Publications (13)16.65 Total impact
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Article: Mycophenolate mofetil therapy for rapidly progressive interstitial lung disease in a patient with clinically amyopathic dermatomyositis.
Modern Rheumatology 04/2013; · 1.58 Impact Factor -
Article: Systemic lupus erythematosus complicated by Crohn's disease: a case report and literature review.
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ABSTRACT: BACKGROUND: Although patients with systemic lupus erythematosus (SLE) may experience various gastrointestinal disorders, SLE and Crohn's disease (CD) rarely coexist. The diseases may have gastrointestinal (GI) manifestations, laboratory results, and radiographic findings that appear similar and consequently differentiating between GI involvement in CD and in SLE may be difficult. We present the case of a patient with SLE and CD who developed continuous GI bleeding and diarrhea that was initially treated as SLE-related colitis to little effect. CASE PRESENTATION: A 55-year-old Japanese woman with systemic lupus erythematosus (SLE) developed continuous gastrointestinal bleeding and diarrhea since the patient was aged 30 years that was initially treated as SLE-related colitis. Although a longitudinal ulcer and aphthous ulcers in the colon were observed every examination, biopsy showed only mild inflammation and revealed neither granuloma nor crypt abscess. The patient underwent surgery for anal fistulas twice at 50 and 54 years of age and her symptoms were atypical of lupus enteritis. Colonoscopy was performed again when the patient was 55 years of age because we suspected she had some type of inflammatory bowel disease (IBD). Cobblestone-like inflammatory polyps and many longitudinal ulcers were detected between the descending colon and the cecum. Macroscopic examination strongly suggested CD. Histopathological examination revealed non-caseating granuloma and no evidence of vasculitis, consistent with CD. Introduction of infliximab dramatically relieved the patient's melena and abdominal symptoms. CONCLUSION: Diagnostic criteria for CD and SLE overlap, making them difficult to diagnose correctly. It is important to consider CD for patients who have SLE with gastrointestinal manifestations. The pathology of lupus enteritis should be clarified through the accumulation of cases of SLE combined with CD.BMC Gastroenterology 12/2012; 12(1):174. · 2.42 Impact Factor -
Article: Inflammation surrounding the vertebral spinous processes as spondylarthritis in Behçet's disease.
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ABSTRACT: Here, we present two Behçet's disease (BD) patients with an uncommon complication: inflammation surrounding the spinous processes revealed by positron emission tomography/computed tomography (PET/CT). Both patients were treated successfully with 20 mg of prednisolone daily. Whether BD should be considered a form of spondylarthritis remains an open question. However, inflammation around the spinous processes should be regarded as a possible uncommon finding of seronegative spondyloarthropathy (SpA) in BD patients.Modern Rheumatology 07/2012; · 1.58 Impact Factor -
Article: Similarities and differences in fluorodeoxyglucose positron emission tomography/computed tomography findings in spondyloarthropathy, polymyalgia rheumatica and rheumatoid arthritis.
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ABSTRACT: OBJECTIVES: We assessed fluorine-18 ((18)F)-labelled fluorodeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) findings in patients with seronegative spondyloarthritis (SpA), polymyalgia rheumatica (PMR), and rheumatoid arthritis (RA). METHODS: We studied 53 patients with SpA (n=21), PMR (n=16), or RA (n=16) admitted to our hospital between 2006 and 2011. Disease activity in the ischial tuberosities, greater trochanters, spinous processes, vertebral bodies, and sacroiliac joints (SIJ) were evaluated by determining FDG accumulation using maximum standardized uptake values (SUV(max)) and FDG scores. RESULTS: SUV(max) for ischial tuberosities was significantly higher in PMR than SpA or RA. SUV(max) for greater trochanters and spinous processes was significantly higher in PMR than RA (P<0.001) and significantly higher in SpA than in PMR or RA for SIJ (P=0.01). No significant difference in vertebral scores was observed among groups (P=0.488). FDG scores yielded similar results. X-ray findings were consistent with PET/CT findings in 3/15 (20%) patients with sacroiliitis, whereas magnetic resonance imaging findings were consistent with PET/CT findings in 4/7 (57.1%) patients. CONCLUSIONS: PET/CT detection of inflammation in the ischial tuberosities, greater trochanters, and spinous processes discriminated between PMR and RA, but not between SpA and PMR. PET/CT findings can distinguish SpA from RA and PMR and are useful for the early diagnosis of sacroiliitis.Joint, bone, spine: revue du rhumatisme 06/2012; · 2.25 Impact Factor -
Article: Thrombotic thrombocytopenic purpura with an autoantibody to ADAMTS13 complicating Sjögren's syndrome: two cases and a literature review.
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ABSTRACT: An association between thrombotic thrombocytopenic purpura (TTP) and Sjögren's syndrome (SS) is rare. This is the first report of two patients with TTP who had inhibitory autoantibodies to ADAMTS13 (a disintegrin-like and metalloprotease with thrombospondin type 1 repeats) complicating primary SS. A rapid diagnosis of TTP, which is a potentially lethal condition, made it possible to treat the two cases successfully. Only eight similar cases with TTP complicating SS have been reported in the literature. The possible presentation of primary SS without classic sicca symptoms, but with haematological abnormalities including TTP, should be recognised. Furthermore, it is important to measure ADAMTS13 activity and anti-ADAMTS13 antibodies, because TTP with SS seems to be a concurrent overlapping autoimmune disorder. We suggest that plasma exchanges in combination with corticosteroids should be administered as early as possible, since they appeared to be effective in treating TTP with SS, including in our cases.Modern Rheumatology 04/2012; · 1.58 Impact Factor -
Article: [Malignant lymphoma presenting as inflammation of unknown origin].
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ABSTRACT: We classified underlying diseases presenting as inflammation of unknown origin in division of rheumatic diseases. On the other hand, patients with malignant lymphoma (ML) varying in cause and symptoms are often admitted to our Rheumatology Division as cases of malignant disease presenting with fever of unknown origin. Clinical records over 5 years were reviewed for 246 cases of undiagnosed inflammatory diseases at hospitalization. The 246 patients included 76 patients with infection, 116 with rheumatic disease, 8 with solid cancer, 9 with drug-induced fever, 10 with other disorders, and 27 with suspected ML. Of theses, malignant lymphoma (ML) was diagnosed in 20 patients (5 Mature T-cell and NK-cell neoplasms, 3 Hodgkin lymphomas, and 12 Mature B cell neoplasms including 5 Intravascular large B-cell lymphoma (IVLBCL)). The reasons for hospitalization were underlying collagen disease in 6 patients, medical workup for fever of unknown origin in 8 patients, and symptoms suggestive of collagen disease in 6. Nearly all of these patients presented with fever and other B symptoms, frequently with concurrent hypoxemia and hemophagocytic syndrome in IVLBCL. For diagnostic purposes, biopsy sites were determined by 18 fluorodexyglucose-positron emission tomography and computed tomography (FDG-PET/CT) in 13 patients, and findings on random skin biopsies were diagnostic in 3 of 5 patients with IVLBCL. Laboratory data on patients with B-cell lymphoma or peripheral T-cell lymphoma revealed tendencies for thrombocytopenia, elevated serum LDH, and increased soluble interleukin-2 receptor (sIL-2R). High serum ferritin values were more frequent in IVLBCL than in other B-cell lymphomas. ML ranked first among causes of inflammation of unknown origin at the division of Rheumatic Diseases, and FDG-PET/CT proved to be useful for biopsy site selection. IVLBCL was not uncommon, and our results show the usefulness of clinical features and random skin biopsies for rapid diagnosis.Japanese Journal of Clinical Immunology 01/2012; 35(2):136-43. -
Article: Hypertrophic pachymeningitis and tracheobronchial stenosis in IgG4-related disease: case presentation and literature review.
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ABSTRACT: Immunoglobulin G4 (IgG4)-related disease is a distinctive mass-forming disorder with frequent systemic involvement, most commonly in the pancreas, salivary glands and lacrimal glands. A few cases of dural involvement and one case of central airway stenosis have also been described. We report here a rare case of IgG4-related disease with intracranial hypertrophic pachymeningitis and irregular tracheobronchial stenosis. We review four previously reported cases of IgG4-related pachymeningitis. We currently lack international standards for the diagnosis of extrapancreatic IgG4-related disease. Based on the findings of the present case and those reported previously, we discuss the distinctive features of IgG4-related pachymeningitis.Internal Medicine 01/2012; 51(8):935-41. · 0.94 Impact Factor -
Article: [Successful early treatment in a case of Cogan's syndrome].
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ABSTRACT: We report a 53-year-old male with Cogan's syndrome. He was admitted to our hospital because of a fever of 2-weeks duration, blurred vision for 10 days, hypoacusis, and numbness of the left hand for 3 days. In addition to uveitis, hypoacusis, and aseptic meningitis, multiple mononeuropathy was diagnosed based on a nerve conduction study. Furthermore, positron emission tomography/computed tomography (PET/CT) revealed diffuse aortitis. Accordingly, the patient was diagnosed with Cogan's syndrome. After starting steroid-pulse therapy followed by 1 mg oral prednisolone/kg/day, the uveitis and hypoacusis improved immediately, while the peripheral neuropathy persisted until effectively treated with intravenous gamma globulin therapy. Prompt steroid therapy for Cogan's syndrome based on a diagnosis made using PET/CT prevented progression of the hypoacusis.Japanese Journal of Clinical Immunology 01/2012; 35(1):92-6. -
Article: Whole-body fluorodeoxyglucose positron emission tomography/computed tomography in patients with active polymyalgia rheumatica: evidence for distinctive bursitis and large-vessel vasculitis.
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ABSTRACT: To investigate fluorodeoxyglucose (FDG) accumulation in large joints, bursas, and large vessels in patients with polymyalgia rheumatica (PMR) using 18-FDG positron emission tomography/computed tomography (PET/CT) and to differentiate PMR from similar diseases. Fourteen untreated patients with active PMR and 17 control patients with rheumatoid arthritis (n = 11) or other active rheumatic diseases (n = 6) underwent 18-FDG PET/CT. FDG uptake in large joints, bursas and vertebral spinous processes was evaluated by calculating maximum standardised uptake values and by visual scoring (scale 0-4). PET scan images were scored in seven vascular regions, and total vascular scores (range 0-21) were calculated. Polymyalgia rheumatica patients showed increased FDG uptake in ischial tuberosities, greater trochanters, and lumbar spinous processes. Positive results at two or more of these sites showed high sensitivity (85.7%) and specificity (88.2%) for the diagnosis of PMR, and shoulder or hip-joint involvement showed low disease specificity. High FDG accumulations were found in the aortas and subclavian arteries of two PMR patients who were asymptomatic for temporal arteritis and scanty synovium and perisynovium, based on FDG uptake. PET/CT images of the 12 PMR patients without apparent vascular involvement showed synovitis and/or perisynovitis. Fluorodeoxyglucose-PET/CT may be useful for the detection of PMR lesions, which are difficult to identify using other methods.Modern Rheumatology 12/2011; 22(5):705-11. · 1.58 Impact Factor -
Article: Prognosis of pneumocystis pneumonia complicated in patients with rheumatoid arthritis (RA) and non-RA rheumatic diseases.
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ABSTRACT: Clinical presentation of pneumocystis pneumonia (PCP) during immunosuppressive therapy for rheumatic diseases was compared between patients with rheumatoid arthritis (RA; n = 7) and those without RA (non-RA; n = 12) based on a chart review. Both RA and non-RA patients with PCP were treated with methotrexate (n = 7) combined with steroids (n = 6) and/or biologics (n = 4). RA-PCP patients were found to have a higher mortality rate than non-RA-PCP patients (3/7 vs. 0/12, respectively; p = 0.036) due to a later exacerbation of interstitial pneumonia and a higher presentation rate of diffuse pulmonary lesions (4/7 vs. 1/12, respectively; p = 0.036) despite lower mean levels of serum beta-D: -glucan (314 ± 214 vs. 1139 ± 1114 pg/ml, respectively; p = 0.02) that suggested a lower burden of Pneumocystis jirovecii. In conclusion, PCP in RA patients with existing pulmonary lesions may trigger subsequent progression to lethal interstitial pneumonia.Modern Rheumatology 10/2011; 22(4):509-14. · 1.58 Impact Factor -
Article: Diffuse alveolar damage in patients with dermatomyositis: a six-case series.
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ABSTRACT: The clinical course of diffuse alveolar damage (DAD) was studied in six consecutive cases of dermatomyositis (DM) based on our hospital records over 8 years. Three patients had severe myopathy at presentation, and the other three patients showed clinically amyopathic DM (CADM). Interstitial pneumonia in all patients developed shortly after they manifested DM. DAD in five deceased patients, which was proven pathologically, did not respond to steroid therapy combined with cyclosporine or tacrolimus. Of these, two patients began receiving combination therapy before suffering respiratory symptoms, and one of them had elevated serum Krebs von der Lungen-6 (KL-6) levels before visible abnormalities appeared on a plain chest X-ray. Only one patient with CADM survived; this patient received intravenously administered pulse cyclophosphamide (IVCY) therapy intravenously for DAD from the early stage. Delayed adjunctive IVCY was ineffective for progressed DAD in the remaining five patients. Elevated serum ferritin levels were observed in all four patients examined and might have predicted the lethal DAD, as in a previous report. In conclusion, promptly beginning IVCY therapy may be beneficial for patients with DM and interstitial pneumonia who show elevated serum levels of ferritin or KL-6 with minimal pulmonary abnormalities.Modern Rheumatology 09/2011; 22(2):243-8. · 1.58 Impact Factor -
Article: Benefit and a possible risk of tocilizumab therapy for adult-onset Still's disease accompanied by macrophage-activation syndrome.
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ABSTRACT: We report a 57-year-old female case of intractable adult-onset Still's disease (AOSD). Initial high-dose prednisolone therapy was ineffective, and macrophage-activation syndrome (MAS) manifested after one session of additional tocilizumab therapy. After successful treatment for MAS with lipo-dexamethasone and cyclosporin, tocilizumab therapy aided in the rapid reduction of the therapeutic steroid dose. Tocilizumab may be useful for maintenance therapy for AOSD, although its efficacy is unclear for the highly active phase of the disease.Modern Rheumatology 02/2011; 21(1):92-6. · 1.58 Impact Factor -
Article: [Successful bosentan therapy in a case of pulmonary arterial hypertention developed during immunosuppressive therapy for lupus nephritis].
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ABSTRACT: We report a 43-year-old female who developed pulmonary arterial hypertension (PAH) during intensive immunosuppressive therapy for systematic lupus erythematosus (SLE). She was diagnosed as SLE at the age of 32 years based on serological and hematological abnormalities, oral ulcers, and facial erythema. She experienced frequent flare-ups of disseminated discoid lupus between the ages of 33 and 36 years and developed immune thrombocytopenia at the age of 39 years. In 2007 when she was 43 years old, she developed lupus nephritis (LN) with elevated serum anti-double stranded DNA antibodies and urine protein of less than 1 g/day. Combination therapy for the LN with 35 mg/day prednisolone and intravenous cyclophosphamide (IVCY) led to renal remission. After the seventh monthly session of IVCY, she developed dyspnea on exertion. PAH was diagnosed based on enlarged main pulmonary arteries on the chest x-ray, right ventricular outflow and a peak tricuspid regurgitant pressure gradient exceeding 45 mmHg on echocardiography, an elevated plasma brain natriuretic peptide (BNP) level of 260 pg/ml, the exclusion of pulmonary thromboembolism, and no lung fibrosis. The PAH was treated successfully with bosentan. At present the tricuspid regurgitation has disappeared, and the plasma BNP level has normalized.Japanese Journal of Clinical Immunology 01/2011; 34(2):99-104.
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Institutions
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2011–2012
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National Center for Global Health and Medicine in Japan
Tokyo, Tokyo-to, Japan
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