Anasarca as the initial presentation of juvenile polymyositis: an uncommon occurrence

Sharma, Deepak; Singh, Surjit; Suri, Deepti; Shava, Upender; Sodhi, Kushaljit Singh

doi:10.1007/s00296-011-2054-0

Cited by 5 publications

(4 citation statements)

References 4 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Eighteen patients reported with the diagnosis of JDM and where edema were found. We summarized the clinical and laboratory characteristic of these patients in Table 1 14‐30 . Considering the evidence from the literature and our patients, we suggest that edema may not be an uncommon finding in JDM and juvenile myositis.…”

Section: Discussionmentioning

confidence: 89%

“…We summarized the clinical and laboratory characteristic of these patients in Table 1. [14][15][16][17][18][19][20][21][22][23][24][25][26][27][28][29][30] Considering the evidence from the literature and our patients, we suggest that edema may not be an uncommon finding in JDM and juvenile myositis. We report that angioedema-like presentation can occur at onset of IIMs and may be the sole skin finding in the early phase of JDM and juvenile myositis.…”

Section: Discussionmentioning

confidence: 97%

See 1 more Smart Citation

Angioedema‐like presentation as the presenting finding of juvenile myositis and juvenile dermatomyositis in 2 patients

Karaca

Güngör

Güneş³

et al. 2022

Int J of Rheum Dis

View full text Add to dashboard Cite

Background Juvenile dermatomyositis (JDM) is the most common subtype of idiopathic inflammatory myopathies in childhood. Gottron’s papules, shawl sign, periorbital heliotrope rash, and periungual telengiectasis are characteristic skin findings of the disease. Besides characteristic skin involvement, some other skin findings, such as angioedema, may be seen prior or in the course of the disease. The presence of angioedema in JDM is emphasized in this report. Case presentations We present 2 unrelated girls, aged 2 (case 1) and 12 years (case 2), who had developed symmetrical weakness in the proximal muscles, muscle pain, elevated muscle enzymes and angioedema. Both cases had abnormal muscle magnetic resonance imaging findings, suggestive of inflammatory myositis. Muscle biopsy was performed only in case 1, and major histocompatibility complex‐1 expression on myofibers was shown consistent with JDM. Cases were diagnosed as probable and definite JDM, respectively. Angioedema was prominent, particularly in the lips and extremities of both cases, without laboratory evidence of C1 inhibitor deficiency or capillary leak syndrome, and absence of family history. Mast cell‐mediated, acquired angioedema was the most likely diagnosis. In both cases, skin and muscle findings improved significantly with steroid treatment. Conclusion We suggest that angioedema may be among the characteristic skin findings in JDM, and may be included in subsequent definitions.

show abstract

Section: Discussionmentioning

confidence: 89%

Section: Discussionmentioning

confidence: 97%

Angioedema‐like presentation as the presenting finding of juvenile myositis and juvenile dermatomyositis in 2 patients

Karaca

Güngör

Güneş³

et al. 2022

Int J of Rheum Dis

View full text Add to dashboard Cite

show abstract

“…The unique feature of our patient was generalized body swelling along with severe dysphagia. In the pediatric literature, the initial presentation of JPM patients with generalized body swelling has been described before [ 4 ]. However, the presentation of patients with accompanying symptoms of dysphagia has not been documented yet.…”

Section: Discussionmentioning

confidence: 99%

“…Further assessment with a laboratory workup and a muscle biopsy revealed the diagnosis of JPM. There have been very few instances reported in the pediatric literature with widespread body swelling as a presenting complaint of JPM [ 4 ].…”

Section: Introductionmentioning

confidence: 99%

An Unusual Presentation of Juvenile Polymyositis in an Adolescent Girl

Patel¹,

Zala²,

Chaudhari³

et al. 2023

Cureus

View full text Add to dashboard Cite

Juvenile idiopathic inflammatory myopathies (JIIMs) are a group of diverse, systemic autoimmune diseases that manifest in childhood and are characterized by weakness and chronic inflammation of skeletal muscles. One of the relatively rare variants of JIIMs is juvenile polymyositis (JPM). JPM patients present with proximal and distal muscle weakness, gait instability with falls, muscle pain and tenderness, and high levels of creatine kinase (CK) during adolescence. There are currently few people being diagnosed with JPM, which raises the question of whether or not it is a distinct disease. We discuss the case of a 13-year-old girl who presented to the hospital with generalized body swelling and difficulty swallowing solid food. She also had drooling of saliva during the presentation and a history of difficulty climbing up and down the stairs for three months. Her extensive laboratory workup showed a positive antinuclear antibody (ANA) test and increased muscle enzyme. A muscle biopsy was ordered, and she was diagnosed with JPM. Such a unique presentation has rarely been reported in the pediatric literature. This case report outlines an unusual JPM presentation that could help clinicians identify the condition and start treatment as soon as possible to minimize complications.

show abstract

Polymyositis Presenting with Isolated Periorbital Edema: a Case-Based Review

Bauzon

Shafi

2021

SN Compr. Clin. Med.

View full text Add to dashboard Cite

Anasarca as the initial presentation of juvenile polymyositis: an uncommon occurrence

Cited by 5 publications

References 4 publications

Angioedema‐like presentation as the presenting finding of juvenile myositis and juvenile dermatomyositis in 2 patients

Angioedema‐like presentation as the presenting finding of juvenile myositis and juvenile dermatomyositis in 2 patients

An Unusual Presentation of Juvenile Polymyositis in an Adolescent Girl

Polymyositis Presenting with Isolated Periorbital Edema: a Case-Based Review

Contact Info

Product

Resources

About