Anti-synthetase syndrome: a rare and challenging diagnosis for bilateral ground-glass opacities—a case report with literature review

Alfraji, Nasam; Mazahir, Usman; Chaudhri, Moiuz; Miskoff, Jeffrey A

doi:10.1186/s12890-020-01388-0

Cited by 21 publications

(15 citation statements)

References 12 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…COVID-19 infection may present with a multitude of pulmonary findings, including diffuse ground-glass opacities (18). Similarly, the pulmonary manifestations of an ASS may present with patchy ground-glass opacities, similar to the interstitial shadows commonly seen in COVID-19-related pneumonia (19). The inflammatory process caused by SARS-CoV-2 infection involves cytokine storm and macrophage consequence of the molecular autoantigen transformation or modifications due to the influence of the SARS-CoV-2 infection (25).…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

New-onset dermatomyositis following COVID-19: A case report

Shimizu

Matsumoto²,

Sasajima³

et al. 2022

Front. Immunol.

View full text Add to dashboard Cite

Coronavirus disease 2019 (COVID-19) is an infectious disease caused by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2). Most of the infected individuals have recovered without complications, but a few patients develop multiple organ involvements. Previous reports suggest an association between COVID-19 and various inflammatory myopathies, in addition to autoimmune diseases. COVID-19 has been known to exacerbate preexisting autoimmune diseases and trigger various autoantibodies and autoimmune disease occurrence. Here we report a case of complicated COVID-19 with anti-synthetase autoantibodies (ASSs) presenting with skin rash, muscle weakness, and interstitial lung disease (ILD) and subsequently diagnosed with dermatomyositis (DM). A 47-year-old Japanese male patient without any previous history of illness, including autoimmune diseases, presented with a high fever, sore throat, and cough. Oropharyngeal swab for SARS-Cov-2 polymerase chain reaction tested positive. He was isolated at home and did not require hospitalization. However, his respiratory symptoms continued, and he was treated with prednisolone (20 mg/day) for 14 days due to the newly developing interstitial shadows over the lower lobes of both lungs. These pulmonary manifestations remitted within a week. He presented with face edema and myalgia 4 weeks later when he was off corticosteroids. Subsequently, he presented with face erythema, V-neck skin rash, low-grade fever, and exertional dyspnea. High-resolution computed tomography of the chest showed ILD. Biochemical analysis revealed creatine kinase and aldolase elevations, in addition to transaminases. Anti-aminoacyl tRNA synthetase (ARS) was detected using an enzyme-linked immunosorbent assay (170.9 U/mL) (MESACUP™ (Medical & Biological Laboratories, Japan), and the tRNA component was identified as anti-PL-7 and anti-Ro-52 antibodies using an immunoblot assay [EUROLINE Myositis Antigens Profile 3 (IgG), Euroimmun, Lübeck,Germany]. The patient was diagnosed with DM, especially anti- synthase antibody syndrome based on the presence of myositis-specific antibodies, clinical features, and pathological findings. The present case suggests that COVID-19 may have contributed to the production of anti-synthetase antibodies (ASAs) and the development of de novo DM. Our case highlights the importance of the assessment of patients who present with inflammatory myopathy post-COVID-19 and appropriate diagnostic work-up, including ASAs, against the clinical features that mimic DM after post-COVID-19.

show abstract

Section: Discussionmentioning

confidence: 99%

“…Coronavirus disease 2019 (COVID- 19), which is caused by the severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), is a life-threatening respiratory illness (1). COVID-19 is a heterogeneous disease ranging from asymptomatic course to multi-organ failure during the inflammatory processes (2).…”

Section: Introductionmentioning

confidence: 99%

New-onset dermatomyositis following COVID-19: A case report

Shimizu

Matsumoto²,

Sasajima³

et al. 2022

Front. Immunol.

View full text Add to dashboard Cite

show abstract

“…The clinical presentation of ILD consists of sudden or gradual onset of exertional dyspnea and difficult-to-control dry cough. In the later course, ILD can be complicated with pulmonary hypertension (PAH) [9][10][11]. Fortunately, control echocardiogram in our patient did not show evolution into PAH.…”

Section: Fig 2 Mechanic Handsmentioning

confidence: 60%

“…Interstitial lung disease (ILD) is one of the most prevalent feature of ASS affecting 71-100% of patients [8], being in correlation with the severity of the disease and emerging as a major determinant of its morbidity and mortality. ILD may precede myopathy in more than one third of cases,in up to 55% of the cases [8,9,10,11]. The severity and type of lung involvement correlates with the prognosis of the disease.…”

Section: Fig 2 Mechanic Handsmentioning

confidence: 99%

Antisynthetase syndrome a rare and challenging diagnosis-case report and literature review

Karanfilovski

Janev²,

Nanovic³

et al. 2022

RBJ

View full text Add to dashboard Cite

This case describes a 46-years old woman who initially presented with myopathy and arthropathy (i.e. musculoskeletal manifestations), which delayed the recognition of antisynthetase syndrome (ASS) and contributed for considering the condition as seronegative rheumatoid arthritis for several years. During the next few years, the patient was progressively worsening, with a disability to stand up from a sitting position, gradual onset of exertional dyspnea, difficult-to-control dry cough and thick, hyperkeratotic skin of both hands (mechanic’s hands). This constellation of symptoms was highly suspicious for ASS and additional serological and radiological examinations were done which confirmed the diagnosis of ASS. The need for further detailed investigation when an interstitial lung disease overlaps with a known rheumatoid condition is obligatory, as shown in this case. A multidisciplinary evaluation is highly recommended to evaluate the clinical, serological and radiological findings in each patient suspected for ASS in order to establish early diagnosis and timely management.

show abstract

“…This syndrome is characterized by the binding of an autoanthexadese antibody to an aminoacyl-transferred RNA synthase, most commonly the Jo-1 antibody[ 4 ]. Clinical manifestations of ASS are diverse, including idiopathic inflammatory myositis (IIM), arthralgia and arthritis, interstitial pneumonia, Raynaud’s phenomenon, and skin lesions characterized by “mechanic’s hands[ 5 ].” The pathogenesis of ASS is unclear, but it is thought to be associated with chronic immune system activation[ 6 ]. It is also unclear whether there is an overlap between ASS and RA, or if ASS in some cases mimics RA[ 4 ].…”

Section: Introductionmentioning

confidence: 99%

Pulmonary hypertension secondary to seronegative rheumatoid arthritis overlapping antisynthetase syndrome: A case report

Huang¹,

Lu²,

Tian³

et al. 2022

WJCC

View full text Add to dashboard Cite

BACKGROUND Polyarthritis is the most frequent clinical manifestation in antisynthetase syndrome (ASS) forms of idiopathic inflammatory myositis and may be misdiagnosed as rheumatoid arthritis (RA), particularly in patients with seronegative RA (SNRA). It is unclear whether there is an overlap between ASS and RA, or if ASS sometimes mimics RA. Pulmonary hypertension (PAH) is common in connective tissue diseases (CTDs). However, published reports on CTD-PAH do not include overlapping CTDs, and its incidence and impact on patient prognosis are unclear. CASE SUMMARY We report the case of a 63-year-old woman who presented with a 3-mo history of symptom aggravation of recurrent symmetrical joint swelling and pain that had persisted for over 10 years. The patient was diagnosed with RA and interstitial lung disease. The patient repeatedly presented to the hospital’s respiratory and rheumatology departments with arthralgia, plus shortness of breath after activity. Relevant tests indicated that anti-CCP and RF remained negative, while anti-J0-1 and anti-Ro-52 were strongly positive. It was not until recently that we recognized that this could be an unusual case of SNRA with concurrent ASS. Joint pain was relieved after regular anti-rheumatic treatment. Chest computed tomography scans showed that pulmonary interstitial changes did not progress significantly over several years; however, they showed gradual widening of the pulmonary artery, and cardiac ultrasound indicated elevated pulmonary artery systolic pressure. The prescribed treatment of PAH was not effective in improving shortness of breath. CONCLUSION Overlap of RA and ASS may be missed. Further research is necessary to facilitate early diagnosis, effective evaluation, and prognosis.

show abstract

Anti-synthetase syndrome: a rare and challenging diagnosis for bilateral ground-glass opacities—a case report with literature review

Cited by 21 publications

References 12 publications

New-onset dermatomyositis following COVID-19: A case report

New-onset dermatomyositis following COVID-19: A case report

Antisynthetase syndrome a rare and challenging diagnosis-case report and literature review

Pulmonary hypertension secondary to seronegative rheumatoid arthritis overlapping antisynthetase syndrome: A case report

Contact Info

Product

Resources

About