Successful treatment of chronic lupus myocarditis with prednisolone and mizoribine

A 36-year-old female patient who was diagnosed with chronic myocarditis as an initial manifestation of systemic lupus erythematosus (SLE) was admitted to our hospital. At her third occurrence of heart failure, we performed an endomyocardial biopsy and proved chronic myocarditis with SLE. Subsequently, she was treated with prednisolone and the immunosuppressive agent mizoribine (MZR), and her cardiac function improved. We describe for the first time treatment with MZR for chronic cardiac involvement of SLE.     

Successful treatment using tacrolimus plus corticosteroid in a patient with RA associated with MDS

We report a case of rheumatoid arthritis (RA) complicated by myelodysplastic syndrome (MDS) successfully treated by tacrolimus. A 57-year-old woman had persistent pain and swelling in bilateral wrist and knee joints, in addition to severe anemia and leukopenia. She was diagnosed with MDS and RA based on the results of bone marrow aspiration and the criteria of RA. Combination therapy with tacrolimus (1.5 mg day⁻¹) and prednisolone (10 mg day⁻¹) improved her bicytopenia and polyarthralgia.     

他克莫司治疗狼疮性肾炎的研究进展

系统性红斑狼疮（systemic lupus erythematosus,SLE）是一种常见的、累及多脏器的自身免疫性疾病。肾脏是SLE最易受累的器官之一,通过肾脏病理检查儿乎所有的SLE患者均有肾损害,通常称之为狼疮性肾炎（lupusnephritis,LN）。狼疮性肾炎严重影响狼疮患者的生存率,约20％的LN患者于10年内发展为尿毒症。随着糖皮质激素及细胞毒药物的应用,尤其是各种新犁免疫抑制剂的出现,LN的预后已有较大改善。     

Successful tocilizumab and tacrolimus treatment in a patient with rheumatoid arthritis complicated by systemic lupus erythematosus

We report a 37-year-old female of intractable rheumatoid arthritis (RA) complicated by systemic lupus erythematosus (SLE), who was successfully treated with a combination of tocilizumab (TCZ) and tacrolimus. She was diagnosed with RA when she was 21 years old, and was administered oral prednisolone, injectable gold and salazosulfapyridine, but deformity of her hands gradually developed. She developed high fever and thrombocytopenia when she was 35 years old. Renal involvement, pericarditis, positive antinuclear antibody and high level of anti-double-stranded DNA antibody were found and the patient was diagnosed with SLE. Polyarthritis and immunological abnormalities developed despite aggressive immunosuppressive therapy including high-dose corticosteroids and intravenously administered cyclophosphamide. Tacrolimus (TAC) therapy gave only partial improvement of joint symptoms. After the initiation of combination therapy with TCZ, not only was a complete remission of RA obtained, but also the serum levels of SLE markers dramatically decreased. Our report suggests the possibility that this combination therapy is effective in treating SLE as well as RA.     

Mizoribine therapy in a patient with lupus nephritis: the association between mizoribine concentration and peritoneal dialysis

We describe a 20-year-old female with systemic lupus erythematosus (SLE) who developed renal failure during continuous ambulatory peritoneal dialysis (CAPD). The patient was treated with mizoribine (MZR). MZR peak concentration was 1.8 μg/ml and was sufficient for clinical efficacy as measured by serological data. Treatment with MZR was safe and useful even while undergoing CAPD. Achieving optimal MZR blood concentration was important for treatment of SLE, even though the patient was in end-stage renal failure.     

Obstructive uropathy as the only manifestation of flare in a patient with systemic lupus erythematosus and anti-phospholipid syndrome

Chronic interstitial cystitis and ureteral stenosis has occasionally been reported in systemic lupus erythematosus, mostly associated with gastrointestinal symptoms. We report a case of obstructive uropathy associated to chronic interstitial cystitis as the only manifestation of lupus flare in a patient with SLE and anti-phospholipid syndrome (APS) who had been in remission for many years. The development of chronic interstitial cystitis in patients with SLE and APS has not been previously reported. Histopathological study of her urinary bladder and ureteral meatus showed chronic inflammatory infiltrate in the subepithelium. Lack of significant lower urinary tract symptoms and gastrointestinal involvement were some of the factors that could have prevented an earlier diagnosis. Obstructive uropathy and renal insufficiency initially improved with immunosuppressive treatment and endoureteral protheses, but poor compliance to the therapy led to ominous ending.     

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??Abstract??Objective To evaluate the efficacy and safety of mizoribine (MZR) for active systemic lupus erythematosus (SLE) in comparison with mycophenolate mofetil (MMF) and cyclophosphamide (CYC).Methods Ninety patients with active SLE were enrolled between April 2012 and March 2013.Thirty patients were given MZR orally at a dose of 300 mg every other day.Thirty patients were taken MMF at 2 g per day in two divided doses.Thirty patients received 0.5 g CYC intravenously every two weeks.Therapeutic effects and adverse events (AEs) were evaluated at the end of 24-week treatment.Results The overall response rates of MZR??MMF and CYC group were 75.86%??78.58%??and 79.31% at week 24 respectively.In MZR??MMF and CYC group??34.48%??39.29%??and 37.93% patients achieved complete remission respectively.There was no significant difference in the overall response rates or complete remission rates among the three groups.SLEDAI scores??24-hour proteinuria and anti-dsDNA titer significantly decreased in all the three groups.All of the three treatments significantly increased leukocyte counts??hemoglobin levels??platelet counts??and serum complement C3 and C4 levels.No significant difference was observed in the parameters above among the three groups.The incidences of AEs in patients treated with MZR or MMF were significantly lower than those in patients treated with CYC.Conclusion MZR is well tolerated and has an effect similar to MMF or CYC in the treatment of active SLE??which may serve as a novel and alternative approach for SLE patients.     

Syndrome of inappropriate secretion of antidiuretic hormone induced by tacrolimus in a patient with systemic lupus erythematosus

We describe the first reported case of a syndrome of inappropriate secretion of antidiuretic hormone (SIADH) induced by low-dose tacrolimus in a patient with autoimmune disease. A 41-year-old man with systemic lupus erythematosus (SLE) developed hyponatremia induced by SIADH after administration of tacrolimus (0.06 mg/kg per day). In this case, the hyponatremia promptly resolved upon withdrawal of tacrolimus. This case strongly suggests that SIADH is a potentially important complication of tacrolimus administration, irrespective of dosage, and should be borne in mind whenever the drug is used.     

Spontaneous Achilles tendon rupture in a patient with systemic lupus erythematosus due to ischemic necrosis after methyl prednisolone pulse therapy

Spontaneous tendon rupture in a patient with systemic lupus erythematosus (SLE) is a rare but potentially disabling complication. Minor trauma, local inflammation and long term corticosteroid therapy are regarded as possible causes. However, ischemic necrosis of the tendon resulting from hypercoagulability and methyl prednisolone (MTP) pulse therapy has not been reported. We present a 20-year old female, newly diagnosed with lupus, who has high titer antiphospholipid antibodies, hyperhomocysteinemia and protein S deficiency. Her severe clinical symptoms of lupus were improved after MTP pulse therapy. Several days later, cold sensation over the right lower leg developed. On day 15 after pulse therapy, acute onset of right heel pain occurred when she was ascending stairs. Rupture of the right Achilles tendon was demonstrated by sonography and MRI. A Doppler sonography revealed narrowing and abrupt cessation of blood flow in the right popliteal artery. Heparin treatment was started. The angiography performed two days after heparinization revealed narrow caliber and decreased flow of the right tibial artery below the right ankle. Surgical repair of the tendon was successful and the pathology of the resected tendon revealed focal necrosis, degeneration and capillary proliferation. MTP pulse therapy in a lupus patient with hypercoaguable state with hyperhomocysteinemia, protein S deficiency and high titer antiphospholipid antibodies may cause spontaneous tendon rupture.     

Successful treatment of a patient with refractory adult Still's disease by tacrolimus

Adult Still's disease (ASD) is a systemic rheumatic disease characterized by high spiking fever, erythema, polyarthritis, and increased levels of C-reactive protein, ferritin, and interleukin (IL)-18. Recently, biological agents targeting proinflammatory cytokines such as tumor necrosis factor (TNF) α, IL-1, and IL-6 have been described as effective treatments for refractory ASD. Herein, we present a patient with ASD, who was successfully treated by tacrolimus concomitant with corticosteroid, while infliximab and etanercept were not effective. Tacrolimus may be one of the drugs for the ASD patients refractory to the conventional treatments including TNF inhibitors.     

Comparison of tacrolimus and mizoribine in a randomized, double-blind controlled study in patients with rheumatoid arthritis

OBJECTIVE: To compare the efficacy and safety of tacrolimus and mizoribine in patients with rheumatoid arthritis (RA). METHODS: Adult patients with RA with an insufficient response to at least one disease modifying antirheumatic drug (DMARD) were randomized to receive 28 weeks of double-blind treatment with tacrolimus 3 mg once daily or mizoribine 50 mg three times daily. The primary efficacy endpoint was the American College of Rheumatology 20% (ACR20) response. Safety was evaluated by adverse events. RESULTS: A total of 204 patients were enrolled for study (103 in the tacrolimus group, 101 in the mizoribine group). Significantly more patients receiving tacrolimus achieved an ACR20 response compared with mizoribine (48.5 vs 10.0%, respectively; p = 0.001). Tacrolimus was also superior to mizoribine in ACR50 and ACR70 response rate, tender and painful joint counts, swollen joint counts and patient and physician assessments of pain, disease activity, and patient's physical function assessment based on the Modified Health Assessment Questionnaire (p < 0.001). Adverse events were more frequent in the tacrolimus group than the mizoribine group (65.0 vs 59.4%); however, there were no statistically significant differences between treatment groups. CONCLUSION: Tacrolimus improves RA symptoms to a significantly greater extent than mizoribine in patients with RA inadequately controlled with at least one prior DMARD. Tacrolimus has the potential to be a useful and highly effective treatment for RA.     

Methyl prednisolone pulse therapy in the treatment of systemic lupus erythematosus.

Twenty patients with active systemic lupus erythematosus (SLE) were treated with methyl prednisolone pulse therapy (MPPT) and followed up for up to 24 weeks (mean 18 weeks). Beneficial effects of MPPT were observed principally on arthralgia, pleuritic pain, vasculitic skin rash, pyrexia, and lymphadenopathy. The serological tests showing the most improvement were ds DNA binding and the serum C3 level. MPPT was found to be both safe and easy to administer. It may be of value in treating patients with SLE whose disease is not controlled by moderate doses of corticosteroids and may also enable the dose of maintenance corticosteroids to be reduced appreciably.     

Drug-related lupus in a patient with rheumatoid arthritis under sulfasalazine treatment

Summary The induction of lupus-like syndromes with the appearance of single-stranded DNA antibodies is a well-known complication of drug therapy. In this report we present a patient with an erosive seropositive rheumatoid arthritis developing the clinical and serological features of systemic lupus erythematosus including the occurrence of double-stranded DNA antibodies under sulfasalazine treatment.     

Myelofibrosis successfully treated with prednisolone in a patient with pachydermoperiostosis

Pachydermoperiostosis (PDP) is a rare disorder of bone and connective tissue growth. A 21-year-old man was referred to our hospital with anemia. He showed characteristics of PDP. Bone marrow biopsy showed myelofibrosis. Chromosomal abnormalities or JAK2 mutation were not found. Anemia gradually progressed, and he became transfusion-dependent. Oral prednisolone was initiated; it gradually improved his anemia and rendered the patient free of transfusion. However, other clinical symptoms such as clubbed fingers and skin hypertrophy remained unimproved. In this case, the serum concentration of vascular endothelial growth factor and transforming growth factor-β levels were increased. Further investigation will be necessary to establish appropriate treatment strategies for this disease.     

Esophagitis in an adolescent patient with Crohn's disease after changing treatment from prednisolone to budesonide

Studies have demonstrated that budesonide is effective in the treatment of active Crohn's disease. Due to its extensive hepatic metabolism, budesonide has much lower adverse events compared to prednisolone. Consequently, the low systemic availability restricts its application to Crohn's disease of the terminal ileum and the colon. Esophageal ulceration is a rare complication of Crohn's disease. This article describes the case of a young lady who presented at the age of 16 with active Crohn's disease of the terminal ileum and the colon without dysphagia or pain in the chest. Her disease was successfully treated with prednisolone for almost two years. Because of weight gain, acne, and moon face she was switched to budesonide. A few days later she presented with intractable pain of the esophagus, dysphagia, and inability to eat. Endoscopy demonstrated aphthous ulcerations of the esophagus and the histology was compatible with Crohn's disease. After two weeks of treatment with prednisolone all symptoms resolved and at follow-up gastroscopy ulcers had disappeared.