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1.
Orv Hetil ; 160(24): 952-957, 2019 Jun.
Artigo em Húngaro | MEDLINE | ID: mdl-31433234

RESUMO

We report a case of a 41-year-old female patient presenting with watery diarrhoea and myalgia in the winter-season. Before her symptoms started she had participated in a pig slaughtering with her family. Some of the family members also became ill. On her physical examination periorbital odema and myalgia were found. Eosinophilia, hypalbuminaemia, elevated lactate dehydrogenase and creatin kinase levels were detected on laboratory investigations. The clinical picture, the laboratory findings and background epidemiological data implied the diagnosis of trichinellosis and albendazol was started. Serum gained on the 22nd post-infectious day turned out to be equivocal for trichinellosis. For this reason and because of the refractory fever a muscle-biopsy was done. Granulomatous myositis described by histology and Trichinella seropositivity from the repeated serum sample on the 62nd post-infectious day finally confirmed the diagnosis. During the course of the disease, we experienced elevation of troponin I suggesting myocarditis, but it was accompanied neither with abnormal ECG signs nor characteristic symptoms. Almost a century ago, a case report was published in Hungarian with a similar introduction. Trichinellosis in that epidemic setting led to the death of five people. Orv Hetil. 2019; 160(24): 952-957.


Assuntos
Diarreia/etiologia , Febre/etiologia , Mialgia/etiologia , Miosite/etiologia , Trichinella/isolamento & purificação , Triquinelose/diagnóstico , Adulto , Albendazol/uso terapêutico , Animais , Anti-Helmínticos/uso terapêutico , Eosinofilia/etiologia , Feminino , Humanos , Músculo Esquelético/parasitologia , Miosite/tratamento farmacológico , Miosite/parasitologia , Estações do Ano , Suínos , Resultado do Tratamento , Triquinelose/sangue , Triquinelose/tratamento farmacológico
2.
J Med Case Rep ; 13(1): 241, 2019 Aug 04.
Artigo em Inglês | MEDLINE | ID: mdl-31376837

RESUMO

BACKGROUND: Antisynthetase syndrome is a relatively uncommon entity, and can be easily missed if not specifically looked for in adults whose initial presentation is with interstitial lung disease. Its presentation with interstitial lung disease alters its prognosis. CASE PRESENTATION: This case report describes a 27-year-old Pakistani, Asian man, a medical student, with no previous comorbidities or significant family history who presented with a 3 months' history of low grade fever and lower respiratory tract infections, associated with exertional dyspnea, arthralgias, and gradual weight loss. During these 3 months, he had received multiple orally administered antibiotics for suspected community-acquired pneumonia. When he presented to us, he was pale and febrile. A chest examination was significant for bi-basal end-inspiratory crackles. Preliminary investigations revealed raised erythrocyte sedimentation rate. High resolution computed tomography of his chest showed fine ground-glass attenuation in posterior basal segments of both lower lobes suggestive of interstitial lung disease. He was started on dexamethasone, to which he responded and showed improvement. However, during the course of events, he developed progressive proximal muscle weakness. Further investigations revealed raised creatinine phosphokinase and lactate dehydrogenase. A thorough autoimmune profile was carried out which showed positive anti-Jo-1 antibodies in high titers. A muscle biopsy was consistent with inflammatory myopathy. Clinical, radiological, serological, and histopathological markers aided in making the definitive diagnosis of antisynthetase syndrome. Antisynthetase syndrome is a variant of polymyositis but with visceral involvement, that is, interstitial lung disease and positive anti-Jo-1 antibodies. Our patient responded very well to glucocorticoids and azathioprine. CONCLUSION: Antisynthetase syndrome is a rare clinical entity which apart from clinical presentation requires specific serological investigations for diagnosis. Concomitant association of interstitial lung disease gives it a guarded prognosis.


Assuntos
Miosite/diagnóstico , Adulto , Anticorpos Antinucleares/sangue , Autoanticorpos/sangue , Azatioprina/administração & dosagem , Creatina Quinase/sangue , Diagnóstico Diferencial , Dispneia/etiologia , Humanos , Doenças Pulmonares Intersticiais/diagnóstico , Masculino , Debilidade Muscular/etiologia , Miosite/tratamento farmacológico , Prednisolona/administração & dosagem , Prednisolona/análogos & derivados
3.
Acta Myol ; 38(1): 13-16, 2019 03.
Artigo em Inglês | MEDLINE | ID: mdl-31309176

RESUMO

Scleromyxedema is a rare cutaneous mucinosis with frequent extracutaneous manifestations. Myopathy in scleromyxedema is a poorly recognized syndrome among neurologists and can mimic idiopathic and connective tissue disease-associated inflammatory myopathy. Diagnosis is suspected by the characterization of the skin lesions and clinched by skin and muscle biopsies. Here, we report a patient with scleromyxedema and myopathy with the characteristic histopathological feature of mucin deposition in skin biopsy. Her muscle biopsy showed a picture consistent with scleromyxedema myopathy with vacuolar and inflammatory changes. The association with paraproteinemia, propensity to life-threatening central nervous system disease and good response to intravenous immunoglobulin necessitate the accurate diagnosis of this condition.


Assuntos
Miosite/diagnóstico , Escleromixedema/diagnóstico , Adulto , Biópsia , Diagnóstico Diferencial , Quimioterapia Combinada , Feminino , Glucocorticoides/uso terapêutico , Humanos , Imunoglobulinas Intravenosas/uso terapêutico , Imunossupressores/uso terapêutico , Miosite/tratamento farmacológico , Prednisolona/uso terapêutico , Escleromixedema/tratamento farmacológico , Talidomida/uso terapêutico
4.
Am J Case Rep ; 20: 1094-1103, 2019 Jul 25.
Artigo em Inglês | MEDLINE | ID: mdl-31344020

RESUMO

BACKGROUND With the advent and advancement of autoantibodies, there has been progress in the diagnosis, prognosis, and treatment of rheumatologic diseases. Antisynthetase syndrome (ASS) is a great example of a disease that initially was described as arthritis, myositis, interstitial lung disease, mechanic's hands, Raynaud's phenomenon, and fever in the presence of the anti-JO-1 antibody, but nowadays it presents with a different spectrum related to new antibodies. CASE REPORT We describe 4 patients with antisynthetase syndrome who were diagnosed with antibodies specific for myositis associated with different clinical findings. All patients responded to immunosuppressive therapy, and rituximab was the most used. CONCLUSIONS It is necessary to search for specific autoantibodies related to the syndrome in suspected clinical cases and in other rheumatological diseases refractory to the usual treatment.


Assuntos
Autoanticorpos/sangue , Miosite/diagnóstico , Adulto , Feminino , Glucocorticoides/uso terapêutico , Humanos , Imunossupressores/uso terapêutico , Masculino , Metotrexato/uso terapêutico , Miosite/tratamento farmacológico , Prednisona/uso terapêutico , Doenças Raras
5.
Rev Med Chil ; 147(3): 342-355, 2019 Mar.
Artigo em Espanhol | MEDLINE | ID: mdl-31344172

RESUMO

Idiopathic inflammatory myopathies (IIM) are a heterogeneous group of acquired immune-mediated diseases, which typically involve the striated muscle with a variable involvement of the skin and other organs. Clinically, they are characterized by proximal muscle weakness, elevation of muscle enzymes, myopathic changes on electromyography and an abnormal muscle biopsy. The different IIM have been classified according to their distinctive histopathologic features in dermatomyositis (DM), polymyositis (PM), inclusion body myositis (IBM) and immune-mediated necrotizing myopathy (IMNM). Several myositis-specific antibodies are associated with the different phenotypes, as well as with different risk of neoplastic disease and systemic complications. The basis for the treatment of DM, PM, and IMNM is immunosuppression. For IBM there are only symptomatic treatments. Steroids, associated or not with other immunosuppressant drugs, are the first line of treatment. Biologic drugs will allow future individualized therapies. The 10-year survival of DM, PM and IMNM is 62 to 90%. The leading causes of death are neoplastic, lung and cardiac complications. IBM does not impair survival, although it affects the quality of life.


Assuntos
Miosite/patologia , Anticorpos , Dermatomiosite/patologia , Eletromiografia , Humanos , Imunossupressores/classificação , Imunossupressores/uso terapêutico , Músculo Esquelético/patologia , Miosite/tratamento farmacológico , Polimiosite/patologia
6.
Acta Reumatol Port ; 44(1): 88-89, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31249282

RESUMO

We present a case of recurrent focal myositis treated with intravenous human immunoglobulin, after ineffectiveness of analgesics, non-steroidal anti-inflammatory drugs, corticosteroids and immunosuppressive therapies. There was a prompt clinical and laboratorial improvement. To the best of our knowledge, this is the first case reporting the administration of this treatment in recurrent focal myositis.


Assuntos
Imunoglobulinas Intravenosas/uso terapêutico , Fatores Imunológicos/uso terapêutico , Miosite/terapia , Adulto , Azatioprina/uso terapêutico , Feminino , Glucocorticoides/uso terapêutico , Humanos , Metotrexato/uso terapêutico , Miosite/tratamento farmacológico , Prednisolona/uso terapêutico , Recidiva
7.
Autoimmun Rev ; 18(7): 658-664, 2019 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-31059843

RESUMO

Anti-OJ autoantibodies are rare myositis-specific autoantibodies that have been described to target isoleucyl-tRNA synthetase. Routinely used multiplex assays perform poorly in detection of anti-OJ antibodies. In this manuscript, we review the existing literature on critical issues in detection of anti-OJ and the clinical features associated with anti-OJ. The challenging detection with line/blot immunoassays and ELISAs is most likely related to the characteristics of the autoantigen involved, which is part of a multi-enzyme synthetase complex. Anti-OJ autoantibodies might therefore be more aptly termed anti-OJ complex autoantibodies. Anti-OJ autoantibodies are associated with the anti-synthetase syndrome, with interstitial lung disease (ILD) frequently being the sole manifestation. Myositis, present in the majority of patients with anti-OJ antibodies, is more severe than in patients with other anti-aminoacyl-tRNA synthetases. Most patients respond to glucocorticoid therapy. As detection of anti-OJ is relevant for treatment, reliable and practical detection is needed. Meanwhile, clinicians need to be aware of the possibility of anti-OJ in patients with ILD, isolated or in combination with myositis.


Assuntos
Autoanticorpos/imunologia , Isoleucina-tRNA Ligase/imunologia , Humanos , Doenças Pulmonares Intersticiais/tratamento farmacológico , Doenças Pulmonares Intersticiais/imunologia , Miosite/tratamento farmacológico , Miosite/imunologia
9.
Clin Exp Rheumatol ; 37(5): 858-861, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31074729

RESUMO

OBJECTIVES: To study the efficacy in terms of muscle strength, and corticosteroid tapering as well as the prevalence of adverse effects in patients with the antisynthetase syndrome (ASyS) treated with azathioprine (AZA) compared to those treated with methotrexate (MTX). METHODS: We compared the clinical outcomes in ASyS patients treated with AZA versus MTX including change in corticosteroid dose, strength, and creatine kinase (CK) as well as the prevalence of adverse effects. RESULTS: Among 169 patients with ASyS, 102 were treated at some point exclusively with either AZA or MTX (± corticosteroids). There were no significant differences in the rate of muscle strength recovery, CK decrease or corticosteroid tapering between those ASyS patients treated with MTX versus AZA. The prevalence of adverse events in patients treated with AZA and MTX was similar (29% vs. 25%, p>0.05); elevated liver enzymes (17% AZA vs. 12% MTX) and gastrointestinal involvement (10% AZA vs. 8% MTX) were the most common adverse events. While no patients treated with AZA developed lung complications, two of the patients treated with MTX experienced reversible pneumonitis with MTX cessation. CONCLUSIONS: AZA and MTX showed similar efficacy and adverse events in patients with ASyS. Pneumonitis is a rare but important event in patients receiving MTX.


Assuntos
Azatioprina , Metotrexato , Miosite/tratamento farmacológico , Corticosteroides , Azatioprina/efeitos adversos , Azatioprina/uso terapêutico , Creatina Quinase/sangue , Humanos , Metotrexato/efeitos adversos , Metotrexato/uso terapêutico , Força Muscular/efeitos dos fármacos , Força Muscular/fisiologia
10.
Rinsho Shinkeigaku ; 59(6): 360-364, 2019 Jun 22.
Artigo em Japonês | MEDLINE | ID: mdl-31142711

RESUMO

An 84-year-old woman developed blepharoptosis, diplopia, weakness of extremities, and dysphagia with elevation of serum CK levels after treatment with nivolumab against renal cell carcinoma. 3 Hz repetitive stimulation showed waning in the trapezius muscle, leading to the diagnosis of myasthenia gravis. Laboratory examination showed that anti-acetylcholine receptor antibody was negative. We performed IVIg and steroid therapy. However, her symptoms did not improve, and she died of respiratory failure, although serum CK levels ameliorated to the normal range. The results of autopsy showed atrophy of muscle fibers and massive infiltration of inflammatory cells in the endomysium of the iliopsoas muscle and diaphragm, indicating occurrence of myositis. Immunohistochemical analysis showed that CD8-positive T cells mainly infiltrates in the endomysium with a small number of CD4-potive T cells. Here, we report an autopsy case of nivolumab-induced myasthenia gravis and myositis.


Assuntos
Antineoplásicos Imunológicos/efeitos adversos , Miastenia Gravis/induzido quimicamente , Miastenia Gravis/patologia , Miosite/induzido quimicamente , Miosite/patologia , Nivolumabe/efeitos adversos , Idoso de 80 Anos ou mais , Antineoplásicos Imunológicos/uso terapêutico , Autopsia , Linfócitos T CD4-Positivos/patologia , Linfócitos T CD8-Positivos/patologia , Carcinoma de Células Renais/tratamento farmacológico , Creatina Quinase/sangue , Evolução Fatal , Feminino , Humanos , Imunoglobulinas Intravenosas/administração & dosagem , Neoplasias Renais/tratamento farmacológico , Miastenia Gravis/diagnóstico , Miastenia Gravis/tratamento farmacológico , Miosite/diagnóstico , Miosite/tratamento farmacológico , Nivolumabe/uso terapêutico , Insuficiência Respiratória/induzido quimicamente
11.
Pan Afr Med J ; 32: 40, 2019.
Artigo em Francês | MEDLINE | ID: mdl-31143345

RESUMO

Anti-synthetase syndrome (ASS) is an inflammatory myopathy commonly associated with pulmonary involvement, especially parenchymal (diffuse infiltrative pneumonitis). Extrathoracic manifestations associated with pulmonary involvement can give an indication to the diagnosis: myalgias, polyarthralgias, Raynaud's syndrome, erythematous palmar hyperkeratosis with fissures and fever. Given the suggestive clinical and radiological picture, the presence of aminoacyl-transfer RNA (tRNA) synthetase antibodies enables to confirm, in particular, Anti Jo-1 antibody activity. Pulmonary involvement is a major prognostic factor, hence the indication for intensive immunosuppressive therapy based on corticosteroids, immunosuppressive medications or the association among them. A better awareness about this disorder revealed by pulmonary manifestations could enable early and adequate management and to improve patient's prognosis.


Assuntos
Imunossupressores/administração & dosagem , Doenças Pulmonares Intersticiais/etiologia , Miosite/diagnóstico , Corticosteroides/administração & dosagem , Adulto , Idoso , Anticorpos Antinucleares/imunologia , Feminino , Humanos , Doenças Pulmonares Intersticiais/tratamento farmacológico , Doenças Pulmonares Intersticiais/fisiopatologia , Masculino , Miosite/tratamento farmacológico , Miosite/imunologia , Prognóstico
12.
BMJ Case Rep ; 12(4)2019 Apr 16.
Artigo em Inglês | MEDLINE | ID: mdl-30996070

RESUMO

A 61-year-old Hispanic man presented to a county hospital for subacute progressive weakness, heliotrope rash and dysphagia. There was initial suspicion for dermatomyositis (DM) given the history; however, the physical exam was not consistent. An MRI followed by a muscle biopsy revealed necrotising autoimmune myositis and anti-3-hydroxy-3-methylglutary-coenzyme A-reductase antibody titers returned positive; the patient was diagnosed with necrotising autoimmune myositis. He was treated with corticosteroids and intravenous immunoglobulin, which resulted in improvement in his weakness and functional status. This case represents a unique instance in which a cardinal feature of DM, the heliotrope rash, prompted an erroneous initial diagnosis. It highlights the necessity of developing abroad differential diagnosis and subsequent thorough investigation into patients presenting with suspected idiopathic immune-mediated myopathies.


Assuntos
Corticosteroides/uso terapêutico , Hispano-Americanos , Imunoglobulinas Intravenosas/uso terapêutico , Fatores Imunológicos/uso terapêutico , Músculo Esquelético/patologia , Miosite/diagnóstico , Necrose/patologia , Autoanticorpos/sangue , Humanos , Masculino , Pessoa de Meia-Idade , Miosite/tratamento farmacológico , Miosite/fisiopatologia , Necrose/tratamento farmacológico , Resultado do Tratamento
13.
Asia Pac J Clin Oncol ; 15(4): 266-269, 2019 Aug.
Artigo em Inglês | MEDLINE | ID: mdl-30815986

RESUMO

We report on the unique case of a patient with antisynthetase syndrome and metastatic non-small cell lung cancer undergoing therapy with the PD-1 checkpoint inhibitor, nivolumab. Despite adequate autoimmune disease control over a period of 12 months, the patient rapidly experienced a flare of interstitial lung disease following initial nivolumab administration, which ultimately proved fatal. The use of immune checkpoint inhibitors in patients with autoimmune disease is becoming more commonplace, however this is the first reported case of the use of these agents in a patient with antisynthetase syndrome. Additionally, the patient's initial clinical presentation with antisynthetase syndrome and simultaneous primary lung cancer, a rare association of which there are few case reports, makes this case interesting and unusual.


Assuntos
Carcinoma Pulmonar de Células não Pequenas/complicações , Carcinoma Pulmonar de Células não Pequenas/tratamento farmacológico , Neoplasias Pulmonares/complicações , Neoplasias Pulmonares/tratamento farmacológico , Miosite/tratamento farmacológico , Receptor de Morte Celular Programada 1/antagonistas & inibidores , Feminino , Humanos , Pessoa de Meia-Idade , Miosite/patologia
15.
Rheumatol Int ; 39(7): 1201-1212, 2019 07.
Artigo em Inglês | MEDLINE | ID: mdl-30778655

RESUMO

To evaluate the relevance of immunoglobulin (IVIg) and/or methylprednisolone pulse therapies in immune-mediated necrotizing myopathy (IMNM). Secondarily, to analyze the muscle damage measured by late magnetic resonance images (MRI). This retrospective study included 13 patients with defined IMNM (nine patients positive for the anti-signal recognition particle and four patients positive for hydroxyl-methyl-glutaryl coenzyme A reductase) who were followed from 2012 to 2018. International Myositis Assessment and Clinical Studies Group (IMACS) scoring assessed the response to a standardized treat-to-target protocol with disease activity core-set measures and late magnetic resonance imaging (MRI). The patients had a mean age of 53.5 years and were predominantly female and of white ethnicity. Median symptom and mean follow-up durations were 4 and 39 months, respectively. All patients received IVIg and/or methylprednisolone pulse therapies. All IMACS core-set measurements improved significantly after initial treatment. Nine patients achieved complete clinical response and among them 2 had complete remission. Eleven patients had discontinued glucocorticoid use by the end of the study. Only 2 patients had moderate muscle atrophy or fat replacement observed by MRI, with the remainder presenting normal or mild findings. Our patients with IMNM treated with an aggressive immunosuppressant therapy had a marked improvement in all IMACS core-set domains. Moreover, the MRI findings suggest that an early treat-to-target approach could reduce the odds of long-term muscle disability. Methylprednisolone and/or IVIg pulse therapies aiming at a target of complete clinical response are potential treatment strategies for IMNM that should be studied in future prospective studies.


Assuntos
Glucocorticoides/uso terapêutico , Imunoglobulinas Intravenosas/uso terapêutico , Fatores Imunológicos/uso terapêutico , Metilprednisolona/uso terapêutico , Miosite/tratamento farmacológico , Adulto , Idoso , Quimioterapia Combinada , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Estudos Retrospectivos , Resultado do Tratamento
16.
Medicina (Kaunas) ; 55(1)2019 Jan 17.
Artigo em Inglês | MEDLINE | ID: mdl-30658484

RESUMO

Background and objectives: Acute proximal muscle weakness has a broad differential. Infectious myositis is difficult to differentiate clinically from inflammatory myopathy, often causing a delayed diagnosis. Infectious myositis should be thought of as a differential for proximal muscle pain and weakness in the right context. Case Presentation: A 40-year-old male with diabetes presented with exquisite pain and weakness of proximal extremities. He denied trauma, recent travel, new medications, or substance use. He denied prior rheumatologic, thyroid, or musculoskeletal disorders. The urine culture revealed staphylococcal infection with negative blood cultures. Rheumatologic and endocrine workups were negative. Random muscle biopsy was negative for inflammatory infiltrate. MRI of thighs and arms showed innumerable foci of nodular and ring enhancement in the proximal muscle groups. The patient noted improvement after about 10 days of antibiotics with complete resolution at 2 months. Discussion and Conclusion: Bacterial myositis is most often due to Staphylococcus aureus (70%) and affects a single muscle. Multifocal abscesses are rare and strongly suggest transient bacteremia. Our patient most likely had transient initiating staphylococcal bacteremia leading to diffuse myositis and hematogenous urinary tract infection (UTI). A delay in treatment can be life-threatening.


Assuntos
Antibacterianos/uso terapêutico , Debilidade Muscular/diagnóstico , Mialgia/diagnóstico , Miosite/diagnóstico , Miosite/tratamento farmacológico , Infecções Urinárias/diagnóstico , Infecções Urinárias/tratamento farmacológico , Doença Aguda , Adulto , Assistência ao Convalescente , Diagnóstico Tardio , Complicações do Diabetes , Diagnóstico Diferencial , Serviço Hospitalar de Emergência , Humanos , Tempo de Internação , Imagem por Ressonância Magnética , Masculino , Debilidade Muscular/diagnóstico por imagem , Mialgia/diagnóstico por imagem , Miosite/diagnóstico por imagem , Miosite/etiologia , Fatores de Risco , Infecções Estafilocócicas/urina , Coxa da Perna/diagnóstico por imagem , Coxa da Perna/fisiopatologia , Resultado do Tratamento , Infecções Urinárias/urina
17.
Clin Exp Rheumatol ; 37(5): 740-747, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-30620290

RESUMO

OBJECTIVES: To assess the efficacy and safety of tacrolimus in combination with corticosteroids in patients with immune-mediated necrotising myopathy. METHODS: The medical records of 20 hospitalised patients with immune-mediated necrotising myopathy (IMNM) who had received tacrolimus combined with oral prednisone from January 2014 to August 2017 were retrospectively reviewed. The recruited patients were shifted to the combined therapy because they failed to respond well to monotherapy with oral prednisone. The clinical efficacy during an average follow-up of 21 months (range, 14-24 months) was assessed by evaluating the changes of serum creatine kinase (CK) levels, the Medical Research Council (MRC) grading of the weakest muscle groups and dosage of oral prednisone. Adverse effects were monitored to assess the safety of tacrolimus. RESULTS: After starting tacrolimus, most of the 20 patients showed significant improvement in muscle strength and remarkable decline in serum CK levels at the follow-up points (p<0.0001). In addition, the daily dosage of prednisone was statistically significantly reduced (p<0.0001) after the combination therapy. No serious adverse events attributable to tacrolimus occurred in the patients. CONCLUSIONS: Early co-administration of tacrolimus with corticosteroid promoted the remission and recovery of patients with IMNM and seemed to be a relatively safe treatment programme for physician managing immune-mediated necrotising myopathy.


Assuntos
Corticosteroides/uso terapêutico , Imunossupressores/uso terapêutico , Miosite , Tacrolimo , Creatina Quinase/sangue , Quimioterapia Combinada , Humanos , Miosite/tratamento farmacológico , Prednisona , Estudos Retrospectivos , Tacrolimo/uso terapêutico , Resultado do Tratamento
18.
Rinsho Shinkeigaku ; 59(1): 21-26, 2019 Jan 30.
Artigo em Japonês | MEDLINE | ID: mdl-30606993

RESUMO

A 69-year-old man was admitted with neck muscle weakness, symmetric proximal muscle weakness, skin rash and elevated serum creatine kinase levels. Muscle biopsy showed perifascicular necrosis and perimysial alkaline phosphatase activity. Chest CT revealed interstitial lung disease and colorectal cancer was diagnosed on colonoscopy. He was serologically positive for anti-EJ antibody, leading to the diagnosis of antisynthetase syndrome (ASS). After laparoscopic low anterior resection of the rectum, he received intravenous methylprednisolone (1,000 mg/d for 3 days) followed by oral prednisolone (50 mg/d). Although his muscle weakness improved after corticosteroid therapy, he developed pericardial effusion with resultant asymptomatic hypotension and arrhythmia possibly due to pericarditis. Corticosteroid monotherapy was insufficient to control the disease, and, we decided to use oral cyclosporin concurrently. After this combined therapy started, pericardial effusion and arrhythmia were improved. We should keep in mind that pericarditis can occur in patients with anti-EJ antibody-positive ASS, and early combined therapy with corticosteroid and immunosuppressive drugs for ASS may improve the patient's prognosis.


Assuntos
Autoanticorpos/sangue , Glicina-tRNA Ligase/imunologia , Miosite/complicações , Miosite/imunologia , Pericardite/etiologia , Idoso , Biomarcadores/sangue , Ciclosporina/administração & dosagem , Progressão da Doença , Humanos , Masculino , Metilprednisolona/administração & dosagem , Miosite/diagnóstico , Miosite/tratamento farmacológico , Pericardite/tratamento farmacológico , Prednisolona/administração & dosagem , Pulsoterapia , Neoplasias Retais/complicações , Neoplasias Retais/cirurgia , Resultado do Tratamento
19.
Int J Hematol ; 109(3): 356-360, 2019 Mar.
Artigo em Inglês | MEDLINE | ID: mdl-30604316

RESUMO

Although programmed cell death (PD)-1 blockade induces immune-related adverse events (irAEs), little is known about the safety of PD-1 blockade after allogeneic hematopoietic stem cell transplantation (HSCT). Here, we describe immune system changes during nivolumab-related myositis in a patient with Hodgkin's lymphoma after allogeneic HSCT; to our knowledge, this is the first such report in the literature. At the onset of myositis, the patient lost lower limb mobility against gravity, and had an activated immune profile with increased cytotoxic CD107a and granzyme B expression, as well as pro-inflammatory cytokines, interferon-γ, tumor necrosis factor-α, interleukin-2 in T and NK cells compared to healthy donor. Pulse steroid therapy decreased creatine kinase levels and induced PD-1 expression and regulatory T cells, but did not improve myositis; previously activated markers remained high. Four-week corticosteroid therapy decreased previously activated markers and the myositis improved. These findings provide new insights into nivolumab-induced irAE pathogenesis and suggest possible optimal treatments for irAEs.


Assuntos
Doença de Hodgkin , Miosite , Nivolumabe , Linfócitos T Reguladores/imunologia , Corticosteroides/imunologia , Adulto , Aloenxertos , Citocinas/imunologia , Transplante de Células-Tronco Hematopoéticas , Doença de Hodgkin/imunologia , Doença de Hodgkin/patologia , Doença de Hodgkin/terapia , Humanos , Células Matadoras Naturais/imunologia , Masculino , Miosite/induzido quimicamente , Miosite/tratamento farmacológico , Miosite/imunologia , Miosite/patologia , Nivolumabe/administração & dosagem , Nivolumabe/efeitos adversos , Receptor de Morte Celular Programada 1/imunologia , Linfócitos T Reguladores/patologia
20.
Clin Rheumatol ; 38(2): 585-590, 2019 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-30218287

RESUMO

To evaluate the efficacy and tolerability of mycophenolate mofetil (MMF) with or without calcineurin inhibitors (CNIs) in patients with inflammatory myopathy taking prednisolone, but refractory to conventional immunosuppressive therapy. The records of patients with inflammatory myopathy who had previously failed treatment with at least one immunosuppressant were retrospectively evaluated. We selected patients treated with MMF and divided them into two groups depending on whether or not there was concomitant use of CNIs. We investigated the efficacy by changes in creatine kinase (CK) levels, forced vital capacity (%FVC), prednisolone dose, and high-resolution computed tomography (HRCT) findings. Interstitial lung disease (ILD) progression was defined by more than 10% decline of %FVC from baseline. We identified 19 patients on MMF treatment. There were seven (36.8%) patients on MMF and CNIs, including five on cyclosporine and two on tacrolimus. At baseline, no significant difference was seen in the prevalence of ILD between patients taking or not taking CNIs (85.7% vs. 75.0%, P = 0.68). Improvement in CK was seen in patients treated with CNIs (P = 0.04) but not in those without (P = 0.39). No significant improvement in %FVC and HRCT findings were found in patients with ILD in either group, and there were no differences in death or ILD progression. The combination of CNIs and MMF might be more effective for decreasing CK levels than MMF alone. Neither treatment arm had a beneficial effect on ILD over a variable observation period.


Assuntos
Antibióticos Antineoplásicos/uso terapêutico , Inibidores de Calcineurina/uso terapêutico , Ácido Micofenólico/uso terapêutico , Miosite/tratamento farmacológico , Adulto , Idoso , Quimioterapia Combinada , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Miosite/complicações , Estudos Retrospectivos , Resultado do Tratamento
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