VIII inhibitors in China (HAVEN 5).Chinese medical journal · 2026Article
- [Evaluation of the efficacy and safety of reduced-dose emicizumab in infants with severe hemophilia A: a single-center real-world study].Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2026Article
- Persons with high Hemophilia Joint Health Scores have reduced functional range of motion during simulated sports activities.Research and practice in thrombosis and haemostasis · 2026Article
- Post Hoc Analyses of A-SURE and PREVENT Confirm the Effectiveness of rFVIIIFc Prophylaxis Across All Ages, BMIs, Severities, and Inhibitor Histories.TH open : companion journal to thrombosis and haemostasis · 2026Article
- Prophylaxis for von Willebrand disease: Is it time for parity with established practice in hemophilia A?Therapeutic advances in hematology · 2026Review
- Outcomes in participants switching from FVIII replacement therapy to efanesoctocog alfa prophylaxis in XTEND-1: a post hoc analysis.Therapeutic advances in hematology · 2026Article
- AOZORA study: 3-year interim analysis of safety and joint health in pediatric people with hemophilia A receiving emicizumab prophylaxis.Research and practice in thrombosis and haemostasis · 2025Article
- Point-of-care musculoskeletal ultrasound for hemophilic arthropathy: a scoping review of scanning protocols by the Imaging Expert Working Group of the International Prophylaxis Study Group.Research and practice in thrombosis and haemostasis · 2025Article
- Inhibitor development and clinical characteristics in children with severe hemophilia A in the ATHN 8 US cohort study.Blood vessels, thrombosis & hemostasis · 2025Article
- TSUBASA Study: Evaluating Association of Physical Activity and Bleeding Events in People With Haemophilia A Without Factor VIII Inhibitors Receiving Emicizumab.Haemophilia : the official journal of the World Federation of Hemophilia · 2025Observational
- An assessment of burden associated with problem joints in children and adults with moderate or severe haemophilia A: analysis of the CHESS-Paediatrics and CHESS II cross-sectional studies.Orphanet journal of rare diseases · 2025Article
- From Vision to Action: National Bleeding Disorders Foundation's Roadmap for Achieving Health Equity, Diversity, and Inclusion.Health equity · 2025Review
- Prospective, Observational Study of the Clinical Outcomes of FVIII Treatment in Adults and Adolescents with Severe Haemophilia A.TH open : companion journal to thrombosis and haemostasis · 2025Article
2 more citing papers are in PubMed but not listed here.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
13 authors at 8 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
The Joint Outcome Study (JOS), a randomized controlled trial, demonstrated that children with severe hemophilia A (HA) initiating prophylactic factor VIII (FVIII) prior to age 2.5 years had reduced joint damage at age 6 years compared with those treated with episodic FVIII for bleeding. The Joint Outcome Continuation Study (JOS-C) evaluated early vs delayed prophylaxis effects on long-term joint health, following JOS participants to age 18 years in an observational, partially retrospective study. Index joint magnetic resonance imaging (MRI) scores of osteochondral (OC) damage (primary outcome), joint physical examination scores, and annualized rates of joint/other bleeding episodes (secondary outcomes) were collected. Thirty-seven of 65 JOS participants enrolled in JOS-C, including 15 randomized to prophylaxis at mean age 1.3 years ("early prophylaxis"); 18 initially randomized to episodic treatment, starting "delayed prophylaxis" at mean age 7.5 years; and 4 with high-titer inhibitors. At JOS-C exit, MRI OC damage was found in 77% of those on delayed and 35% of those on early prophylaxis for an odds ratio of OC damage, in the delayed vs early prophylaxis group, of 6.3 (95% confidence interval, 1.3, 29.9; P = .02). Annualized bleeding rates were higher with delayed prophylaxis (mean plus or minus standard deviation, 10.6 ± 6.6 vs 3.5 ± 2.1; P < .001), including when only comparing time periods on prophylaxis (6.2 ± 5.3 vs 3.3 ± 1.9; P < .05). In severe HA, early initiation of prophylaxis provided continued protection against joint damage throughout childhood compared with delayed initiation, but early prophylaxis was not sufficient to fully prevent damage. This trial was registered at www.clinicaltrials.gov as #NCT01000844.
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Registered trials
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.