Autoantibody to PL-12 (Anti-Alanyl-tRNA Synthetase) in an African American Girl with Juvenile Dermatomyositis and Resolution of Interstitial Lung Disease

Vega, Patricia; Ibarra, Maria; Prestridge, Adrienne; Pachman, Lauren M.

doi:10.3899/jrheum.100608

Cited by 11 publications

(5 citation statements)

References 13 publications

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“…Case reports also support this reversibility with treatment: a case of acute respiratory distress syndrome secondary to ILD in IIM was resolved with aggressive treatment with tacrolimus (). Similarly, resolution of PFT and chest HRCT abnormalities after aggressive therapy has been described in a pediatric patient with IIM and the presence of anti–PL‐12 (). Based on this evidence, it is imperative to identify those patients with risk of pulmonary abnormalities, most specifically ILD, early in their disease course.…”

Section: Discussionmentioning

confidence: 66%

“…Adults prospectively followed with PFTs had normalization of TLC and DL CO over time in one‐third of patients after treatment (). Similarly, resolution of PFT and chest high‐resolution computed tomography (HRCT) abnormalities after aggressive therapy has been described in a pediatric patient with IIM and anti–PL‐12 (). Based on this evidence, it is imperative to identify those children at risk for pulmonary damage, most specifically ILD, early in their disease course.…”

Section: Introductionmentioning

confidence: 79%

“…The authors report the improvement in lung function after treatment with cyclosporine despite the fact that the patients had minimal symptoms (). One case report of anti–PL‐12 in a pediatric patient documented reversibility of ILD with aggressive treatment (). Early corticosteroid therapy sufficient to normalize enzymes apparently led to better resolutions of ILD ().…”

Section: Discussionmentioning

confidence: 99%

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Pulmonary Function Tests in Idiopathic Inflammatory Myopathy: Association With Clinical Parameters in Children

Prestridge

Morgan

Ferguson

et al. 2013

Arthritis Care & Research

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Objective. To determine the association of decreased lung function in children with idiopathic inflammatory myopathies (IIMs) with specific clinical parameters. Methods. This study of 38 children ages 6 -23 years diagnosed with definite/probable IIM evaluated the association of myositis-specific/-associated antibodies (MSAs/MAAs), duration of untreated disease at diagnosis, Disease Activity Score for muscle (DAS-M), muscle-derived enzymes (aldolase, lactate dehydrogenase [LDH], aspartate transaminase, and creatine phosphokinase [CPK]), neopterin and von Willebrand factor antigen, and the Childhood Myositis Assessment Scale (CMAS) scores with data from pulmonary function testing (PFT). Results. Impaired PFTs were defined as total lung capacity (TLC) or diffusing capacity for carbon monoxide (DLCO) of <80% predicted. The PFTs documented that 37% of the children (14 of 38) had either decreased TLC or decreased DLCO; 5% (2 of 38) had both. Children with decreased TLC alone (7 [18%] of 38) were older both at the time of PFT and diagnosis, had anti-Jo-1 and anti-Scl-70 antibody, and had elevated levels of CPK and neopterin. Children with decreased DLCO alone (5 [13%] of 38) had a shorter duration of untreated disease at diagnosis, had higher DAS-M and total DAS, were positive for anti-Ro and anti-PL-12, had increased LDH, and had elevated levels of neopterin and aldolase, with low CMAS scores for items 1, 3, 10, 11, and 14. Conclusion. Assessment of PFTs in children with IIMs should be considered, since more than one-third of patients were found to be impaired. The presence of MSAs/MAAs, an elevated serum neopterin level (mean ؎ SD 12.4 ؎ 9.6 nmoles/liter, normal value <10.5), older age at diagnosis, and shorter duration of untreated disease at diagnosis suggest the presence of potential lung pathology.

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Section: Discussionmentioning

confidence: 66%

Section: Introductionmentioning

confidence: 79%

Section: Discussionmentioning

confidence: 99%

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Pulmonary Function Tests in Idiopathic Inflammatory Myopathy: Association With Clinical Parameters in Children

Prestridge

Morgan

Ferguson

et al. 2013

Arthritis Care & Research

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“…A new biomarker, Krebs von den Lungen-6 (KL-6), appears to be useful in identifying children with ILD and is associated with increased IL-18 and ferritin [61]. ILD is increased in DM patients positive for MDA-5 who also may be clinically amyopathic (CADM) [51], as well as in children with inflammatory myopathy and other MSA, but can be reversed by aggressive therapy [62]. IV corticosteroids and cyclosporine A are frequently used for JDM with ILD; some biologics are promising.…”

Section: Nucleoside Monophosphatesmentioning

confidence: 99%

Juvenile Dermatomyositis: New Clues to Diagnosis and Therapy

Pachman

Nolan

DeRanieri

et al. 2021

Curr Treat Options in Rheum

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Purpose of review To identify clues to disease activity and discuss therapy options. Recent findings The diagnostic evaluation includes documenting symmetrical proximal muscle damage by exam and MRI, as well as elevated muscle enzymes—aldolase, creatine phosphokinase, LDH, and SGOT—which often normalize with a longer duration of untreated disease. Ultrasound identifies persistent, occult muscle inflammation. The myositis-specific antibodies (MSA) and myositis-associated antibodies (MAA) are associated with specific disease course variations. Anti-NXP-2 is found in younger children and is associated with calcinosis; anti-TIF-1γ+ juvenile dermatomyositis has a longer disease course. The diagnostic rash—involving the eyelids, hands, knees, face, and upper chest—is the most persistent symptom and is associated with microvascular compromise, reflected by loss of nailfold (periungual) end row capillaries. This loss is associated with decreased bioavailability of oral prednisone; the bioavailability of other orally administered medications should also be considered. At diagnosis, at least 3 days of intravenous methyl prednisolone may help control the HLA-restricted and type 1/2 interferon–driven inflammatory process. The requirement for avoidance of ultraviolet light exposure mandates vitamin D supplementation. Summary This often chronic illness targets the cardiovascular system; mortality has decreased from 30 to 1–2% with corticosteroids. New serological biomarkers indicate occult inflammation: ↑CXCL-10 predicts a longer disease course. Some biologic therapies appear promising.

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“…To complicate matters, a child with any of the MSAs may also have a MAA, for example anti-Ro-52 (6%), associated with ILD (52). Reversal of ILD with aggressive medical therapy was reported in a child positive for anti-PL-12 (53).…”

Section: Juvenile Dermatomyositis Etiology: Genetics and Environmentmentioning

confidence: 99%

Advances in Juvenile Dermatomyositis: Myositis Specific Antibodies Aid in Understanding Disease Heterogeneity

Pachman

Khojah

2018

The Journal of Pediatrics

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Autoantibody to PL-12 (Anti-Alanyl-tRNA Synthetase) in an African American Girl with Juvenile Dermatomyositis and Resolution of Interstitial Lung Disease

Cited by 11 publications

References 13 publications

Pulmonary Function Tests in Idiopathic Inflammatory Myopathy: Association With Clinical Parameters in Children

Pulmonary Function Tests in Idiopathic Inflammatory Myopathy: Association With Clinical Parameters in Children

Juvenile Dermatomyositis: New Clues to Diagnosis and Therapy

Advances in Juvenile Dermatomyositis: Myositis Specific Antibodies Aid in Understanding Disease Heterogeneity

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