Juvenile Dermatomyositis and Development of Malignancy: 2 Case Reports and a Literature Review

Cannon, Laura; Dvergsten, Jeffrey; Stingl, Cory

doi:10.3899/jrheum.190929

Cited by 5 publications

(3 citation statements)

References 7 publications

(8 reference statements)

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“…This has raised the possibility that anti-Sp4 autoantibodies may reflect a more effective anti-tumor response in adults. The risk of malignancy in juveniles with myositis, however, is exceedingly low (5,6). Nonetheless, it is now evident that anti-Sp4 autoantibodies are present in a subset of these patients.…”

Section: Sp4 Autoantibodies In Juvenile Myositismentioning

confidence: 99%

“…Although there is much overlap between adult and juvenile patients with myositis with the same MSA, critical differences exist. For instance, adults with anti-transcription intermediary factor 1 (anti-TIF1) autoantibodypositive dermatomyositis (DM) have a significantly higher risk of malignancy, whereas in juvenile-onset IIM malignancy has not been found to be associated with these autoantibodies and overall is exceedingly rare (3)(4)(5)(6).…”

Section: Introductionmentioning

confidence: 99%

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Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis

Sherman

Pak

Pinal‐Fernandez

et al. 2023

Arthritis & Rheumatology

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ObjectiveAutoantibodies recognizing specificity protein 4 (Sp4) were recently discovered in adults with idiopathic inflammatory myopathies (IIM). Anti‐Sp4 autoantibodies co‐occurred in patients with anti–transcription intermediary factor 1 (anti‐TIF1) autoantibody‐positive dermatomyositis (DM) and were associated with a reduced risk of cancer. In the present study, the prevalence and clinical features associated with anti‐Sp4 autoantibodies in juvenile‐onset IIM were investigated.MethodsSerum samples from 336 patients with juvenile myositis in a cross‐sectional cohort and 91 healthy controls were screened for anti‐Sp4 autoantibodies using enzyme‐linked immunosorbent assay. Clinical characteristics, outcomes, and HLA alleles of those with and those without anti‐Sp4 autoantibodies were compared.ResultsAnti‐Sp4 autoantibodies were present in 23 patients (7%) with juvenile myositis and were not present in any of the controls. Anti‐Sp4 autoantibodies were found among each clinical myositis subgroup. The frequency of TIF1 autoantibody positivity was significantly higher among those with anti‐Sp4 autoantibodies (21 [91%] versus 92 [30%], P < 0.001). In the anti‐TIF1 autoantibody–positive subgroup, Raynaud's phenomenon (8 [38%] versus 2 [2%], P < 0.001) was more common and peak aspartate aminotransferase was significantly lower in those with anti‐Sp4 autoantibodies. None of the patients with anti‐Sp4 autoantibodies required a wheelchair. Among White patients, DQA1*04 and DRB1*08 were associated with anti‐Sp4 autoantibodies.ConclusionAnti‐Sp4 autoantibodies were found in patients with juvenile‐onset IIM, predominantly those with coexisting anti‐TIF1 autoantibodies. Patients with anti‐Sp4 autoantibodies represent a phenotypic subset of anti‐TIF1 autoantibody–positive myositis characterized by frequent Raynaud's phenomenon and less pronounced muscle involvement, similar to adults with these autoantibodies. Novel immunogenetic risk factors for White patients with IIM were identified among juveniles with anti‐Sp4 autoantibodies.

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Section: Sp4 Autoantibodies In Juvenile Myositismentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis

Sherman

Pak

Pinal‐Fernandez

et al. 2023

Arthritis & Rheumatology

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“…In adults, there is a wellestablished relationship between DM and malignancies. However, the paraneoplastic phenomenon has very rarely been noted in pediatric patients, and the current data are limited to a few cases in the literature [6,7].…”

Section: Introductionmentioning

confidence: 95%

The Significance of Autoantibodies in Juvenile Dermatomyositis

Kwiatkowska

Reich

2021

BioMed Research International

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Juvenile dermatomyositis is a chronic and rare autoimmune disorder classified into the spectrum of idiopathic inflammatory myopathies. Although this entity is mainly characterized by the presence of pathognomonic cutaneous lesions and proximal muscle weakness, the clinical manifestation can be highly heterogeneous; thus, diagnosis might be challenging. Current treatment recommendations for juvenile dermatomyositis, based mainly upon case series, include the use of corticosteroids, immunomodulatory, and immunosuppressive agents. Recently, several specific autoantibodies have been shown to be associated with distinct clinical phenotypes of classic dermatomyositis. There is a need to further evaluate their relevance in the formation of various clinical features. Furthermore, while providing more personalized treatment strategies, one should consider diversity of autoantibody-related subgroups of juvenile dermatomyositis.

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Biologic drugs in the treatment of juvenile dermatomyositis: a literature review

Sener,

Cam,

Ozen

et al. 2023

Clin Rheumatol

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Juvenile Dermatomyositis and Development of Malignancy: 2 Case Reports and a Literature Review

Cited by 5 publications

References 7 publications

Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis

Autoantibodies Recognizing Specificity Protein 4 Co‐occur With Anti–Transcription Intermediary Factor 1 and Are Associated With Distinct Clinical Features and Immunogenetic Risk Factors in Juvenile Myositis

The Significance of Autoantibodies in Juvenile Dermatomyositis

Biologic drugs in the treatment of juvenile dermatomyositis: a literature review

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