ArticleInternational ophthalmology2016
Successful treatment with adalimumab for severe multifocal choroiditis and panuveitis in presumed (early-onset) ocular sarcoidosis.
Article in International ophthalmology, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 17 papers, 2 of them syntheses that pooled it.
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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.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
17 citing papers in PubMed, 2 syntheses or guidelines pooled it, 28 citations in OpenAlex.
- Neurosarcoidosis in children: A systematic review and summary of cases, imaging and management.Journal of neuroimmunology · 2022Pooled it
- Anti-TNF Drugs for Chronic Uveitis in Adults-A Systematic Review and Meta-Analysis of Randomized Controlled Trials.Frontiers in medicine · 2019Pooled it
- Phenotype of Takayasu-like vasculitis and cardiopathy in patients with Blau syndrome.Clinical rheumatology · 2024Article
- Biologic Therapies for the Management of Cutaneous Findings in Genodermatoses: A Review.American journal of clinical dermatology · 2022Review
- Potential Benefits of TNF Targeting Therapy in Blau Syndrome, a NOD2-Associated Systemic Autoinflammatory Granulomatosis.Frontiers in immunology · 2022Review
- A novel mutation in early-onset sarcoidosis/Blau syndrome: an association with Propionibacterium acnes.Pediatric rheumatology online journal · 2021Article
- Distinguishing Blau Syndrome from Systemic Sarcoidosis.Current allergy and asthma reports · 2021Review
- A young female with early onset arthritis, uveitis, hepatic, and renal granulomas: a clinical tryst with Blau syndrome over 20 years and case-based review.Rheumatology international · 2021Review
- Blau Syndrome and Early-Onset Sarcoidosis: A Six Case Series and Review of the Literature.Archives of rheumatology · 2020Article
- Periodic fever syndromes and the autoinflammatory diseases (AIDs).Journal of translational autoimmunity · 2020Review
- Effective treatment of TNFα inhibitors in Chinese patients with Blau syndrome.Arthritis research & therapy · 2019Review
- Pediatric Sarcoidosis: A Review with Emphasis on Early Onset and High-Risk Sarcoidosis and Diagnostic Challenges.Diagnostics (Basel, Switzerland) · 2019Review
- Familial Blau syndrome:First molecularly confirmed report from India.Indian journal of ophthalmology · 2019Article
- A Case of Sporadic Blau Syndrome with an Uncommon Clinical Course.Case reports in rheumatology · 2018Article
- Adalimumab for the treatment of refractory noninfectious paediatric uveitis.International ophthalmology · 2017Article
- Blau Syndrome (Juvenile Systemic Granulomatosis): State-Of-The-Art Review.Pediatric dermatologyReview
- Multimodal imaging in pediatric uveitis.Therapeutic advances in ophthalmologyReview
Corrections and comments
- Erratum issued
- Erratum issued
Authors and funding
8 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Early-onset sarcoidosis (EOS) and Blau syndrome are rare auto-inflammatory diseases characterized by a triad of skin rash, granulomatous uveitis, and symmetrical polyarthritis occurring in early childhood. In this paper, we describe a case report very interesting for the multidisciplinary management (pediatric rheumatologist and ophthalmologist), the challenging diagnosis and the difficult choice of the best treatment. We describe a case report of an 8-year old with recurrent episodes of acute uveitis that developed bilateral granulomatous panuveitis initially treated with topical and systemic steroids. Genetic testing for NOD2/CARD15 revealed a heterozygous mutation on exon 4 in the NBD domain (P268S/SNP5). Therefore, an incomplete EOS was suspected. Because uveitis worsening with multifocal chorioretinitis aggravation, intravenous boluses of methylprednisolone were administered. During the steroids tapering, she flared again, and methotrexate was started along with corticosteroids pulse therapy. However, new ocular granuloma appeared, macular oedema with poor visual outcome occurred, and therefore, adalimumab was added to MTX and steroids. After 6 months since the new therapy started, she had a complete visual recovery, and she was able to stop steroid treatment. At 2 years of follow-up, she is still in remission on treatment, and her visual acuity is normal. No side effects were observed. In our patient, we found a heterozygous mutation on exon 4 in the NBD domain (P268S/SNP5) of NOD2/CARD15 gene and an incomplete EOS was hypothesized. The role of this variant is currently under study. Adalimumab use dramatically changed the course of eye disease, prompting to stop steroid treatment and preserving visual acuity.
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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.