In silico validation of the Autoinflammatory Disease Damage Index.
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Ошибки и ограничения
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Ter Haar Nienke M, van Delft Amber Laetitia Justine, Annink Kim Valerie, van Stel Henk, Al-Mayouf Sulaiman M, Amaryan Gayane, Anton Jordi, Barron Karyl S, Benseler Susanne, Brogan Paul A, Cantarini Luca, Cattalini Marco, Cochino Alexis-Virgil, de Benedetti Fabrizio, Dedeoglu Fatma, de Jesus Adriana Almeida, Demirkaya Erkan, Dolezalova Pavla, Durrant Karen L, Fabio Giovanna, Gallizzi Romina, Goldbach-Mansky Raphaela, Hachulla Eric, Hentgen Veronique, Herlin Troels, Hofer Michaël, Hoffman Hal M, Insalaco Antonella, Jansson Annette F, Kallinich Tilmann, Kone-Paut Isabelle, Kozlova Anna, Kuemmerle-Deschner Jasmin Beate, Lachmann Helen J, Laxer Ronald M, Martini Alberto, Nielsen Susan, Nikishina Irina, Ombrello Amanda K, Özen Seza, Papadopoulou-Alataki Efimia, Quartier Pierre, Rigante Donato, Russo Ricardo, Simon Anna, Trachana Maria, Uziel Yosef, Ravelli Angelo, Schulert Grant, Gattorno Marco, Frenkel Joost
Аннотация
METHODS: The 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.
RESULTS: The 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.
CONCLUSION: The ADDI is a reliable and valid instrument to quantify damage in individual patients and can be used to compare disease outcomes in clinical studies.