Tacrolimus in refractory juvenile dermatomyositis: Case report and review of literature

Rao, Anand Prahalad; Kolli, Vinyasa; Rajarathinam, Indhuja; Raghuram, Jyothi

doi:10.4103/injr.injr_128_20

Cited by 2 publications

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“…Evidence for the use of azathioprine comes from historical studies that included small numbers of patients, and although this drug can be used as an adjunctive treatment, it has become less favoured over the last two decades for the treatment of IIM in paediatric practice 147 , 148 . Some evidence is available on the use of tacrolimus to treat JIIM but is limited by the small number of patients involved 149 – 151 . Adult data suggest that tacrolimus or ciclosporin alongside corticosteroids should be considered for patients with myositis-associated ILD, and although these data are often extrapolated to JIIM, insufficient data are available to form evidence-based recommendations for this complication in childhood-onset disease 133 .…”

Section: Managementmentioning

confidence: 99%

Juvenile idiopathic inflammatory myositis: an update on pathophysiology and clinical care

et al. 2023

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The childhood-onset or juvenile idiopathic inflammatory myopathies (JIIMs) are a heterogenous group of rare and serious autoimmune diseases of children and young people that predominantly affect the muscles and skin but can also involve other organs, including the lungs, gut, joints, heart and central nervous system. Different myositis-specific autoantibodies have been identified that are associated with different muscle biopsy features, as well as with different clinical characteristics, prognoses and treatment responses. Thus, myositis-specific autoantibodies can be used to subset JIIMs into sub-phenotypes; some of these sub-phenotypes parallel disease seen in adults, whereas others are distinct from adult-onset idiopathic inflammatory myopathies. Although treatments and management have much improved over the past decade, evidence is still lacking for many of the current treatments and few validated prognostic biomarkers are available with which to predict response to treatment, comorbidities (such as calcinosis) or outcome. Emerging data on the pathogenesis of the JIIMs are leading to proposals for new trials and tools for monitoring disease.

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Section: Managementmentioning

confidence: 99%

Juvenile idiopathic inflammatory myositis: an update on pathophysiology and clinical care

et al. 2023

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Methotrexate/methylprednisolone/prednisolone

2021

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In a case report, a 8-year-old boy and 11-year-old boy were described, who exhibited lack of efficacy to methotrexate, methylprednisolone and prednisolone while receiving treatment for juvenile dermatomyositis (JDM) [not all routes stated].Case 1: An 11-year-old boy, who had JDM, started receiving treatment with IV pulse methylprednisolone 30 mg/kg/dose for 3 days followed by oral prednisolone 1 mg/kg daily along with parenteral methotrexate at 15 mg/m 2 weekly. Despite treatment with steroids for a period of 4 weeks along with methotrexate, worsening of his condition was noted along with recurrent fever spikes, persistent muscle weakness. His childhood myositis assessment scale (CMAS) score was 22/52. Also, he developed hypoalbuminemia and anasarca along with severe abdominal pain, which were consistent with a possible gastrointestinal vasculitis. Therefore, tacrolimus was added in the treatment regimen. This resulted in improvement in the first 4 weeks of treatment with resolution of oedema. Also, improvement in muscle power and in truncal and neck flexor weakness was noted. His CAMS score was improved to 52/52. Treatment with steroids and methotrexate was tapered initially and then discontinued.Case 2: An 8-year-old boy, who had JDM, started receiving treatment with methylprednisolone pulse therapy followed by oral prednisolone 1 mg/kg daily and methotrexate 15 mg/m 2 weekly. Despite receiving therapy for 2 months, he had persistent skin and muscle disease activity. His Manual Muscle Testing-8/childhood myositis assessment scale (MT8/CAMS) score was 30/100. Therefore, mycophenolate mofetil 600 mg/m 2 /dose in twice-daily dose was added to the treatment regimen. He continued to receive methylprednisolone pulses at 30 mg/kg for 3 days every month along with prednisolone and methotrexate. In spite of this treatment, his cutaneous disease activity and persistent myositis were continued. Also, his hybrid MT8/CMAS score was 45/100. As a result, he was started on tacrolimus. This resulted in improvement in his condition, and the dose of prednisolone was reduced to 0.5 mg/kg daily. His cutaneous disease was noted to be disappeared. After 18 months of tacrolimus initiation, his hybrid MMT8/ CMAS score was 70/100 with no evidence of cutaneous disease activity and was receiving 0.4 mg/kg of oral prednisolone along with stable doses of methotrexate and mycophenolate mofetil.

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Tacrolimus in refractory juvenile dermatomyositis: Case report and review of literature

Cited by 2 publications

References 0 publications

Juvenile idiopathic inflammatory myositis: an update on pathophysiology and clinical care

Juvenile idiopathic inflammatory myositis: an update on pathophysiology and clinical care

Methotrexate/methylprednisolone/prednisolone

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