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A long-term study with the IL-1 receptor blocker anakinra/kineret® in patients with neonatal onset multisystem inflammatory disease (NOMID/CINCA syndrome)

Conditions
neonatal onset multisystem inflammatory disease (NOMID/CINCA)
Therapeutic area: Diseases [C] - Immune System Diseases [C20]
Registration Number
EUCTR2013-000300-42-Outside-EU/EEA
Lead Sponsor
ational Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS)
Brief Summary

Not available

Detailed Description

Not available

Recruitment & Eligibility

Status
A
Sex
All
Target Recruitment
50
Inclusion Criteria

No age limitation (valid from July 2007)
2. Patients fulfilled at least 2 of the following 3 clinical manifestations:
•Typical NOMID rash
•CNS involvement (papilledema, cerebrospinal fluid (CSF) pleiocytosis, sensorineural hearing loss)
•Typical arthropathic changes on radiographs (epiphyseal and/or patellar overgrowth)
3. Onset of manifestations of NOMID/CINCA at =6 months of age
4. Stable dose of steroids, NSAIDs, DMARDs for 4 weeks prior to enrolment visit
5. Washout period for biologics: 6 half-lives before Kineret administration for all drugs with anti-TNF properties. For etanercept (6 half-lives=24 days), this calculated to drug discontinuation 3 days before enrolment into the observation period, infliximab and adalimumab (6 half-lives=48 days) drug were to be discontinued 27 days before the observation period, and thalidomide (6 half lives=3 days) were to be discontinued 3 days prior to Kineret administration
6. Patient’s or legal guardian’s ability and willingness to give informed consent
7. Females of childbearing potential (young women who have had at least one menstrual period regardless of age) had to have a negative urine pregnancy test at baseline prior to performance of any radiologic procedure or administration of study medication. Women of childbearing age and men able to father a child, who were sexually active, were asked to use a form of effective birth control, including abstinence
8. Negative purified protein derivative (PPD) test using 5 T.U. intradermal testing per CDC guidelines with exception of inclusion criteria no. 9 below
9. Patients with latent tuberculosis (TB) (positive PPD test) had to have adequate therapy for TB initiated prior to first dose of study medication as recommended in published guidelines.

Are the trial subjects under 18? yes
Number of subjects for this age range: 36
F.1.2 Adults (18-64 years) yes
F.1.2.1 Number of subjects for this age range 7
F.1.3 Elderly (>=65 years) no
F.1.3.1 Number of subjects for this age range

Exclusion Criteria

Having received live virus vaccine during 3 months prior to baseline visit (first visit to NIH)
2. Active infections or a history of pulmonary TB infection with or without documented adequate therapy, patients with current active TB, or recent close exposure to an individual with active TB
3. Positive testing for HIV, Hepatitis B or C known or documented at screening, enrolment or baseline visit
4. A history of, or concomitant diagnosis of, congestive heart failure.
5. A history of malignancy
6. Prior use of anti-CD4 antibody
7. Known hypersensitivity to E. coli derived products or any components of Kineret
8. Presence of any other rheumatic disease or major chronic infectious/ inflammatory/ immunologic disease (e.g. inflammatory bowel disease, psoriatic arthritis, spondyloarthropathy, SLE in addition to NOMID/CINCA)
9. Presence of the following at enrolment visit: ALT or AST >2.0 x upper limit of normal (ULN) of the local laboratories values, creatinine >1.5 x ULN, WBC <3.6 x 109/L; platelet count <150,000 mm3
10. Enrolment in any other investigational clinical study or receiving an investigational agent, or had not yet completed at least 4 weeks since ending another investigational device or drug trial
11. Existing concern about compliance with the protocol procedures by patient and/or parent/s and legally acceptable representative/s
12. Lactating females or pregnant females
13. Patients with asthma were only included after evaluation by a pulmonary and infectious disease consultation
14. The use of other anti-IL-1 inhibiting agents or the initiation of a longer acting IL-1 inhibiting agent while on study led to non-enrolment or termination respectively.

Study & Design

Study Type
Interventional clinical trial of medicinal product
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
Secondary Outcome Measures
NameTimeMethod
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