ReviewEuropean journal of pediatrics2015
Treatment of hyperimmunoglobulinemia D syndrome with biologics in children: review of the literature and Finnish experience.
Review in European journal of pediatrics, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers, 2 of them syntheses that pooled it.
What it found
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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.
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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
18 citing papers in PubMed, 2 syntheses or guidelines pooled it, 37 citations in OpenAlex.
- Mevalonate kinase deficiency: an updated clinical overview and revision of the SHARE recommendations.Frontiers in immunology · 2024Pooled it
- Consensus protocols for the diagnosis and management of the hereditary autoinflammatory syndromes CAPS, TRAPS and MKD/HIDS: a German PRO-KIND initiative.Pediatric rheumatology online journal · 2020Guideline
- Tocilizumab effectively reduces flares of hyperimmunoglobulin D syndrome in children: Three cases in China.Molecular genetics and metabolism reports · 2024Article
- A case report of Hyper-IgD syndrome in a 5-year-old girl with recurrent fever, skin rash, and arthralgia; novel MVK mutation (Clinical case reports · 2024Article
- Efficacy of Anakinra Treatment in two Moroccan Patients With Mevalonate Kinase Deficiency.Global pediatric health · 2023Article
- AAV-monoclonal antibody expression protects mice from Ebola virus without impeding the endogenous antibody response to heterologous challenge.Molecular therapy. Methods & clinical development · 2022Article
- Twists and turns of the genetic story of mevalonate kinase-associated diseases: A review.Genes & diseases · 2022Review
- Management of Monogenic IL-1 Mediated Autoinflammatory Diseases in Childhood.Frontiers in immunology · 2021Review
- Real-Life Indications of Interleukin-1 Blocking Agents in Hereditary Recurrent Fevers: Data From the JIRcohort and a Literature Review.Frontiers in immunology · 2021Review
- The off-label use of anakinra in pediatric systemic autoinflammatory diseases.Therapeutic advances in musculoskeletal disease · 2020Review
- A case report of mevalonate kinase deficiency in a 14-month-old female with fevers and lower extremity weakness.BMC pediatrics · 2019Article
- Monogenic Periodic Fever Syndromes: Treatment Options for the Pediatric Patient.Paediatric drugs · 2017Review
- Hyperimmunoglobulinaemia D syndrome: a rare cause of prolonged fever and treatment with anti-interleukin 1 agent.BMJ case reports · 2016Article
- Natural history of mevalonate kinase deficiency: a literature review.Pediatric rheumatology online journal · 2016Review
- Perinatal manifestation of mevalonate kinase deficiency and efficacy of anakinra.Pediatric rheumatology online journal · 2016Review
- Observational Study of a French and Belgian Multicenter Cohort of 23 Patients Diagnosed in Adulthood With Mevalonate Kinase Deficiency.Medicine · 2016Observational
- Article
- Targeting the inflammasome in rheumatic diseases.Translational research : the journal of laboratory and clinical medicine · 2016Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
4 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
unlabelledHyperimmunoglobulinemia D syndrome (HIDS) is an autoinflammatory disorder that is caused by mevalonate kinase deficiency (MKD). Recent advances in the pathogenesis of MKD, including the proposed mechanisms of inflammasome activation, provide the basis for the development of new treatment modalities. So far, feedback on the treatment of HIDS with biological medicines has come from case reports with limited numbers of patients. In this review, we summarize the data that is currently available on the treatment of HIDS in children, with the emphasis on new therapies, and present three Finnish pediatric cases treated with anakinra. Case reports have been published on 33 pediatric HIDS patients who have been treated with biological medicines, and in some cases, they were treated with more than one drug. Of these patients, 21 were treated with anakinra and 16 with etanercept, resulting in complete or partial responses in 90 and 50% of cases, respectively. A further five patients were treated with canakinumab, with complete or partial responses.
conclusionThe accumulating evidence on the efficacy and safety of biological drugs in pediatric HIDS suggests that the anti-interleukin-1 agent anakinra is the drug of choice for HIDS in children. WHAT IS KNOWN: • Various biologic drugs have been tried for the treatment of HIDS. What is New: • Based on the 90% response rate, anakinra seems to be the drug of choice for HIDS in children.
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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.