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Cyclosporin A in hyperimmunoglobulin E syndrome

A Etzioni¹, N Shehadeh, A Brecher

¹Department of Pediatrics and Clinical Immunology, Rambam Medical Center, Haifa, Israel.

Annals of Allergy, Asthma & Immunology : Official Publication of the American College of Allergy, Asthma, & Immunology

|April 1, 1997

Summary

Low-dose Cyclosporin A (CsA) shows promise for treating hyperimmunoglobulinemia E syndrome (HIES). This immunosuppressive therapy improved clinical symptoms and immune function in a young patient unresponsive to other treatments.

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Area of Science:

Immunology
Clinical Medicine

Background:

Hyperimmunoglobulinemia E syndrome (HIES) is a severe primary immunodeficiency with unknown causes and limited treatment options.
Cyclosporin A (CsA), an immunosuppressant, was investigated for its potential therapeutic effects in HIES.

Observation:

A 3-year-old boy with severe HIES, refractory to multiple treatments, received low-dose CsA (3 mg/kg/d) for six months.
Humoral and cellular immune functions, alongside clinical status, were monitored throughout the treatment period.

Findings:

Cyclosporin A therapy resulted in significant clinical improvement.
Key immunological improvements included a marked reduction in serum IgE levels and enhanced neutrophil chemotactic function.
No alterations in CD40 ligand expression were noted, and CsA blood levels remained below 150 ng/mL.

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Implications:

Low-dose Cyclosporin A (3-5 mg/kg/d) appears beneficial for managing severe hyperimmunoglobulinemia E syndrome.
CsA should be considered as a therapeutic option for HIES patients who have not responded to conventional treatments.