An HLA-B27-positive patient diagnosed with ulcerative colitis 15 years after the onset of arthropathy

Abstract

A 28-year-old woman had persistent pain of both hip joints since the age of 13 years. X-ray analysis showed destructive changes in both hip joints and ossification of sacroiliitic joints. The patient had mild diarrhea and slight abdominal pain for 8 years. Blood-stained stool was not noticed. Barium enema showed changes consistent with the diagnosis of ulcerative colitis (UC). Inflammatory bowel syndrome should be considered in patients with persistent coxitis, even in the absence of severe abdominal symptoms.     

An HLA-B27-positive patient diagnosed with ulcerative colitis 15 years after the onset of arthropathy

 A 28-year-old woman had persistent pain of both hip joints since the age of 13 years. X-ray analysis showed destructive changes in both hip joints and ossification of sacroiliitic joints. The patient had mild diarrhea and slight abdominal pain for 8 years. Blood-stained stool was not noticed. Barium enema showed changes consistent with the diagnosis of ulcerative colitis (UC). Inflammatory bowel syndrome should be considered in patients with persistent coxitis, even in the absence of severe abdominal symptoms. Received: September 17, 2001 / Accepted: April 9, 2002 Correspondence to:A. Tsutsumi     

Successful treatment with etanercept in a patient with hepatotoxicity closely related to infliximab

To date, hepatotoxicity with anti-TNF therapy has been associated with concomitant liver-toxicity drugs, infection or malignant diseases. We report the case of one patient with spondyloarthropathty who presented severe liver dysfunction related to infliximab. After the second infusion serum controls showed an slightly increase of transaminases. Before the administration of fifth infusion, infliximab therapy was stopped due to severe liver damage (AST 327 mU/ml, ALT 656 mU/mL, GGT 140 mU/mL, alkaline phosphate 227 mU/mL). Ten weeks after infliximab discontinuation serum concentrations of liver blood tests were normal but ankylosing spondylitis symptoms had relapsed. Therefore, he was treated with etanercept with a rapid and sustained improvement. Serum concentrations of albumin, AST, ALT, GGT and alkaline phosphate were followed and did not change for five months.     

Azathioprine-induced severe pancytopenia due to a homozygous two-point mutation of the thiopurine methyltransferase gene in a patient with juvenile HLA-B27-associated spondylarthritis

Severe pancytopenia due to azathioprine (AZA) toxicity in patients with autoimmune diseases is not uncommon. We describe a 14-year-old girl with HLA-B27+ spondylarthritis who was treated with AZA 3 mg/kg/day and who suddenly developed severe pancytopenia in the seventh week of treatment. Analysis of the catabolic pathway of AZA revealed a homozygous deficiency of thiopurine methyltransferase (TPMT) on the basis of a combined 2-point mutation at nucleotide positions 460 and 719 in the gene for TPMT, causing a toxic level of the metabolic active 6-thioguanine nucleotides (6-TGN) (2,394 pmoles/8 × 10⁸ red blood cells). The patient was transfusion dependent and finally recovered 8 weeks after the development of the pancytopenia. At that time, 6-TGN had already returned to normal therapeutic levels. Family studies revealed another homozygous deficiency in the mother, while the other family members were heterozygous.     

Dissection of the ascending aorta in a patient with HLA-B27 associated ankylosing spondylitis

We describe a 38-year-old patient with ankylosing spondylitis complicated by a non-traumatic dissection of the ascending aorta without concomitant Marfan's syndrome.     

Toxic hepatitis induced by infliximab in a patient with rheumatoid arthritis with no relapse after switching to etanercept

We present a case of toxic hepatitis related to infliximab treatment in a 38-year-old woman with rheumatoid arthritis (RA). The patient had previously been treated with different disease-modifying drugs (DMARDs) alone or in combination but had never revealed signs of liver dysfunction. Due to high disease activity, treatment with infliximab (3 mg/kg i.v.) was initiated in combination with methotrexate (MTX) (25 mg/week) and folic acid (5 mg/week). The patient stopped MTX and folic acid on her own initiative after 3 weeks due to improvement of joint symptoms. After seven infusions, progressive elevations of the transaminases up to five times the upper normal limit were noted and treatment with infliximab was terminated. Serological tests for viral and autoimmune hepatitis and for ANA and anti-dsDNA were all negative. Specific infliximab antibodies could not be detected. Ultrasound of the liver was normal. Liver biopsy showed late signs of acute toxic hepatitis without MTX-related fibrosis. This is one the first cases that convincingly demonstrates that infliximab treatment may cause toxic hepatitis. Moreover, the case suggests a lack of hepatic cross-toxicity between infliximab and etanercept as the patient continued with etanercept without new episodes of liver dysfunction.     

Reiter's syndrome caused by Streptococcus viridans in a patient with HLA-B27 antigen

A 26-year-old male patient with mitral valve prolapse and HLA-B27 antigen received endodontic treatment for dental caries. Two weeks later fever, dysuria, diarrhea, sterile inflammatory arthritis of lower limbs, enthesitis, dactylitis, conjunctivitis, and uveitis consecutively developed. Blood culture performed at the time of active arthritis yielded Streptococcus viridans. He did not have any history of psoriasis, acute infectious diarrhea, chronic inflammatory bowel diseases, or sexually transmitted diseases. Laboratory studies also excluded the possibility of infections by human immunodeficiency virus, hepatitis B or C virus, chlamydia, and streptococci from the upper airway. This report indicates that Streptococcus viridans can be the triggering microorganisms of Reiter's syndrome in some circumstances.     

Decreased incidence of anterior uveitis in patients with ankylosing spondylitis treated with the anti-tumor necrosis factor agents infliximab and etanercept

OBJECTIVE: To estimate the incidence of anterior uveitis in patients with ankylosing spondylitis (AS) who underwent anti-tumor necrosis factor (anti-TNF) therapy, using data from recently performed trials. METHODS: Data from 4 placebo-controlled studies with anti-TNF agents in AS (2 with etanercept and 2 with infliximab) and 3 open-label studies were analyzed for the prestudy prevalence and the incidence of reported flares of anterior uveitis. RESULTS: A total of 717 patients who received treatment for anterior uveitis during the course of published clinical studies were identified by a systematic literature search using Medline. Followup information on the course of anterior uveitis was available for 397 patients. Of these, 297 were exposed to etanercept and 90 were exposed to infliximab for a total of 430 and 146.4 years, respectively. Among 190 patients who received placebo, the overall exposure was 70.5 years. The frequency of flares of anterior uveitis in the placebo group was 15.6 per 100 patient-years (95% confidence interval 7.8-27.9), while the patients treated with anti-TNF agents had a mean of only 6.8 anterior uveitis flares per 100 patient-years (P = 0.01). Flares of anterior uveitis occurred less frequently (although not significantly) in patients treated with infliximab than in patients treated with etanercept (3.4 per 100 patient-years and 7.9 per 100 patient-years, respectively). CONCLUSION: Treatment of AS patients with biologic agents directed against TNFalpha is associated with a significant decrease in the number of anterior uveitis flares. This reduction was slightly more marked among patients treated with infliximab, but the difference was not significant.     

Ankylosing spondylitis and HLA-B27: restriction fragment length polymorphism and sequencing of an HLA-B27 allele from a patient with ankylosing spondylitis.

Two groups of patients with ankylosing spondylitis (AS) from England and Poland were examined for restriction fragment length polymorphisms (RFLPs) associated with the disease. No preferential association was found between the 9.2 kb PvuII fragment in HLA-B27 positive patients with AS compared with HLA-B27 healthy subjects as had been previously reported. In the English group, however, a 14 kb PvuII fragment was more common in HLA-B27 positive subjects with AS than in normal controls. Also 4.6 and 3.7 kb PvuII fragments were more prevalent in subjects without AS than in the group with AS, but these results were confined to the English group. Furthermore, the sequence of an HLA-B*2705 gene isolated from a patient with AS was examined, and no significant differences were found compared with the sequence isolated from a healthy subject. There do not seem to be significant genetic differences in the coding or in the regulatory region in HLA-B27 alleles, in subjects with or without AS.     

Pneumonia with bacteremia due to Yersinia enterocolitica in a diabetic patient carrying HLA-B27

INTRODUCTION: We report a new case of pneumonia and bacteremia due to Yersinia enterocolitica (YE) in a diabetic patient with HLA-B27 positive spondylarthritis. OBSERVATION: A 75-year-old man was admitted for a pneumonia. He was suffering from HLA-B27 positive spondylarthritis and stable diabetes mellitus. Amoxicillin with clavulanic acid was ineffective. Two blood and stool cultures were positive for YE. There was no evidence of septic metastases, immunodepression and iron overload. Outcome was uneventful after 21 days of ofloxacin. CONCLUSION: YE pneumonia is rare. In this patient, diabetes mellitus and spondylarthritis with HLA-B27 may have played a role in the infection but their imputability remain questionable.     

Influence of antibodies against infliximab and etanercept on the treatment effectiveness of these agents in Japanese patients with rheumatoid arthritis

We investigated the influence of antibodies against infliximab and etanercept on the serum trough levels of these agents and the influence of these antibodies on the effectiveness of treatment in patients with rheumatoid arthritis treated with these agents. Forty patients treated with infliximab for 54?weeks and 40 patients treated with etanercept for 32?weeks were enrolled. They were divided into responder and non-responder groups. Serum trough levels of and antibodies against these agents were measured by enzyme-linked immunosorbent assay or radioimmunoassay. Of the 40 patients treated with infliximab, 14 (35%) had anti-infliximab antibodies. Serum trough levels were significantly lower in the non-responder group (14 patients) than in the responder group (26 patients) 6?weeks after initiation of infliximab (p?相似文献