Objectives We aimed to assess the clinical features of human T-cell leukaemia virus type 1 (HTLV-1)-positive rheumatoid arthritis (RA) patients. Furthermore, we investigated the impact of HTLV-1 infection on incidences of serious infections requiring hospitalisation (SIH) and malignancies. Methods A total of 150 sex- and age-matched HTLV-1-negative and 50 HTLV-1-positive RA patients were enrolled from the HTLV-1 RA Miyazaki Cohort Study. Clinical and laboratory data were collected from this cohort database. The incidence rate (IR) for SIH and malignancies from 2015 to 2020 was analysed. Results The median age and female ratio in the study population were 70 years old and 80%, respectively. Although no differences were found in inflammatory marker values between the two groups, the patient global assessment and Health Assessment Questionnaire scores were higher in HTLV-1-positive RA patients. In HTLV-1-negative RA patients, the IR for SIH was 6.37/100 person-years (PY) and 1.32/100 PY for malignancies. In HTLV-1-positive RA patients, SIH occurred in 11.1/100 PY and malignancies in 2.46/100 PY. The crude IR ratio comparing SIH between two groups was 1.74 (95% confidence interval, 1.04–2.84), which was a significant increase. Conclusions HTLV-1-positive RA patients may worsen RA symptoms. HTLV-1 may be a risk factor for SIH.
Background CD4-positive T cells are the main target of human T-cell leukemia virus type 1 (HTLV-1). Interferon-γ release assays rely on the fact that T-lymphocytes release this cytokine when exposed to tuberculosis-specific antigens and are useful in testing for latent tuberculosis infection before initiating biologic therapy, such as anti-tumor necrosis factor agents. However, the reliability of interferon-γ release assays in detecting tuberculosis infection among HTLV-1-positive patients with rheumatoid arthritis (RA) remains unclear. The present study aimed to evaluate the use of the T-SPOT.TB assay in HTLV-1-positive RA patients. Methods Overall, 29 HTLV-1-positive RA patients and 87 age-and sex-matched HTLV-1-negative RA patients (controls) were included from the HTLV-1 RA Miyazaki Cohort Study. Results of the T-SPOT.TB assay for latent tuberculosis infection screening were collected from medical records of patients. Results Approximately 55% of the HTLV-1-positive RA patients showed invalid T-SPOT.TB assay results (odds ratio: 108, 95% confidence interval: 13.1-890, p < 0.0001) owing to a spot count of >10 in the negative controls. HTLV-1 proviral load values were significantly higher in patients with invalid results compared with those without invalid results (p = 0.003).
BackgroundAutoantibodies (abs) directed against Ro52/TRIM21 are common in systemic sclerosis (SSc) but their clinical significance remains uncertain. Some reports suggested that anti-Ro52/TRIM21 abs-positive SSc patients present interstitial lung disorders (SSc-ILD). However, it is not clear whether positive for anti-Ro52/TRIM21 related to other clinical manifestations in patients with SSc.ObjectivesThe aim of this study is to clarify the prevalence of anti-Ro52/TRIM21 abs in patients with SSc. Then, we investigated the clinical manifestations between anti-Ro52/TRIM2 abs-positive and negative patients with SSc.MethodsThis study is a retrospective case control study. The medical records of 42 patients who were diagnosed as having SSc admitted to our hospital were reviewed. We evaluated the clinical manifestations at the first-onset of SSc such as Rodnan skin score, digital ulcer, abnormal subcutaneous calcification, esophageal reflux, pulmonary hypertension, myositis and arthralgia. Co-existing rheumatic diseases were also reviewed such as Sjogren syndrome (SjS), rheumatoid arthritis (RA), and polymyositis. All subjects underwent SSc –associated abs testing using EUROLINE immunoblot assay. The autoantibodies include anti-Scl-70, anti-centromere A and B (CENP-A, CENP-B), anti-RNA polymerase III (RP-11, RP-155), anti-fibrillarin (U3RNP), anti- 90-kd nucleolar protein (NOR-90), anti-Th/To, anti-PM/Scl-100, anti-PM/Scl-75, anti-Ku, anti-platelet-derived growth factor receptor (PDGFR), and tripartite motif–containing protein 21(Ro-52). The association between clinical features and autoantibody profile was evaluated. Diagnosis of ILD was evaluated by chest high-resolution CT (HRCT).ResultsThirty-five patients with SSc are female (87%). Twenty-one and nineteen patients with SSc are positive and negative for anti-Ro52/TRIM21 abs, respectively (47 v.s 52%). There is no difference in population of diffuse type of SSc between anti-Ro52/TRIM21 abs-positive and negative SSc patients. In addition, the prevalence of ILD is not different between two groups (57 v.s 52%, p=0.76). The prevalence of SjS is tended to be higher in anti-Ro52/TRIM21 abs-positive SSc patients than in negative SSc patients (68 v.s 33%, p=0.05). Unexpectedly, 57% of anti-Ro52/TRIM21 abs-negative SSc patients present poly-arthralgia at the onset of SSc. In addition, there were not any complications such as osteoarthritis and RA in anti-Ro52/TRIM21 abs-negative SSc patients. The prevalence of arthralgia was higher in anti-Ro52/TRIM21 abs-negative SSc patients than in positive SSc patients (p=0.02).ConclusionsThe prevalence of anti-Ro52/TRIM21 abs in SSc patients of this study seems to be high compared to other reports. In addition, there seems to be no difference in the prevalence of ILD regardless of existence of anti-Ro52/TRIM21 abs in SSc patients. Anti-Ro52/TRIM21 abs-negative SSc patients were tended to present poly-arthralgia. This musculoskeletal disorder of anti-Ro52/TRIM21 abs-negative SSc patients may be not associated with other rheumatic diseases s...
Rationale:Kawasaki disease (KD) is an acute febrile illness predominantly affecting children less than 5 years of age and characterized by systemic inflammation in all medium-sized arteries. Adult-onset KD (AKD) is rare with only 105 case reports published. Recently, the efficacy of infliximab (IFX) for patients with refractory KD has been demonstrated.Patient concerns:A previously healthy 24-year-old man was admitted because of a persistent fever, and elevated serum level of AST, ALT, LDH, and CRP.Diagnosis:The patients met the diagnostic criteria for KD based on the findings of persistent fever, polymorphous exanthema, unilateral cervical lymphadenopathy, non-purulent palpebral conjunctivitis and membranous desquamation. Echocardiogram revealed the dilatation at the proximal sites of the right coronary artery (7.9 mm) and left anterior descending artery (5 mm). The patient was treated with high-dose intravenous immunoglobulin (1 g/kg/day for 2 days) and ASA (100 mg daily). However, his fever and arthralgia persisted.Interventions:He was administered single 5 mg/kg doses of IFX.Outcomes:He became afebrile the next day and his arthralgia improved.Lessons:We report the first case of administration of IFX in a patient with AKD refractory to intravenous immunoglobulin (IVIG), and successful reduction of systemic inflammation. However, the effectiveness of IFX in the regression of coronary artery aneurysm remains to be determined.
A 61-year-old woman with human T-cell leukemia virus type 1 (HTLV-1)-associated myelopathy (HAM)/ tropical spastic paraparesis (TSP) and interstitial pneumonia (IP) was admitted to our hospital. She complained of sicca symptoms, polyarthralgia, and swollen joints. She was diagnosed with rheumatoid arthritis (RA) and Sjogren's syndrome. Methotrexate and anti-tumor necrosis factor therapy were not utilized because of the inclusion of severe respiratory disorders among the complications and the neurological symptoms of HAM/TSP. Tocilizumab monotherapy improved the RA disease activity without exacerbating HAM/TSP. The present case suggests that tocilizumab might be a suitable treatment option in patients with RA and HAM/ TSP.
ObjectiveTo compare the findings of muscle magnetic resonance imaging (MRI) between anti-signal recognition particle antibody-positive myopathy (anti-SRP myopathy) and anti-aminoacyl-tRNA synthetase antibody-positive myositis (anti-ARS myositis). Methods Of the patients newly diagnosed with polymyositis (PM)/dermatomyositis (DM) and immune-mediated necrotising myopathy (IMNM) admitted to our Department between April 2012 and December 2021, those who met the eligibility criteria of positive for anti-SRP or anti-ARS antibodies and thigh MRI at the time of diagnosis were included.We compared the lesion sites and MRI findings of the thigh muscles that were classified into oedema, fascial oedema, fatty replacement, and muscle atrophy between the three groups of anti-SRP myopathy, anti-Jo-1 antibody-positive myositis, and non-Jo-1 antibody-positive myositis. Results Of the 98 PM/DM and IMNM patients, five anti-SRP myopathy patients and 11 anti-Jo-1-positive and 22 non-Jo-1antibody-positive patients with myositis were included. The SRP group showed significantly higher blood levels of myogenic enzymes such as serum creatinine kinase (CK) than the other groups (p=0.01). In thigh MRI findings, despite oedema in most cases in anti-SRP and anti-ARS groups, fascial oedema was identified only in the ARS group, frequently in Jo-1 positive patients in particular. Moreover, gluteus maximus muscle lesions occurred more frequently in the SRP group than in the ARS group (p=0.008). Conclusion A comparison of thigh MRI between anti-SRP myopathy and anti-ARS myositis showed different findings and lesionsites reflecting the different pathophysiology that may contribute to their diagnosis.
Neuropsychiatric systemic lupus erythematosus (NPSLE) with cerebral vasculitis is rare, and its prognosis is unfavorable. High-dose glucocorticoids and cyclophosphamide are widely used for the treatment of NPSLE, but cyclophosphamide has a risk of cervical intraepithelial neoplasia and ovarian insufficiency, which may discourage its use in young women. We experienced a case of NPSLE with cerebral vasculitis and lupus nephritis that responded successfully to glucocorticoids and mycophenolate mofetil (MMF). MMF might be a treatment option for NPSLE without concern for reproductive toxicity. However, there are only a few reports on the efficacy of MMF in NPSLE, and further investigations are needed.
Background:In clinical rheumatology, interferon-γ release assays (IGRAs) have been reported as a useful diagnostic test for latent tuberculosis infection (LTBI) before beginning the administration of biologics such as anti-TNF therapies (1). CD4-positive T cells are the main target in Human T-cell leukaemia virus type 1 (HTLV-1) infection. Several reports suggest that the reaction of tuberculin skin test (TST) is attenuated in HTLV-1-positive individuals compared with that in HTLV-1-negative individuals (2). However, it remains unclear whether IGRAs are reliable for detecting TB infection among HTLV-1-positive RA patients.Objectives:The present study aimed to investigate the usefulness of the T-SPOT.TBassay in HTLV-1-positive RA patients. In addition, the association between the existence of IFN-γ producing T cells and HTLV-1 proviral loads (PVLs) in HTLV-1-positive RA patients was analysed on the basis of the T-SPOT.TBassay results.Methods:We reviewed the medical records of 75 HTLV-1-negative and 29 HTLV-1-positive RA patients were suspected cases of LTBI and evaluated using the T-SPOT.TBassay as a clinical practice from April 2012 to July 2019. The results of T-SPOT.TBwere collected from medical records, retrospectively. Peripheral blood samples were obtained from HTLV-1-positive RA patients for the analysis of HTLV-1 PVLs values. The study protocol was approved by the research ethics committees of our hospitals.Results:Approximately 55% of the HTLV-1-positive RA patients showed invalid results for the T-SPOT.TBassay (p < 0.0001); the cause of invalid results was a spot-forming count of >10 spots in the negative controls of the T-SPOT.TBassay among HTLV-1-positive RA patients. Among HTLV-1-positive RA patients, HTLV-1 PVL values were significantly higher in 16 patients who showed invalid results than in 13 patients who did not (p = 0.003). There were no between-group differences in female patient ratio, age, RA disease activity and therapeutic regimens. IFN-γ producing cells were detected in the peripheral blood of HTLV-1-positive RA patients without stimulation with TB-specific antigens.Conclusion:The incidence of invalid results for the T-SPOT.TBassay has been reported to be as low as 0.6% (3). The results of this assay for screening of LTBI in HTLV-1-positive RA patients should be interpreted with caution. Furthermore, our results show that an increase in IFN-γ producing T cell numbers due to HTLV-1 infection in RA patients may affect the pathogenesis of RA.References:[1]Iannone, F., et al.J. Rheumatol. Suppl.91, 41-46 (2014).[2]Tachibana, N., et al.Int. J. Cancer42, 829-831 (1988).[3]Rego, K., et al.Tuberculosis (Edinb.)108, 178-185 (2018).Acknowledgments:We would like to thank Dr Yuki Hashikura and Ms Yuki Kaseda of the University of Miyazaki for their technical support in this work. We would also like to acknowledge Ms Yumiko Kai at the Institute of Rheumatology, Zenjinkai Shimin-no-Mori Hospital, for her help in data management.A part this work was supported by a grant from the Practical Research Project for Rare/Intractable Diseases of the Japan Agency for Medical Research and Development (Grant No. JP19ek0109356), a Health and Labor Sciences Research Grant on Rare and Intractable Diseases from the Ministry of Health, Labor and Welfare of Japan (Grant No. 19FC1007), and a Grant-in-Aid for Clinical Research from Miyazaki University Hospital.Disclosure of Interests:Kunihiko Umekita Paid instructor for: Astellas Pharma Inc. Chugai Pharma Inc. Tanabe-Mitsubishi Pharma Inc., Speakers bureau: Bristol-Myers Squibb, Yayoi Hashiba: None declared, Risa Kudou: None declared, Shunichi Miyauchi: None declared, Masatoshi Kimura: None declared, Motohiro Matsuda: None declared, Chihiro Iwao: None declared, Yumi Kariya: None declared, Takeshi Kawaguchi: None declared, Katoko Takajo: None declared, Koushou Iwao: None declared, Yuuki Rikitake: None declared, Ichiro Takajo: None declared, Toshihiko Hidaka Paid instructor for: Astellas Pharma Inc. Chugai Pharma Inc. Tanabe-Mitsubishi Pharma Inc., Speakers bureau: Astellas Pharma Inc. Chugai Pharma Inc. Tanabe-Mitsubishi Pharma Inc., Akihiko Okayama: None declared
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