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Phase II Study on Gusperimus in Patients With Refractory Wegener's Granulomatosis

Phase 2
Completed
Conditions
Wegener's Granulomatosis
Interventions
Registration Number
NCT00530075
Lead Sponsor
Nippon Kayaku Co., Ltd.
Brief Summary

Wegener's granulomatosis is a primary systemic vasculitis characterized by granulomatous and necrotizing inflammation predominantly affecting the respiratory tract and the kidneys. Conventional therapy of Wegener's granulomatosis with cyclophosphamide and corticosteroids is limited by incomplete remissions and a high relapse rate. Patients accumulate irreversible damage due to the disease and the consequences of prolonged drug exposure. The efficacy and safety of an alternative immunosuppressive drug, gusperimus, was evaluated in patients with refractory disease. A prospective, international, nulti-centre, single limb, open label study. Entry required active Wegener's granulomatosis with a Birmingham Vasculitis Activity Score (BVAS) \>=4 and previous therapy with cyclophosphamide or methotrexate. Immunosuppressive drugs were withdrawn at entry and prednisolone doses adjusted according to clinical status. Gusperimus, 0.5mg/kg/day, was self-administered by subcutaneous injection in six treatment cycles of 21 days with a seven day washout between cycles. Cycles were stopped early for white blood count \< 4,000/mm3. The primary endpoint was complete remission (BVAS=0 for at least 2 months) or partial remission (BVAS\<50% of entry score). After the sixth cycle azathioprine was commenced and follow-up continued for a further six months.

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
45
Inclusion Criteria
  • Documented diagnosis of WG according to American College of Rheumatology (ACR) and Chapel Hill Consensus Conference (CHCC) definition
  • BVAS >= 4
  • Total disease duration >= 3 months treated with CYC or >= 6 months with MTX
  • Age 18 - 80
  • WBC >= 4,000/mm3, haemoglobin >= 8g/dl, neutrophils >= 2,500/mm3, platelets >= 100,000/mm3
  • ALT, bilirubin and alkaline phosphatase levels within 2x the upper limits of normal
  • Documented to be non-pregnant by serum/urine pregnancy test
  • Willing to participate in this study
  • Provide signed informed consent
  • Able and prepared to self-administer the study drug or have a close friend/relative able to do this
Exclusion Criteria
  • Participation in another clinical research study
  • Pregnant or nursing mothers and women of childbearing age not using appropriate contraception
  • Clear evidence of active disease due to bacteria/viral infection
  • Patient has an unacceptable risk for participation in a study of immunosuppressive therapy
  • History of substance abuse or psychotic disorders
  • Previous treatment with Gusperimus

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
1GusperimusGusperimus
Primary Outcome Measures
NameTimeMethod
Remission of VasculitisAt Entry (Day 1 of Cycle 1), Day 22 of cycles 1-6, up to 24 weeks

The primary efficacy outcome measure was remission of vasculitis. Complete remission was defined as a Birmingham vasculitis activity score (BVAS) of 0 sustained for at least 2 months. Partial remission was defined as a reduction in BVAS of 50% or more, sustained for at least 2 months, when compared with the BVAS at entry.

Entry required active Wegener's granulomatosis with a BVAS \>= 4. Their disease had to be active, as measured with BVAS in which clinical manifestations caused by active vasculitis are scored on a list of predefined organ-specific items.

Secondary Outcome Measures
NameTimeMethod
HaematuriaAt Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks

Assessment of anti-inflammatory activity of gusperimus using surrogate marker: number of hematuria-positive patients.

Duration of Clinical ResponseAt Entry (Day 1 of Cycle 1), Day 22 of cycles 1-6, up to 24 weeks, End of treatment period, and 3 and 6 months of follow-up period

Time from Complete Remission or Partial Remission to Relapse.

ANCAAt Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks

Assessment of anti-neutrophil cytoplasmic antibody (ANCA): Number of ANCA-positive patients was counted.

ANCA are highly associatred with active WG, with c-ANCA titres observed in 90% of WG. In addition to their diagnostic value, it has been suggested that ANCA may have a predictive value for relapse in patients with systemic vasculitis.

CRPAt Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks

Assessment of anti-inflammatory activity of gusperimus using surrogate marker: serum C-reactive protein level.

Vasculitis Damage Index (VDI)At Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks, 6 months of follow-up period

Assessment of the degree of irreversible damage due to the vasculitis using VDI scoring system. The VDI comprises 64 items of damage (grouped into 11 organ-based systems). Total VDI score is 0 - 64. The higher scores represent the more severe damage occurred in patients. The VDI score can either increase or remain the same over time.

CreatinineAt Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks

Assessment of anti-inflammatory activity of gusperimus using surrogate marker: serum creatinine level

SF-36At Entry (Day 1 of Cycle 1), End of treatment period, up to 24 weeks

Assessment of the impact of gusperimus on general health using the Short form-36 (SF-36) questionaire. The SF-36 is a self-report, 36 item survey measuring health-related quality-of-life. Thirty-five items are used to construct 8 scales: (1) physical functioning, (2) role physical, (3) bodily pain, (4) general health, (5) vitality, (6) social function, (7) role emotional, and (8) mental health. Raw scores are calculated as the sum of re-coded scale items and transformed to a 0 to 100 scale. If scores for all 8 scales are available, two summary measures known as component scores are derived: the Physical Health Component Score (PCS) and the Mental Health Component Score (MCS). First each scale standardized to the relevant population. Then PCS and MCS are calculated as the weighted sum of standardized scores. All scales and the component scores are positively scored so that higher scores represent better health-related quality-of-life.

Trial Locations

Locations (7)

Karolinska University Hospital

🇸🇪

Stockholm, Sweden

University Hospital Maastricht

🇳🇱

Maastricht, Netherlands

Addenbrookes Hospital

🇬🇧

Cambridge, United Kingdom

Universitatsklinikum Schleswig-Holstein

🇩🇪

Luebeck, Germany

General Faculty Hospital

🇨🇿

Prague, Czech Republic

Western General Hospital

🇬🇧

Edinburgh, Scotland, United Kingdom

Reumatologisk Klinik

🇩🇰

Copenhagen, Denmark

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