ArticleBlood advances2018
Allogeneic hematopoietic stem cell transplantation for severe, refractory juvenile idiopathic arthritis.
Article in Blood advances, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 29 papers, 2 of them syntheses that pooled it.
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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.
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Who cites it
29 citing papers in PubMed, 2 syntheses or guidelines pooled it, 60 citations in OpenAlex.
- Indications for haematopoietic cell transplantation and CAR-T for haematological diseases, solid tumours and immune disorders: 2025 EBMT practice recommendations.Bone marrow transplantation · 2025Guideline
- Current treatment in macrophage activation syndrome worldwide: a systematic literature review to inform the METAPHOR project.Rheumatology (Oxford, England) · 2025Pooled it
- Allogeneic HSCT for Autoimmune Diseases: A Retrospective Study From the EBMT ADWP, IEWP, and PDWP Working Parties.Frontiers in immunology · 2019Trial
- Outcomes of patients with hematological malignancies complicated with autoimmune diseases after allogeneic hematopoietic stem cell transplantation: A ten-year real-world study.Journal of translational internal medicine · 2026Article
- Case Report: Telitacicept in the treatment of refractory juvenile idiopathic arthritis: clinical experience from three cases.Frontiers in pediatrics · 2026Article
- The evolving landscape of rheumatic drugs and their indications in paediatric and adolescent care.Rheumatology (Oxford, England) · 2025Review
- Allogeneic haematopoietic stem-cell transplantation for children with refractory systemic juvenile idiopathic arthritis and associated lung disease: outcomes from an international, retrospective cohort study.The Lancet. Rheumatology · 2025Article
- A rapid ecologic analysis, confirmed by a case-control study, identifies class I HLA alleles correlated to the risk of COVID-19.Journal of translational medicine · 2025Article
- Advances in the mechanisms, imaging characteristics and management strategies for immune checkpoint inhibitor-related pneumonitis.Frontiers in immunology · 2025Review
- Pharmacological and non-pharmacological treatments for refractory paediatric Still's disease: a scoping review.Rheumatology advances in practice · 2025Review
- Treating and triggering hyperinflammation: tackling hemophagocytic lymphohistiocytosis and HLH-like syndromes in the pediatric cell therapy and critical care setting.Frontiers in oncology · 2025Review
- Review
- Patterns and determinants of response to novel therapies in juvenile and adult-onset polyarthritis.Rheumatology (Oxford, England) · 2024Review
- Part 5: Allogeneic HSCT in refractory SJIA with lung disease; recent cases from centers in North America & Europe.Pediatric rheumatology online journal · 2024Article
- Case report: Success of allogeneic hematopoietic stem cell transplantation for refractory systemic-onset juvenile idiopathic arthritis.Frontiers in medicine · 2023Article
- Systemic juvenile idiopathic arthritis: The Great Ormond Street Hospital experience (2005-2021).Frontiers in pediatrics · 2023Article
- Case report: Emapalumab for active disease control prior to hematopoietic stem cell transplantation in refractory systemic juvenile idiopathic arthritis complicated by macrophage activation syndrome.Frontiers in pediatrics · 2023Article
- Refractory systemic onset juvenile idiopathic arthritis: current challenges and future perspectives.Annals of medicine · 2022Review
- Systemic Juvenile Idiopathic Arthritis/Pediatric Still's Disease, a Syndrome but Several Clinical Forms: Recent Therapeutic Approaches.Journal of clinical medicine · 2022Article
- Autologous ATG-free hematopoietic stem cell transplantation for refractory autoimmune rheumatic diseases: a Latin American cohort.Clinical rheumatology · 2022Article
Corrections and comments
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Authors and funding
25 authors at 10 institutions in 3 countries.
Funding
Abstract
Patients with juvenile idiopathic arthritis (JIA) can experience a severe disease course, with progressive destructive polyarthritis refractory to conventional therapy with disease-modifying antirheumatic drugs including biologics, as well as life-threatening complications including macrophage activation syndrome (MAS). Allogeneic hematopoietic stem cell transplantation (allo-HSCT) is a potentially curative immunomodulatory strategy for patients with such refractory disease. We treated 16 patients in 5 transplant centers between 2007 and 2016: 11 children with systemic JIA and 5 with rheumatoid factor-negative polyarticular JIA; all were either refractory to standard therapy, had developed secondary hemophagocytic lymphohistiocytosis/MAS poorly responsive to treatment, or had failed autologous HSCT. All children received reduced toxicity fludarabine-based conditioning regimens and serotherapy with alemtuzumab. Fourteen of 16 patients are alive with a median follow-up of 29 months (range, 2.8-96 months). All patients had hematological recovery. Three patients had grade II-IV acute graft-versus-host disease. The incidence of viral infections after HSCT was high, likely due to the use of alemtuzumab in already heavily immunosuppressed patients. All patients had significant improvement of arthritis, resolution of MAS, and improved quality of life early following allo-HSCT; most importantly, 11 children achieved complete drug-free remission at the last follow-up. Allo-HSCT using alemtuzumab and reduced toxicity conditioning is a promising therapeutic option for patients with JIA refractory to conventional therapy and/or complicated by MAS. Long-term follow-up is required to ascertain whether disease control following HSCT continues indefinitely.
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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.