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Proposal for a prospective observational study of patients with atypical SCID

Conditions
D81.9
Combined immunodeficiency, unspecified
Registration Number
DRKS00000292
Lead Sponsor
Centrum für Chronische Immmundefizienz (CCI)
Brief Summary

Not available

Detailed Description

Not available

Recruitment & Eligibility

Status
Complete
Sex
All
Target Recruitment
73
Inclusion Criteria

1. All Patients with biallelic mutations in a gene where complete loss of function always leads to a SCID phenotype (that is RAG1/2, Artemis, IL-2R-alpha, IL7-R-gamma, DNA Ligase IV, JAK3, ADA) are eligible for this study.

2. T-cells (CD3+) > 100/µl. This threshhold is arbitrarily chosen as indicative of residual function of the affected gene product.

Exclusion Criteria

1. Patients with mutations in the following SCID genes due to the presence of T cells in a significant proportion of patients with null mutations: Fox N1 (WHN), CD45, CD3 (delta, epsilon, gamma, theta), Cernunnous, Coronin A1, Orai-1, ZAP-70, MHC II, RMRP, PNP and del22q.11. Patients with mutations in AK2 are also excluded.

2. Maternal T-cells (this must be investigated for a patient to be included)

3. Clinical presentation of classical SCID:
a.) Opportunistic infections (e.g. PCP-related pneumonia, invasive fungal infections, mycobacterial infections) in the first year of life
b.) Severe infections (major-infections) combined with failure to thrive, respectively persistent (> 4 weeks)/ invasive Candida infection in the first year of life

4.Clinical presentation of Omenn syndrome:
(e.g. erythematous rash, lymphoproliferation, severe/opportunistic infections, failure to thrive in the first year of life)

Study & Design

Study Type
observational
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
prevalence of each clinical and immunological feature in the full cohort of patients with atypical SCID
Secondary Outcome Measures
NameTimeMethod
percentage of patients with normal numbers of T-, B- and NK- or naïve T cells, nomal levels of immunoglobulins or specific antibodies in order to further emphasize the observation that these results do not exclude the diagnosis of SCID || Time to BMT, survival time
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