幼年皮肌炎的罕见皮肤表现:眶周水肿和面部肿胀。

IF 1.4 4区 医学 Q3 PEDIATRICS Paediatrics and International Child Health Pub Date : 2024-09-01 Epub Date: 2024-10-02 DOI:10.1080/20469047.2024.2406735
Merve Cansu Polat, Meryem Hilal Altaş, Alkım Öden Akman, Ali Kansu Tehçi, Didem Ardıçlı, Banu Çelikel Acar, Vildan Güngörer
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引用次数: 0

摘要

幼年皮肌炎(JDM)是一种自身免疫性疾病,以肌肉无力和典型的皮肤症状为特征。虽然眶周水肿和面部肿胀与 JDM 的皮肤症状相符,但却极为罕见。据报道,一名 7 岁男孩出现眶周水肿和面部肿胀,但无肌无力。此外,他还伴有胆汁淤积和明显的全血细胞减少,这在 JDM 中并不常见,病因主要考虑为恶性肿瘤和代谢紊乱。除了发病时肌酶升高外,他没有其他肌肉骨骼方面的不适,但在随访期间出现了肌无力,肌肉活检结果与炎症性肌病相符。他对常规治疗反应良好,在随访的第 14 个月时没有出现身体受限或皮肤症状。虽然具有典型临床特征的患者很容易诊断,但非典型皮肤症状对临床医生来说却具有挑战性。如果除了肌酶升高外,还有不典型的皮肤和临床表现,则应在鉴别诊断中考虑 JDM:缩写:AHCE:无症状高钾血症;AST:天冬氨酸氨基转移酶;C:补体;CK:肌酸激酶;IVIG:静脉注射免疫球蛋白;IIM:特发性炎症性肌病;JDM:幼年皮肌炎;LDH:乳酸脱氢酶;MAA:肌炎相关抗体;MDA5:MRC:医学研究委员会;MRI:磁共振成像;MSA:肌炎特异性抗体;MTX:氨甲喋呤 NXP2:核基质蛋白 2;STIR:短 tau 反转恢复;US:超声波。
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Rare skin manifestation of juvenile dermatomyositis: peri-orbital oedema and facial swelling.

Juvenile dermatomyositis (JDM) is an auto-immune disease characterised by muscle weakness and typical skin findings. Although peri-orbital oedema and facial swelling are compatible cutaneous findings in JDM, they are extremely rare. A 7-year-old boy who presented with peri-orbital oedema and facial swelling without muscle weakness is reported. In addition, he had cholestasis and marked cytopenia, which are uncommon in JDM, and malignancy and metabolic disorders were primarily considered in the aetiology. He had no musculoskeletal complaints other than elevated muscle enzymes on presentation but developed muscle weakness during follow-up, and a muscle biopsy was compatible with inflammatory myopathy. He responded favourably to conventional treatment and there were no physical limitations or skin findings by the 14th month of follow-up. Although patients presenting with typical clinical features are easy to diagnose, atypical skin findings are challenging for the clinician. In the presence of atypical skin and clinical findings in addition to muscle enzyme elevation, JDM should be considered in the differential diagnosis.Abbreviations: AHCE: asymptomatic hyper-CKemia; AST: aspartate aminotransferase; C: complement; CK: creatine kinase; IVIG: intravenous immunoglobulin; IIM: idiopathic inflammatory myopathy; JDM: juvenile dermatomyositis; LDH: lactate dehydrogenase; MAA: myositis-associated antibodies; MDA5: melanoma differentiation-associated gene 5; MRC: Medical Research Council; MRI: magnetic resonance imaging; MSA: myositis-specific antibodies; MTX: methotrexate NXP2: nuclear matrix protein 2; STIR: short tau inversion recovery; US: ultrasound.

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来源期刊
CiteScore
3.30
自引率
0.00%
发文量
19
审稿时长
6-12 weeks
期刊介绍: Paediatrics and International Child Health is an international forum for all aspects of paediatrics and child health in developing and low-income countries. The international, peer-reviewed papers cover a wide range of diseases in childhood and examine the social and cultural settings in which they occur. Although the main aim is to enable authors in developing and low-income countries to publish internationally, it also accepts relevant papers from industrialised countries. The journal is a key publication for all with an interest in paediatric health in low-resource settings.
期刊最新文献
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