ArticleTherapeutic advances in musculoskeletal disease2022
Clinical and immunological study of Tofacitinib and Baricitinib in refractory Blau syndrome: case report and literature review.
Article in Therapeutic advances in musculoskeletal disease, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.
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Who cites it
11 citing papers in PubMed, 16 citations in OpenAlex.
- Noninfectious uveitis: Management with biologic agents.Indian journal of ophthalmology · 2026Review
- Clinical features, treatment strategies, and long-term outcomes of Blau syndrome: a 10-year experience from a Chinese cohort.Advances in rheumatology (London, England) · 2026Observational
- A case report: Guillain-Barré syndrome probably associated with TNF inhibitor in Blau syndrome.BMC pediatrics · 2026Article
- Mechanisms outpacing medicine in autoinflammatory diseases.Frontiers in immunology · 2026Review
- Janus kinase inhibitors in pulmonary and extra-pulmonary sarcoidosis: A case series and a systematic review of the literature.Sarcoidosis, vasculitis, and diffuse lung diseases : official journal of WASOG · 2025Article
- Potential benefits of JAK inhibitor therapy in Blau syndrome: a case report.Frontiers in immunology · 2025Article
- Phenotype of Takayasu-like vasculitis and cardiopathy in patients with Blau syndrome.Clinical rheumatology · 2024Article
- A peptide mimic of SOCS1 modulates equine peripheral immune cellsFrontiers in immunology · 2024Article
- Uveitis Associated with Monogenic Autoinflammatory Syndromes in Children.Ocular immunology and inflammation · 2023Review
- Blau syndrome with hypertension and hepatic granulomas: a case report and literature review.Frontiers in pediatrics · 2023Article
- Tofacitinib in uveitis and scleritis.Oman journal of ophthalmologyReview
Corrections and comments
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Authors and funding
12 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Blau syndrome (BS) is an autoinflammatory disorder characterized by non-caseating granulomatous dermatitis, arthritis, and uveitis. We present a case of refractory and severe BS that was treated with the Janus kinase inhibitors (JAKINIBS), Tofacitinib (TOFA) and then Baricitinib (BARI). Our aim was to describe the clinical and immunological outcomes after treatment with JAKINIBS. Blood tests and serum samples were obtained during follow-up with TOFA and BARI. We assessed their effects on clinical outcomes, acute phase reactants, absolute lymphocyte counts (ALCs), lymphocyte subset counts, immunoglobulins, and cytokine levels. A review of the literature on the use of JAKINIBS for the treatment of uveitis and sarcoidosis was also conducted. TOFA led to a rapid and maintained disease control and a steroid-sparing effect. A decrease from baseline was observed in ALC, CD3+, CD4+, CD8+, and natural killer (NK) cell counts. B-cells were stable. Serum levels of interleukin (IL)-4 and tumor necrosis factor alpha (TNF-α) increased, whereas IL-2, IL-6, IL-10, and IL-17 maintained stable. TOFA was discontinued after 19 months due to significant lymphopenia. The initiation of BARI allowed maintaining adequate control of disease activity with an adequate safety profile. The literature review showed seven patients with uveitis and five with sarcoidosis treated with JAKINIBS. No cases of BS treated with JAKINIBS were found. We report the successful use of JAKINIBS in a patient with refractory and severe BS.
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