MedPath

IVIG With Rituximab vs Rituximab as First Line Treatment of Pemphigus

Phase 2
Active, not recruiting
Conditions
Pemphigus
Interventions
Other: IVIg
Registration Number
NCT04400994
Lead Sponsor
The University of Hong Kong
Brief Summary

Pemphigus is a rare acquired autoimmune disease in which immunoglobulin G (IgG) antibodies target desmosomal proteins to produce intraepithelial, and mucocutaneous blisters. It is potentially fatal and the average mortality of pemphigus vulgaris (PV) was 75% before the introduction of corticosteroids in the early 1950s.

Traditionally, treatment of pemphigus included high dose systemic corticosteroids with or without adjuvant immunosuppressants. However; the prolonged use of high dose steroids carries significant side effects. A recent randomized trial has proved the efficacy of Rituximab, a monoclonal anti-CD20 antibody against B-lymphocytes, as an efficacious therapy for pemphigus. Early use of rituximab was associated with better clinical outcomes, hence combination treatment of rituximab and intravenous immunoglobulins (IVIG) has shown to be effective for refractory pemphigus cases and can potentially induce long-term complete remission and lower risks infectious complications.

In this study, investigators will evaluate the efficacy and safety of early use of rituximab with or without IVIG in patients with moderate to severe pemphigus using protocols that were similar to those previously published, investigators will also aim to measure the impact of health care economics and in doing so, assess the cost and benefits of both treatment arms.

Detailed Description

Pemphigus is a rare acquired autoimmune disease in which immunoglobulin G (IgG) antibodies target desmosomal proteins to produce intraepithelial, and mucocutaneous blisters. It is potentially fatal and the average mortality of pemphigus vulgaris (PV) was 75% before the introduction of corticosteroids in the early 1950s.

Traditionally, treatment of pemphigus included high dose systemic corticosteroids with or without adjuvant immunosuppressants. However; the prolonged use of high dose steroids carries significant side effects. A recent randomized trial has proved the efficacy of Rituximab, a monoclonal anti-CD20 antibody against B-lymphocytes, as an efficacious therapy for pemphigus. Furthermore, early use of rituximab was associated with associated with better clinical outcomes. Moreover, combination treatment of rituximab and intravenous immunoglobulins (IVIG) has shown to be effective for refractory pemphigus cases and can potentially induce long-term complete remission and lower risks infectious complications.

Cost effectiveness is an important issue and while combination of IVIG and rituximab has been advocated, the cost of such treatment is substantial and whether it poses any benefit over rituximab alone, or with other more conventional immunosuppressive agents, has not been established. Both treatment approaches have been previously published in high impact journals.

In this study, investigators aim to evaluate the efficacy and safety of early use of rituximab with or without IVIG in patients with moderate to severe pemphigus using protocols that were similar to those previously published. Apart from complete remission and adverse effects, investigators will also aim to measure the impact of health care economics and in doing so, assess the cost and benefits of both treatment arms.

Recruitment & Eligibility

Status
ACTIVE_NOT_RECRUITING
Sex
All
Target Recruitment
20
Inclusion Criteria
  • Written informed consent obtained from patient
  • Ages Eligible for Study: 18 years to 75 years (Adult, Older Adult)
  • Newly or recently diagnosed (less than 18 months) diagnosed pemphigus vulgaris or pemphigus foliaceus based on clinical features; histological features of acantholysis via skin or mucosal biopsy; and intercellular staining pattern of indirect immunofluorescence or serological detection of DSG 1 or DSG 3 by enzyme-linked immunosorbent assay (ELISA)
  • Moderate to severe active disease, as defined by overall PDAI >= 15 or skin involvement BSA>= 5%. 9 [Annex 1]
  • Receiving standard-of-care oral prednisolone up to 1.5 mg/kg/day
  • Women who are sexually active and not postmenopausal, agreement to remain abstinent or use 2 effective methods of contraception.
  • Ability to comply with study protocol as deemed by investigator's assessment

Exclusion criteria:

  • Age <18 or >75
  • Pregnant women or nursing mother
  • Already diagnosed pemphigus patients diagnosed > 18 months
  • Non-consenting patients, or patient who cannot be followed up regularly
  • Patient with history of serious allergy or anaphylactic reaction to monoclonal antibody treatment
  • Severe heart failure (NYHA Class III or IV)
  • Unstable angina or myocardiac infarction within last 3 months or post-infarction heart failure
  • Anaemia (haemoglobin <10g/dL), Neutropenia (<1000/mm3), Lymphopenia (<900/mm3), thrombocytopenia (<100,000/mm3)
  • Renal insufficiency eGFR <60
  • Liver insufficiency of ALT/ALT > 2 times normal limit range
  • Positive test results for hepatitis C (HCV) serology at screening *Patients who are HepBs Ag positive, or HepBs Ag negative and anti-HepBc Ab - positive: Patients who are HepBs Ag positive - will be started on entecavir 0.5mg daily, and will be referred to a gastroenterologist for further follow up.

Patients who are HepBs Ag negative, and HBc Ab positive, with detectable HepB DNA levels - will be started on entecavir 0.5mg daily, and will be referred to a gastroenterologist for further follow up.

Patients who are HepBs Ag negative, HBc Ab positive, with no detectable HepB DNA levels - will be started on entecavir 0.5mg daily, and will be continued on entecavir for at least 18 months after completion of last dose of rituximab.

  • Blood test positive for HIV
  • Signs of active infection on CXR
  • Positive interferon gamma release assay Quantiferon or T.Spot TB test: must be treated with at least 4 weeks post initiation of isoniazid or other TB therapy
  • Inherited or acquired severe immunodeficiency
  • History of malignancy
  • Patient with active severe infection (excluding fungal infections of the nail), which has required antibiotic treatment within 2 week prior to study enrolment
  • Infection requiring hospitalisation or intravenous antibiotic treatment within the last 8 weeks prior to enrolment
  • Past history of osteomyelitis, or fasciitis, septic arthritis within the last one year
  • Patients with drug induced pemphigus. A thorough medication history will be taken to rule out drug induced pemphigus including D-penicillamine, angiotensin-converting enzyme inhibitors, angiotensin receptor blockers and cephalosporins
  • Evidence of any new or uncontrolled concomitant disease that in the investigators' judgement would preclude the patients participation
  • Patients with history of allergy or adverse events to IVIG or rituximab treatment10
  • Treatment with intravenous immunoglobulins, plasmaphoresis within the last 8 weeks prior to randomization
  • Previous treatment with rituximab or any monoclonal antibody inducing profound lymphopenia
  • Treatment with live or attenuated vaccine within the last 28 days prior to randomization
Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Rituximab and IVIGIVIg* Rituximab (375 mg/m2 BSA) once a week for 4 weeks (week 1, 2, 3); * Week 4: Rituximab + IVIG 2g per kg * Week 5, 6, 7: Above treatment repeated for 2nd cycle, infusion of rituximab (375 mg/m2 BSA) once a week for 4 weeks (week 5, 6, 7); * Week 8: Rituximab + IVIG 2g/kg * In months 3, 4, 5, 6, patients received a single infusion of rituximab (375 mg/m2 BSA) plus infusion of 2g/kg IVIG * Thus in 6-month period patients received a total of 12 infusions of rituximab and 7 infusions of IVIG * If a patient was clinically free of disease at end of 6 months, additional infusions of IVIG will be given at week 30, 38, 48, 60 and 76 * A total of 12 doses of rituximab and 12 cycles of IVIG will be given
Rituximab onlyRituximab* Rituximab infusion 375mg/m2 body surface area (BSA) weekly for 4 weeks from baseline (week 0, 1, 2, 3) * Rituximab infusion 375mg/m2 BSA weekly for 4 weeks at week 24 (week 24, 25, 26, 27) * Rituximab infusion 375mg/m2 BSA weekly for 2 weeks at week 52 (week 52, 53) * Rituximab infusion 375mg/m2 BSA weekly for 4 weeks at week 76 (week 76, 77) * A total of 12 doses of rituximab will be given in 55 weeks
Rituximab and IVIGRituximab* Rituximab (375 mg/m2 BSA) once a week for 4 weeks (week 1, 2, 3); * Week 4: Rituximab + IVIG 2g per kg * Week 5, 6, 7: Above treatment repeated for 2nd cycle, infusion of rituximab (375 mg/m2 BSA) once a week for 4 weeks (week 5, 6, 7); * Week 8: Rituximab + IVIG 2g/kg * In months 3, 4, 5, 6, patients received a single infusion of rituximab (375 mg/m2 BSA) plus infusion of 2g/kg IVIG * Thus in 6-month period patients received a total of 12 infusions of rituximab and 7 infusions of IVIG * If a patient was clinically free of disease at end of 6 months, additional infusions of IVIG will be given at week 30, 38, 48, 60 and 76 * A total of 12 doses of rituximab and 12 cycles of IVIG will be given
Primary Outcome Measures
NameTimeMethod
relapse-free complete remissionFrom baseline up to 208 weeks

Percentage of participants who achieve relapse-free complete remission

Secondary Outcome Measures
NameTimeMethod
Time to protocol defined disease flareFrom baseline up to 208 weeks

Time to protocol defined disease flare

Number of protocol defined disease flaresFrom baseline up to 208 weeks

Number of protocol defined disease flares

Time to initial complete remissionFrom baseline up to 208 weeks

Time to initial complete remission, evaluated by the PDAI activity score

Duration of complete remissionFrom baseline up to 208 weeks

Duration of complete remission, evaluated by the PDAI activity score

Blood lymphocyte level (CBC)Baseline week 0, week 4, 12, 24, 36, 48, 60, 72, 84, 96, 120, 144, 168, 192

Blood lymphocyte level (CBC)

Blood DSG 1 and 3 levelsBaseline week 0, week 4, 12, 24, 36, 48, 60, 72, 84, 96, 120, 144, 168, 192

Blood DSG 1 and 3 levels

Blood CD19/20 mean B cell counts percentageWeek 0, week 4, 12, 24, 36, 48, 60, 72, 84, 96, 120, 144, 168, 192

Blood CD19/20 mean B cell counts percentage

Occurrence of severe treatment adverse eventsBaseline, week 4, 12, 24, 36, 48, 60, 72, 96, 120, 144, 168, 192

Safety endpoints: Occurrence of treatment adverse events, serious adverse events (grade 3 or 4) based on common terminology criteria for adverse events (CTCAE). Death from any cause. Adverse events leading to discontinuation, vital signs, and laboratory tests

Change in health-related quality of life: Dermatology Life Quality Index (DLQI) ScoreBaseline, Week 4, 12, 24, 36, 48, 60, 72, 84, 96, 120, 144, 168, 192

Change in health-related quality of life as measured by the Dermatology Life Quality Index (DLQI) Score.

The DLQI is calculated by summing the score of each question resulting in maximum of 30 and a minimum of 0. The higher the score, the more quality of life is impaired.

Number of rescue therapy givenBaseline up to Week 208

Number of rescue therapy given

Trial Locations

Locations (1)

Department of Medicine

🇭🇰

Central, Hong Kong

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