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      • Record: found
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      Is Open Access

      Live‐attenuated vaccines in a cryopyrin‐associated periodic syndrome patient receiving canakinumab treatment during infancy

      case-report

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          Key Clinical Message

          We successfully immunized the neonatal‐onset multisystem inflammatory disease ( NOMID) patient with live‐attenuated vaccines for measles, rubella, varicella, and mumps and achieved sufficient antibody titer under canakinumab therapy without complications.

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          Most cited references18

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          Familial autoinflammatory diseases: genetics, pathogenesis and treatment.

          Silvia Stojanov, D Kastner (2005)
          The systemic autoinflammatory diseases are characterized by seemingly unprovoked inflammation, without major involvement of the adaptive immune system. This review focuses mainly on a subset of these illnesses, the hereditary recurrent fevers, which include familial Mediterranean fever, the tumor necrosis factor receptor-associated periodic syndrome, the hyperimmunoglobulinemia D with periodic fever syndrome, and cryopyrin-associated periodic syndromes. This review elucidates how recent advances have impacted diagnosis, pathogenesis, and treatment. More than 170 mutations have been identified in the four genes underlying the six hereditary recurrent fevers. Genetic testing has broadened the clinical and geographic boundaries of these illnesses, given rise to the concept of the cryopyrin-associated periodic syndromes as a disease spectrum, and permitted diagnosis of compound heterozygotes for mutations in two different hereditary recurrent fever genes. Genetics has also advanced our understanding of amyloidosis, a complication of the hereditary recurrent fevers, and suggested a possible role for common hereditary recurrent fever variants in other inflammatory conditions. Recent advances in molecular pathophysiology include the elucidation of the N-terminal PYRIN domain in protein-protein interactions, the description of the NALP3 (cryopyrin) inflammasome as a macromolecular complex for interleukin-1beta activation, and the identification of signaling defects other than defective receptor shedding in patients with tumor necrosis factor receptor-associated periodic syndrome. These molecular insights form the conceptual basis for targeted biologic therapies. Advances in molecular genetics extend our ability to recognize and treat patients with systemic autoinflammatory diseases and inform our understanding of the regulation of innate immunity in humans.
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            Pattern of interleukin-1beta secretion in response to lipopolysaccharide and ATP before and after interleukin-1 blockade in patients with CIAS1 mutations.

            Marco Gattorno, Sara Tassi, Sonia Carta … (2007)
            To examine the synthesis, processing, and secretion of interleukin-1beta (IL-1beta), as well as the clinical and biologic effects of IL-1 blockade, in patients with chronic infantile neurologic, cutaneous, articular (CINCA) syndrome and Muckle-Wells syndrome (MWS), in an effort to understand the molecular mechanisms linking mutations of the CIAS1 gene and IL-1beta hypersecretion, and the underlying response to IL-1 receptor antagonist (IL-1Ra). Six patients with CINCA syndrome or MWS were treated with IL-1Ra and followed up longitudinally. Monocytes obtained from the patients and from 24 healthy donors were activated with lipopolysaccharide (LPS) for 3 hours, and intracellular and secreted IL-1beta levels were determined by Western blotting and enzyme-linked immunosorbent assay before and after exposure to exogenous ATP. LPS-induced IL-1beta secretion was markedly increased in monocytes from patients with CIAS1 mutations. However, unlike in healthy subjects, secretion of IL-1beta was not induced by exogenous ATP. Treatment with IL-1Ra resulted in a dramatic clinical improvement, which was paralleled by an early and strong down-regulation of LPS-induced IL-1beta secretion by the patients' cells in vitro. Our results showed that the requirements of ATP stimulation for IL-1beta release observed in healthy individuals are bypassed in patients bearing CIAS1 mutations. This indicates that cryopyrin is the direct target of ATP and that the mutations release the protein from the requirement of ATP for activation. In addition, the dramatic amelioration induced by IL-1Ra treatment is at least partly due to the strong decrease in IL-1beta secretion that follows the first injections of the antagonist. These findings may have implications for other chronic inflammatory conditions characterized by increased IL-1beta.
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              EULAR recommendations for vaccination in paediatric patients with rheumatic diseases.

              Leonardo Pileggi, Marloes Heijstek, A Fasth … (2011)
              Evidence-based recommendations for vaccination of paediatric patients with rheumatic diseases (PaedRD) were developed by following the EULAR standardised procedures for guideline development. The EULAR task force consisted of (paediatric) rheumatologists/immunologists, one expert in vaccine evaluation, one expert in public health and infectious disease control, and one epidemiologist. A systematic literature review was conducted in MEDLINE, EMBASE, and abstracts of the EULAR and American College of Rheumatology meetings of 2008/9. The level of evidence and strength of recommendation were based on customary scoring systems. Delphi voting was applied to assess the level of agreement between task force members. 107 papers and eight abstracts were used. The majority of papers considered seasonal influenza (41) or pneumococcal (23) vaccination. 26 studies were performed specifically in paediatric patients, and the majority in adult rheumatoid arthritis and systemic lupus erythematosus patients. Fifteen recommendations were developed with an overall agreement of 91.7%. More research is needed on the safety and immunogenicity of (live-attenuated) vaccination in PaedRD, particularly in those using biologicals, and the effect of vaccination on prevention of infections.
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                Author and article information

                Contributors
                Misa Watanabe:
                ORCID: http://orcid.org/0000-0001-5856-9628
                misa-watanabe@med.toho-u.ac.jp
                Journal
                Journal ID (nlm-ta): Clin Case Rep
                Journal ID (iso-abbrev): Clin Case Rep
                Journal ID (doi): 10.1002/(ISSN)2050-0904
                Journal ID (publisher-id): CCR3
                Title: Clinical Case Reports
                Publisher: John Wiley and Sons Inc. (Hoboken )
                ISSN (Electronic): 2050-0904
                Publication date (Electronic): 12 September 2017
                Publication date Collection: November 2017
                Volume: 5
                Issue: 11 ( doiID: 10.1002/ccr3.2017.5.issue-11 )
                Pages: 1750-1755
                Affiliations
                [ 1 ] Department of Pediatrics Toho University School of Medicine Ota‐ku Tokyo Japan
                [ 2 ] Department of Pediatrics Graduate School of Medicine Kyoto University Kyoto Japan
                [ 3 ] Advanced and Integrated Cardiovascular Research Course in the Young and Adolescence Tokyo Japan
                Author notes
                [*] [* ] Correspondence

                Misa Watanabe, Department of Pediatrics, Toho University School of Medicine, 6‐11‐1, Omori‐Nishi, Ota‐ku, Tokyo 143‐8541, Japan. Tel: 703‐3762‐4151; Fax: 703‐3298‐8217; E‐mail: misa-watanabe@ 123456med.toho-u.ac.jp

                Author information
                http://orcid.org/0000-0001-5856-9628
                Article
                Publisher ID: CCR31149
                DOI: 10.1002/ccr3.1149
                PMC ID: 5676279
                SO-VID: ca199cdd-1185-42a1-bd47-04cd183f013b
                Copyright © © 2017 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd.
                License:

                This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 30 April 2017
                Date revision received : 29 July 2017
                Date accepted : 05 August 2017
                Page count
                Figures: 2, Tables: 1, Pages: 6, Words: 3325
                Categories
                Subject: Case Report
                Subject: Case Reports
                Custom metadata
                source-schema-version-number 2.0
                component-id ccr31149
                cover-date November 2017
                details-of-publishers-convertor Converter:WILEY_ML3GV2_TO_NLMPMC version:5.2.1 mode:remove_FC converted:08.11.2017

                Keywords: canakinumab,cryopyrin‐associated periodic syndrome,infant,live‐attenuated vaccine,neonatal‐onset multisystem inflammatory disease
                Data availability:
                Keywords: canakinumab, cryopyrin‐associated periodic syndrome, infant, live‐attenuated vaccine, neonatal‐onset multisystem inflammatory disease

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