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Clinical manifestations and outcome of anti-PL7 positive patients with antisynthetase syndrome.
Marie, I; Josse, S; Decaux, O; Diot, E; Landron, C; Roblot, P; Jouneau, S; Hatron, P Y; Hachulla, E; Vittecoq, O; Menard, J-F; Jouen, F; Dominique, S.
Afiliación
  • Marie I; Department of Internal Medicine, CHU Rouen, University of Rouen IFRMP, Institute for Biochemical Research, Rouen, France. isabelle.marie@chu-rouen.fr
Eur J Intern Med ; 24(5): 474-9, 2013 Jul.
Article en En | MEDLINE | ID: mdl-23375620
BACKGROUND: The aims of the present study were to determine both clinical manifestations and outcome of anti-PL7 patients with antisynthetase syndrome (ASS). METHODS: The medical records of 15 consecutive anti-PL7 patients with biopsy proven ASS were retrospectively analyzed without prior selection. RESULTS: Anti-PL7 patients exhibited polymyositis (n=14) and dermatomyositis (n=1); extra-pulmonary manifestations of ASS included: Raynaud's phenomenon (40%), mechanic's hands (33.3%), joint impairment (26.7%), pericardial effusion (20%) and esophageal/gastrointestinal involvement (20%). The outcome of myositis was as follows: remission/improvement (91.7%) and deterioration (8.3%). Fourteen patients (93.3%) experienced interstitial lung disease (ILD). ILD preceded ASS diagnosis (n=5), was identified concomitantly with ASS (n=8) and occurred after ASS diagnosis (n=1). Patients could be divided into 3 groups according to their presenting lung manifestations: acute onset of lung disease (n=1), progressive onset of lung signs (n=11) and asymptomatic patients exhibiting abnormalities consistent with ILD on PFT and HRCT-scan (n=2). No patient had resolution of ILD, whereas 64.3% and 35.7% experienced improvement and deterioration of ILD, respectively. ILD resulted in respiratory insufficiency requiring O2 therapy in 14.3% of cases. Two patients died. Predictive parameters of ILD deterioration were: DLCO<45% at ILD diagnosis and HRCT-scan pattern of usual interstitial pneumonia (UIP). CONCLUSION: Our series mainly underscores that ILD is frequent in anti-PL7 patients, leading to high morbidity. Our study further suggests that patients with predictive factors of ILD deterioration may require more aggressive therapy, especially the group of patients with DLCO<45% at ILD diagnosis and UIP pattern on HRCT-scan.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Autoanticuerpos / Treonina-ARNt Ligasa / Enfermedades Pulmonares Intersticiales / Miositis Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Female / Humans / Male / Middle aged Idioma: En Revista: Eur J Intern Med Asunto de la revista: MEDICINA INTERNA Año: 2013 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Países Bajos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Autoanticuerpos / Treonina-ARNt Ligasa / Enfermedades Pulmonares Intersticiales / Miositis Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Female / Humans / Male / Middle aged Idioma: En Revista: Eur J Intern Med Asunto de la revista: MEDICINA INTERNA Año: 2013 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Países Bajos