Extended myositis-specific and -associated antibodies profile in systemic sclerosis: A cross-sectional study

Leurs, Amélie; Dubucquoi, Sylvain; Machuron, François; Balden, Maïté; Renaud, François; Rogeau, Stéphanie; López, Benjamín; Lambert, Marc; Morell-Dubois, Sandrine; Maillard, Hélène; Behal, Hélène; Hachulla, É.; Launay, David; Sobanski, Vincent

doi:10.1016/j.jbspin.2020.06.021

Cited by 15 publications

(11 citation statements)

References 35 publications

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“…25 Concerning autoantibody characteristics, the anti-Ro-52 was more prevalent in both groups, whereas the anti-centromere was more common in the Japanese group. This finding differs from previous studies in the United Kingdom, France [26][27][28][29] and Japan 6 , in which the most prevalent autoantibodies were anti-Ku, and anti-PM/Scl75. In our study, only six (13.3%) patients had anti-Ku being that of these three (6.6%) were Japanese.…”

Section: Discussioncontrasting

confidence: 99%

“…In the past five decades, epidemiological, histopathological, and laboratory studies have attempted to determine which phenotypes of patients with SSc would develop Myos and what impact this impairment has on these patients’ prognosis. [ 9 , 19 , 27 ] Several case reports from the 19 th century[ 2 , 3 ] referred to severe outcomes for these pathologies. In addition, patients with Myo-SSc are more severe, they tend to have a worse prognosis,[ 20 ] greater morbidities, impaired quality of life[ 11 ] and incidence of cardiac involvement.…”

Section: Discussionmentioning

confidence: 99%

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Patients with inflammatory myopathies overlapping with systemic sclerosis: A Brazilian-Japanese bicentric study

et al. 2022

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Objectives: This study aims to describe and compare the demographic, clinical, and laboratory characteristics and follow-up of representative samples of patients with myopathies and systemic sclerosis overlap syndromes (Myo-SSc) from two tertiary centers. Patients and methods: This is a cross-sectional and retrospective study conducted between January 2000 and December 2020. Fourty-five patients were analyzed with Myo-SSc (6 males, 39 females; mean age: 50.2±15.4 years; range, 45 to 65 years) from two tertiary centers (n=30 from Brazil and n=15 from Japan). Results: The median follow-up was 98 (range, 37 to 168) months. Muscle impairment started simultaneously with the diagnosis of systemic sclerosis in 57.8% (26/45) of cases. Muscle involvement occurred before the onset of systemic sclerosis in 35.5% (16/45) of cases, and after in 6.7% (3/45). Polymyositis was observed in 55.6% (25/45) of cases, followed by dermatomyositis in 24.4% (11/45) and antisynthetase syndrome in 20.0% (9/45). Concerning systemic sclerosis, the diffuse and limited forms occurred in 64.4% (29/45) and 35.6% (16/45) of the cases, respectively. Comparing the subgroups, Myo or SSc onset was earlier in Brazilian patients, and they had a higher frequency of dysphagia (20/45, [66.7%]) and digital ulcers (27/45, [90%]), whereas Japanese patients had higher modified Rodnan skin scores (15 [9 to 23]) and prevalence of positive anti-centromere antibodies (4/15 [23.7%]). The current disease status and mortality were similar in both groups. Conclusion: In the present study, Myo-SSc affected middle-aged women, and its manifestation spectrum varied according to geographic distribution.

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Section: Discussioncontrasting

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Patients with inflammatory myopathies overlapping with systemic sclerosis: A Brazilian-Japanese bicentric study

et al. 2022

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“…We also found a high proportion of patients positive for anti-MDA5 antibodies compared with previous reports. 6 Because the majority of the MDA5 titers were low, and these patients did not demonstrate classic MDA5 clinical presentations such as rapidly progressive ILD or digital ulcerations, 13 further testing should be performed to confirm these positive results. Among the less prevalent MSA/MAA, we also found an association between Mi-2b and elevated MRSS and an association between PM-75 antibodies and digital ulcers.…”

Section: Discussionmentioning

confidence: 99%

Clinical and Autoantibody Associations in Antinuclear Antibody–Positive Systemic Sclerosis Lacking Prototypic Autoantibodies

Kruzer¹,

Marangoni

Heckler

et al. 2022

J Clin Rheumatol

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“…Our case was anti-Ro-52 positive, which is considered a SSc-associated autoantibody. Myositis-specific antibodies, as anti-Mi-2b found in our patient, are typically thought to be rare in jSSc, although Leurs et al have recently reported a prevalence of 8.0% in SSc [ 19 ].…”

Section: Discussionmentioning

confidence: 76%

Juvenile diabetes and systemic sclerosis: just a coincidence?

et al. 2022

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Background Limited joint mobility (LJM), previously known as cheiroarthropathy, refers to the presence of reduced extension at the finger joints in people with diabetes and may be associated with scleroderma-like syndromes such as diabetic sclerodactyly. While scleroderma-like syndromes and LJM have been observed in patients with long-term diabetes and associated complications, the coexistence of diabetes with Juvenile systemic sclerosis (jSSc) is rarely described. Case presentation We describe the case of a 14-year-old boy with long-lasting type 1 diabetes (T1D) and suspected LJM associated with Raynaud phenomenon, sclerodactyly and tapering of the fingertips. A comprehensive work-up showed positive autoantibodies (ANA, anti-Ro-52, anti-Mi-2b), abnormal nailfold capillaroscopy with a scleroderma pattern, interstitial lung disease and cardiac involvement. The overall clinical picture was consistent with the diagnosis of jSSc. Conclusions LJM can be the initial sign of underlying systemic sclerosis. Nailfold capillaroscopy may help differentiate jSSc from classical LJM in pediatric patients with T1D and finger contractures or skin induration of no clear origin. This case report provides a starting point for a novel hypothesis regarding the pathogenesis of jSSc. The association between T1D and jSSc may be more than a coincidence and could suggest a relationship between glucose metabolism, fibrosis and microangiopathy.

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Extended myositis-specific and -associated antibodies profile in systemic sclerosis: A cross-sectional study

Cited by 15 publications

References 35 publications

Patients with inflammatory myopathies overlapping with systemic sclerosis: A Brazilian-Japanese bicentric study

Patients with inflammatory myopathies overlapping with systemic sclerosis: A Brazilian-Japanese bicentric study

Clinical and Autoantibody Associations in Antinuclear Antibody–Positive Systemic Sclerosis Lacking Prototypic Autoantibodies

Juvenile diabetes and systemic sclerosis: just a coincidence?

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