Evidence map›Paper›PMID 30077992›Full record

ArticleAnnals of the rheumatic diseases2018

In silico validation of the Autoinflammatory Disease Damage Index.

Nienke M Ter Haar, Amber Laetitia Justine van Delft, Kim Valerie Annink, Henk van Stel, Sulaiman M Al-Mayouf, Gayane Amaryan, Jordi Anton, Karyl S Barron, Susanne Benseler, Paul A Brogan and 41 more

Abstract readValidation Study
PubMed Full text at PMC Publisher
In one paragraph

Article in Annals of the rheumatic diseases, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
20citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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.

2 · The registry

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.

3 · Its place in the literature

Who cites it

20 citing papers in PubMed, 1 synthesis or guideline pooled it.

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  20. Familial Mediterranean Fever: Assessing the Overall Clinical Impact and Formulating Treatment Plans.Mediterranean journal of hematology and infectious diseases · 2019
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

51 authors.

Nienke M Ter HaarLaboratory for Translational Immunology, University Medical Centre Utrecht, Utrecht, The Netherlands.
Amber Laetitia Justine van DelftDepartment of Paediatrics, Universitair Medisch Centrum Utrecht-Locatie Wilhelmina Kinderziekenhuis, Utrecht, The Netherlands.
Kim Valerie AnninkDepartment of Paediatrics, Universitair Medisch Centrum Utrecht-Locatie Wilhelmina Kinderziekenhuis, Utrecht, The Netherlands.
Henk van StelDepartment of Paediatrics, Universitair Medisch Centrum Utrecht-Locatie Wilhelmina Kinderziekenhuis, Utrecht, The Netherlands.
Sulaiman M Al-MayoufDepartment of Pediatrics, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.
Gayane AmaryanNational Paediatric Centre for Familial Mediterranean Fever and Gastroenterology Service, Arabkir Medical Centre-Institute of Child and Adolescent Health, Yerevan, Armenia.
Jordi AntonPaediatric Rheumatology Unit, Hospital Sant Joan de Déu, Barcelona, Spain.
Karyl S BarronDivision of Intramural Research and National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, Maryland, USA.
Susanne BenselerDepartments of Paediatrics and Rheumatology, Alberta Children's Hospital, Calgary, Alberta, Canada.
Paul A BroganDepartment of Infection, Inflammation and Rheumatology, University College London Institute of Child Health, London, UK.
Luca CantariniRheumatology Unit, Department of Medical Sciences, Surgery and Neurosciences, University of Siena, Siena, Italy.
Marco CattaliniPaediatric Clinic, University of Brescia and Spedali Civili di Brescia, Brescia, Italy.
Alexis-Virgil CochinoDepartment of Paediatrics, National Institute for Mother and Child Health Alessandrescu-Rusescu, Bucharest, Romania.
Fabrizio de BenedettiDivision of Rheumatology, Ospedale Pediatrico Bambino Gesù, Rome, Italy.
Fatma DedeogluRheumatology Program, Division of Immunology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Adriana Almeida de JesusTranslational Autoinflammatory Disease Section, NIAMS/NIH, Bethesda, Maryland, USA.
Erkan DemirkayaWestern University Children's Hospital, London Health Sciences Centre, London, UK.
Pavla DolezalovaDepartment of Paediatrics and Adolescent Medicine, Charles University, General University Hospital, Praha, Czech Republic.
Karen L DurrantAutoinflammatory Alliance, San Francisco, California, USA.
Giovanna FabioDepartment of Internal Medicine, Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy.
Romina GallizziDepartment of Paediatric Rheumatology, AOUG Martino, Messina, Italy.
Raphaela Goldbach-ManskyTranslational Autoinflammatory Disease Section, NIAMS/NIH, Bethesda, Maryland, USA.
Eric HachullaDépartement de Médecine Interne et Immunologie Clinique, Université de Lille, Lille, France.
Veronique HentgenReference Centre for Autoinflammatory Diseases (CEREMAI), Versailles Hospital, Le Chesnay, France.
Troels HerlinDepartmentof Paediatrics, Aarhus University Hospital, Aarhus, Denmark.
Michaël HoferDepartment of Paediatric Rheumatology, University of Lausanne, Lausanne, Switzerland.
Hal M HoffmanDepartment of Paediatrics, University of California, San Diego, California, USA.
Antonella InsalacoDipartimento di Medicina Pediatrica, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy.
Annette F JanssonPaediatric Pneumology and Immunology and Interdisciplinary Centre for Social Paediatrics, Charite University Medicine Berlin, Berlin, Germany.
Tilmann KallinichPaediatric Pneumology and Immunology and Interdisciplinary Centre for Social Paediatrics, Charite University Medicine Berlin, Berlin, Germany.
Isabelle Kone-PautDepartment of Paediatric Rheumatology and CEREMAI, Bicêtre Hospital, APHP, University of Paris Sud, Paris, France.
Anna KozlovaDepartment of Immunology, Federal Research and Clinical Centre for Paediatric Haematology, Oncology and Immunology, Moscow, Russian Federation.
Jasmin Beate Kuemmerle-DeschnerDivision of Pediatric Rheumatology, Department of Pediatrics, University Hospital Tübingen, Tübingen, Germany.
Helen J LachmannDivision of Medicine, University College London, London, UK.
Ronald M LaxerDepartment of Paediatrics and Medicine, University of Toronto and the Hospital for Sick Children, Toronto, Ontario, Canada.
Alberto MartiniDirezione Scientifica, Istituto Giannina Gaslini, Genova, Liguria, Italy.
Susan NielsenPaediatric Rheumatology Unit 4272, Rigshospitalet, Copenhagen, Denmark.
Irina NikishinaDepartment of Paediatric Rheumatic Diseases, VA Nasonova Research Institute of Rheumatology, Moscow, Russian Federation.
Amanda K OmbrelloInflammatory Disease Section, National Human Genome Research Institute, Bethesda, Maryland, USA.
Seza ÖzenPediatric Rheumatology, Hacettepe University, Ankara, Turkey.
Efimia Papadopoulou-AlatakiFourth Department of Pediatrics, Aristotle University of Thessaloniki, Thessaloniki, Greece.
Pierre QuartierDepartment of Paediatric Immunology-Hematology and Rheumatology Unit and IMAGINE Institute, Institution Necker-Enfants Malades Hospital and Paris Descartes University, Paris, Île-de-France, France.
Donato RiganteInstitute of Paediatrics, Fondazione Policlinico Universitario A Gemelli, Università Cattolica Sacro Cuore, Rome, Italy.
Ricardo RussoServicio de Inmunología/Reumatología, Hospital de Pediatria Juan P Garrahan, Buenos Aires, Argentina.
Anna SimonDepartment of General Internal Medicine, Radboud Expertise Centre for Immunodeficiency and Autoinflammation, Radboud University Medical Centre, Nijmegen, The Netherlands.
Maria TrachanaPaediatric Immunology and Rheumatology Referral Centre, First Paediatric Clinic, Aristotle University of Thessaloniki, Thessaloniki, Greece.
Yosef UzielDepartment of Paediatrics, Meir Medical Centre, Kfar Saba, Israel.
Angelo RavelliInstitution Università degli Studi di Genova and G Gaslini Institute, Genova, Italy.
Grant SchulertDivision of Rheumatology, Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA.
Marco GattornoInstitution Università degli Studi di Genova and G Gaslini Institute, Genova, Italy.
Joost FrenkelDepartment of Paediatrics, Universitair Medisch Centrum Utrecht-Locatie Wilhelmina Kinderziekenhuis, Utrecht, The Netherlands.

Funding

Pathogenesis, and Outcome of Autoinflammatory Diseases, NOMID/CAPS, DIRA, CANDLE, SAVI, NLRC4-MAS, Stills-like Diseases, and other Undifferentiated Autoinflammatory DiseasesZIAAI001220 · NIAID · NATIONAL INSTITUTE OF ALLERGY AND INFECTIOUS DISEASES · PI GOLDBACH-MANSKY, RAPHAELA · 2017 to 2025
$22.3M
Intramural NIH HHS Z99 AI999999
6 · The paper itself

Abstract

introductionAutoinflammatory diseases can cause irreversible tissue damage due to systemic inflammation. Recently, the Autoinflammatory Disease Damage Index (ADDI) was developed. The ADDI is the first instrument to quantify damage in familial Mediterranean fever, cryopyrin-associated periodic syndromes, mevalonate kinase deficiency and tumour necrosis factor receptor-associated periodic syndrome. The aim of this study was to validate this tool for its intended use in a clinical/research setting.

methodsThe ADDI was scored on paper clinical cases by at least three physicians per case, independently of each other. Face and content validity were assessed by requesting comments on the ADDI. Reliability was tested by calculating the intraclass correlation coefficient (ICC) using an 'observer-nested-within-subject' design. Construct validity was determined by correlating the ADDI score to the Physician Global Assessment (PGA) of damage and disease activity. Redundancy of individual items was determined with Cronbach's alpha.

resultsThe ADDI was validated on a total of 110 paper clinical cases by 37 experts in autoinflammatory diseases. This yielded an ICC of 0.84 (95% CI 0.78 to 0.89). The ADDI score correlated strongly with PGA-damage (r=0.92, 95% CI 0.88 to 0.95) and was not strongly influenced by disease activity (r=0.395, 95% CI 0.21 to 0.55). After comments from disease experts, some item definitions were refined. The interitem correlation in all different categories was lower than 0.7, indicating that there was no redundancy between individual damage items.

conclusionThe ADDI is a reliable and valid instrument to quantify damage in individual patients and can be used to compare disease outcomes in clinical studies.

Indexed as

Severity of Illness IndexAdolescentAdultChildComputer SimulationCryopyrin-Associated Periodic SyndromesFamilial Mediterranean FeverHereditary Autoinflammatory DiseasesHumansMevalonate Kinase DeficiencyObserver VariationRegistriesReproducibility of ResultsYoung Adultfamilial Mediterranean feverfever syndromesinflammation

Identifiers

PMID30077992
PMCPMC8411437

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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.