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Clinical Trial to Evaluate Efficacy and Safety of Dupilumab in Localized Scleroderma

Phase 2
Completed
Conditions
Localized Scleroderma
Interventions
Registration Number
NCT04200755
Lead Sponsor
University of Cologne
Brief Summary

The DupiMorph study evaluates the efficacy of Dupilumab in localized scleroderma patients. Dupilumab is approved in the US and EU for the treatment of moderate/severe atopic dermatitis and since 2018 in the US for severe asthma therapy.

Detailed Description

Localized scleroderma (LS) comprises a heterogenous group of sclerotic skin disorders. The incidence of LS is reported to be approximately 27 cases/ 1x106 and is hence approximately 2-3-fold higher compared to systemic scleroderma. Although in most cases not lethal the disease can significantly impact quality of life. Depending on the location of fibrosis, the disorder can cause bone deformities, alopecia, skin atrophy or lesions with severe hypo-/hyperpigmentation.

The disease is pathomechanistically poorly understood and no effective therapy is currently approved. The most promising treatments up to date include methotrexate ± pulsed corticosteroids or phototherapy with PUVA or UVA1. Yet, the number of treated patients in these studies is low. The response rates are low and inconsistent with approximately 50% of patients treated with UVA1 experience a recurrence within three years. There are no studies on efficacy of topical corticosteroids in LS. Small pilot studies and few case reports describe regression of lesions after topical calcineurin inhibitors. In addition, current therapies can only be applied for a short time during the acute phase due to the side effect profile after long-term use (e.g. skin atrophy in response to topical steroids, skin cancer in response to long term UV therapy, multiple side effects by long- term use of methotrexate and/or corticosteroids). Hence, this study evaluates, in comparison with placebo, the efficacy of Dupilumab administered subcutaneously every 14 days in patients with Morphea (plaque type) or generalized localized scleroderma (affecting at least three anatomic sites).

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
45
Inclusion Criteria
  • Subject is a male or female ≥18 years of age on the day the study informed consent is signed
  • Out-patient status
  • Caucasian
  • Morphea (plaque type) or Generalized localized scleroderma (affecting at least three anatomic sites)
  • At least one lesions with lilac ring (active phase of the disease);
  • Activity of LS within the last 12 month (as defined by progression of size or new developing plaque)
  • For women of childbearing potential: negative pregnancy test at Visit 1
  • For women of childbearing potential: Use of effective method of contraception from 4 weeks prior to enrolment, throughout study treatment until 12 weeks after the last IMP dose.
  • Written informed consent signed
Exclusion Criteria
  • Systemic immunosuppressive therapy or UV therapy less than 3 months before enrollment.
  • Participation in another trial of IMPs or devices parallel to, or less than 6 months before or previous participation in this trial
  • Pregnancy or breastfeeding mother
  • Diagnosis of other significant chronic inflammatory or autoimmune disorders. Patients with the following autoimmune disorders are excluded from the study: Multiple sclerosis, primary biliary cirrhosis, type I diabetes mellitus. Patients with the following autoimmune disorders are regarded as eligible: Lichen sclerosus, vitiligo, alopecia arthritis, thyroid diseases (e.g. Hashimoto disease). Patients with any autoimmune disorder not listed above should only be included after consultation with the principal coordinating investigator.
  • Topical immunosuppressive therapy less than 1 month before enrollment
  • Concurrent phototherapy
  • Known infection with helminths (helminthosis)
  • Any condition or laboratory abnormality that, in the judgment of the investigator, would put the subject at unacceptable risk for participation in the study or may interfere with the assessments included in the study. E.g. uncontrolled psychiatric illness or history of clinical relevant drug abuse.
  • Known hypersensitivity to any components of the IMP
  • Treatment with a live (attenuated) vaccine within 3 months prior to enrollment
  • History of malignancy (except patients with completely treated in situ carcinoma of the cervix, completely treated and resolved non-metastatic squamous or basal cell carcinoma of the skin)
  • Known diagnosis of active tuberculosis or non-tuberculous mycobacterial infection or latent untreated tuberculosis unless it is well documented by a specialist that the patient has been adequately treated
  • Known diagnosis of HIV, HBV or HCV infection
  • Regular use (more than 2 visits per week) of a tanning booth/parlor
  • Known diagnosis of asthma

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
DupilumabDupilumab 300Mg Solution for Injection30 patients; Dupilumab s.c. injection; 2 ready-to-use syringes (600 mg) initial (V1), 1 ready-to-use syringe (300 mg) every 14 days (V2- V13) Dupilumab s.c. injection in healthy skin, 24 weeks
PlaceboPlacebo15 patients; placebo s.c. injection; 2 ready-to-use syringes initial (V1), 1 ready-to-use syringe every 14 days (V2-V13) placebo s.c. injection in healthy skin, 24 weeks
Primary Outcome Measures
NameTimeMethod
LoSCAT target lesionBaseline to End of Treatment Visit, 24 weeks

Treatment response is assessed using the LoSCAT (Localized Scleroderma Cutaneous Assessment Tool). Target lesion will be assessed at Baseline and End of Treatment. Score reduction by 50% after 24 weeks (End of Treatment Visit V14) compared to Baseline Visit (V1) is defined as treatment response.

Secondary Outcome Measures
NameTimeMethod
LoSDI all lesionsBaseline to Follow-Up Visit, 48 weeks

Change in LoSDI (Localized Scleroderma Skin Damage Index) of all existing lesions during treatment, at End of Treatment Visit and during follow-up

Body temperatureBaseline to Follow-Up Visit, 48 weeks

Body temperature will be documented throughout the study.

Anti-nuclear antibodies (ANAs) levelsBaseline to Follow-Up Visit, 48 weeks

Change in anti-nuclear antibodies (ANAs) levels

mLoSSI all lesionsBaseline to Follow-Up Visit, 48 weeks

Change in mLoSSI (Localized Scleroderma Skin Activity Index) of all existing lesions during treatment, at End of Treatment Visit and during follow-up

RT-qPCRBaseline to End of Treatment Visit, 24 weeks

Quantification of change in gene expression of genes identified by RNAseq in tissue of localized scleroderma patients after Dupilumab treatment: RT-qPCR of tissue biopsies obtained from the lesion (lilac ring, optional: center) before (baseline visit V1) and after treatment (End of Treatment Visit V14) and healthy skin before treatment

Adverse events (AEs)Baseline to Follow-Up Visit, 48 weeks

Adverse events will be documented throughout the study

Body weightBaseline to Follow-Up Visit, 48 weeks

Body weight will be documented throughout the study.

DLQIBaseline to Follow-Up Visit, 48 weeks

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

Blood pressureBaseline to Follow-Up Visit, 48 weeks

Blood pressure will be documented throughout the study.

Pulse rateBaseline to Follow-Up Visit, 48 weeks

Pulse rate will be documented throughout the study.

Haematocrit (HcT)Baseline to Follow-Up Visit, 48 weeks

Haematocrit (HcT) will be documented at End of Treatment Visit and during follow-up

HaemoglobinBaseline to Follow-Up Visit, 48 weeks

Haemoglobin (Hgb) will be documented at End of Treatment Visit and during follow-up

Blood cell countBaseline to Follow-Up Visit, 48 weeks

Platelet count and differential White blood cell count will be documented at End of Treatment Visit and during follow-up

Clinical ChemistryBaseline to Follow-Up Visit, 48 weeks

Clinical Chemistry parameters (Creatinine) will be documented at End of Treatment Visit and during follow-up

Number of lesionsBaseline to Follow-Up Visit, 48 weeks

Count of all existing non-target and new lesions on the entire integument during treatment, at End of Treatment Visit and during follow-up

RNAseqBaseline to End of Treatment Visit, 24 weeks

Gene signature of localized scleroderma after Dupilumab treatment: RNA- seq of tissue biopsies obtained from the lesion (lilac ring, optional: center) before (baseline visit V1) and after treatment (EoT V14) and healthy skin before treatment

Physical examinationBaseline to Follow-Up Visit, 48 weeks

Physical examination (general appearance including skin, head and throat (head, eyes, ears, nose, and throat), lymph nodes, respiratory, cardiovascular, musculoskeletal, and neurological systems) will be documented throughout the study.

Serum cytokine levelsBaseline to Follow-Up Visit, 48 weeks

Change in serum levels of IL-4, IL-5, IL-13, periostin, dipeptidyl peptidase-4

Blood EnzymesBaseline to Follow-Up Visit, 48 weeks

Clinical Chemistry parameters (Aspartate aminotransferase (AST), Alanine aminotransferase (ALT), Gamma-glutamyl transferase (GGT), Lactic dehydrogenase (LDH)) will be documented at End of Treatment Visit and during follow-up

Trial Locations

Locations (4)

Charité - Universitätsmedizin Berlin Klinik für Dermatologie, Venerologie und Allergologie

🇩🇪

Berlin, Germany

Uniklinik Köln, Klinik für Dermatologie und Venerologie

🇩🇪

Köln, Germany

Helios St. Elisabeth Klinik Oberhausen, Klinik für Dermatologie, Venerologie und Allergologie

🇩🇪

Oberhausen, Germany

Universitäts-Hautklinik Tübingen

🇩🇪

Tübingen, Germany

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