The long-term outcome of anti-Jo-1-positive inflammatory myopathies

Späth, M.; Schröder, Mira; Schlotter‐Weigel, Beate; Walter, Maggie C.; Haŭtmann, Hŭbert; Leinsinger, Gerda; Pongratz, D.; Müller‐Felber, Wolfgang

doi:10.1007/s00415-004-0449-5

Cited by 64 publications

(32 citation statements)

References 21 publications

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“…5,8,10,11,13,17,19,21 Morbidity in both PM/DM and the anti-Jo-1 syndrome is related to acute respiratory decompensation at presentation and our study confirms this association and points out that the morphologic equivalent may be diffuse alveolar damage alone or acute lung injury superimposed on underlying fibrotic interstitial pneumonias such as usual interstitial pneumonia and fibrotic nonspecific interstitial pneumonia. 5,8,17,19,21 The limitations to this study are notable. First, this is a retrospective study, where clinical information, largely based on the referral nature of the patient population to the University of Pittsburgh, is potentially restricted.…”

Section: Discussionsupporting

confidence: 79%

“…The most common of these is the antiJo-1 antisynthetase syndrome in which anti-histidyl tRNA synthetase (anti-Jo-1) antibodies are accompanied by clinical arthritis, Raynaud's phenomenon, myositis, mechanic's hands, and interstitial lung disease (ILD). [1][2][3][4][5][6][7][8][9][10] The anti-Jo-1 tRNA synthetase syndrome has a higher incidence of ILD, relative to classical PM/DM, approximating 90% in some series, and the presence of anti-Jo-1 antibodies correlates with poor patient prognosis. [11][12][13][14][15][16][17][18][19][20][21][22] In this report, we studied the pulmonary histopathologic changes of a select group of IIM patients with the anti-Jo-1 tRNA synthetase syndrome, generating the first and largest biopsy study focused on this specific group of patients.…”

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confidence: 99%

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The pulmonary histopathologic manifestations of the anti-Jo-1 tRNA synthetase syndrome

et al. 2010

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Section: Discussionsupporting

confidence: 79%

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confidence: 99%

The pulmonary histopathologic manifestations of the anti-Jo-1 tRNA synthetase syndrome

et al. 2010

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“…Moreover, certain causes require specific treatments. These treatments could be essential, as the long-term prognosis and the survival of patients with ASS, based on retrospective studies, showed a clear correlation with lung involvement [2,[18][19][20]. However, to date, the influence of cofactors associated with ILD, such as PH, has rarely been evaluated in large series.…”

Section: Introductionmentioning

confidence: 99%

Pulmonary hypertension in antisynthetase syndrome: prevalence, aetiology and survival

et al. 2013

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Antisynthetase syndrome is characterised by the association of interstitial lung disease and myositis with different anti-tRNA-synthetase antibodies. The occurrence, aetiology and prognosis of pulmonary hypertension have not yet been evaluated.Among 203 consecutive patients, transthoracic echocardiogram and right heart catheterisation results were retrospectively analysed in the light of clinico-biological, morphological and functional parameters. Definitions of pulmonary hypertension were based on the European Society of Cardiology/European Respiratory Society 2009 guidelines, with severe pulmonary hypertension being defined by a mean pulmonary arterial pressure .35 mmHg.Pulmonary hypertension was suspected by transthoracic echocardiogram in 47 (23.2%) cases, corresponding to pulmonary hypertension ''possible'' (n527, 13.3%) or ''likely'' (n520, 9.9%). Right heart catheterisation was performed in 21 patients, excluding pulmonary hypertension in five and confirming pre-capillary pulmonary hypertension in 16 (7.9%). Although related to interstitial lung disease in all cases, pre-capillary pulmonary hypertension was severe in 13 (81.3%) patients (mean¡SD pulmonary arterial pressure 46¡9 mmHg), frequently associated with low cardiac index (mean¡SD 2.3¡0.8 L?min -1 ?m -2 ) and high forced vital capacity/diffusing capacity of the lung for carbon monoxide ratio (2.5¡0.6). Pulmonary hypertension was significantly associated with a lower survival rate (p,0.001), with a 3-year survival rate of 58%.The occurrence of pulmonary hypertension in antisynthetase syndrome is significant and dramatically worsens the prognosis. Although systematically associated with interstitial lung disease, pulmonary hypertension was usually severe, suggesting a specific pulmonary vascular involvement. @ERSpublications PH in antisynthetase syndrome significantly worsens the prognosis, suggesting a specific pulmonary vascular involvement

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“…4,31,33,34 Furthermore, positive anti-Jo-1 and/or interstitial pneumopathy as prognostic factors remain controversial. [34][35][36][37] In our patients, there was one death attributed to septic shock (hospital bronchopneumonia).…”

Section: Discussionmentioning

confidence: 99%

“…[1][2][3] Since it is a rare syndrome, its prevalence in the general population is unknown. Moreover, ASS epidemiological studies [4][5][6] are scarce in the literature, except for a few case reports and serial cases. 3,[7][8][9][10][11] Therefore, we reported a cohort retrospective study of 18 patients with anti-Jo-1 ASS from our tertiary service.…”

Section: -3mentioning

confidence: 99%

Síndrome antissintetase anti-Jo-1

Shinjo¹,

Levy‐Neto²

2010

Rev. Bras. Reumatol.

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Objective: Given a lack of population-based studies, we report an epidemiological-clinic study of anti-Jo-1 antisynthetase syndrome (ASS). Patients and Methods: To study a retrospective cohort of a single-center from 1980 to 2010. Clinical-laboratory and demographic data were obtained from medical files. All patients fulfilled the Bohan and Peter criteria (1975) and presented anti-Jo-1, articular, muscle and lung involvement. Eighteen patients with anti-Jo-1 ASS were analyzed. Results: The mean age at disease onset was 39.9 ± 15.7 years and average disease duration was 9.7 ± 7.0 years. All subjects were white, and 94.4% were female. Constitutional symptoms occurred in 50 % of cases. There was cutaneous and gastrointestinal tract involvement in 66.6% and 55.6% of cases, respectively. No cases manifested neurologic or cardiac involvement. Half of the patients showed incipient pneumopathy, ground-glass opacities and basal pulmonary fibrosis. There was one case of tuberculosis, three of herpes zoster and one of non-Hodgkin lymphoma. One death occurred due to sepsis shock (hospital bronchopneumonia). All patients received prednisone (1mg/kg/day) and 12 (66.7%) participants received methyl prednisolone pulse therapy (1g/day, 3 days). Various immunosuppressants were used as corticosteroid tapers, depending on tolerance, side effects and/or refractoriness. Although disease relapse (clinical and/or laboratory) occurred in 87.5% of cases, 12 out of 16 patients (75%) were in disease remission at study endpoint. Conclusion: In the present study, almost all patients were white females and the disease relapse rate was high.

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The long-term outcome of anti-Jo-1-positive inflammatory myopathies

Cited by 64 publications

References 21 publications

The pulmonary histopathologic manifestations of the anti-Jo-1 tRNA synthetase syndrome

The pulmonary histopathologic manifestations of the anti-Jo-1 tRNA synthetase syndrome

Pulmonary hypertension in antisynthetase syndrome: prevalence, aetiology and survival

Síndrome antissintetase anti-Jo-1

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