http://dx.doi.org/10.1007/s00431-008-0718-x
scopus: cited 17 times
web of science: cited 15 times
Clinical heterogeneity can hamper the diagnosis of patients with ZAP70 deficiency
January 2009
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One of the severe combined immunodeficiencies (SCIDs), which is caused by a genetic defect in the signal transduction pathways involved in T-cell activation, is the ZAP70 deficiency. Mutations in ZAP70 lead to both abnormal thymic development and defective T-cell receptor (TCR) signaling of peripheral T-cells. In contrast to the lymphopenia in most SCID patients, ZAP70-deficient patients have lymphocytosis, despite the selective absence of CD8+ T-cells. The clinical presentation is usually before 2 years of age with typical findings of SCID. Here, we present three new ZAP70-deficient patients who vary in their clinical presentation. One of the ZAP70-deficient patients presented as a classical SCID, the second patient presented as a healthy looking wheezy infant, whereas the third patient came to clinical attention for the eczematous skin lesions simulating atopic dermatitis with eosinophilia and elevated immunoglobulin E (IgE), similar to the Omenn syndrome. This study illustrates that awareness of the clinical heterogeneity of ZAP70 deficiency is of utmost importance for making a fast and accurate diagnosis, which will contribute to the improvement of the adequate treatment of this severe immunodeficiency.
- patient
- t-cell
- mutation
- deficiency
- 70 deficiency
- immunodeficiency
- month
- kinase
- expression
- defect
- cd 8+ t-cells
- protein
- infection
- absence
- t-cell receptor
- patients 1
- 70 gene
- tyrosine kinase domain
- receptor
- cd 4+ t-cells