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How should eosinophilic cystitis be treated in patients with chronic granulomatous disease?
Alessia Claps1, Martina Della Corte, Simona Gerocarni Nappo
1Unit of Immunology and Infectious Diseases, University-Hospital Pediatric Department, Bambino Gesù Children Hospital, IRCCS, Rome, Italy.
Insights
Eosinophilic cystitis (EC) in children with chronic granulomatous disease (CGD) presents a prolonged and recurrent course, unlike in immunocompetent children. This study focuses on effective therapies for EC in CGD patients.
Area of Science:
- Immunology
- Pediatrics
- Urology
Background:
- Chronic granulomatous disease (CGD) is a primary immunodeficiency affecting phagocytic cell oxidative mechanisms, leading to severe infections and inflammation.
- Eosinophilic cystitis (EC) is a rare inflammatory bladder condition, with varied therapeutic approaches.
- EC may be part of the inflammatory spectrum observed in CGD patients.
Background:
Chronic granulomatous disease (CGD) is a primary immunodeficiency resulting from the absence or malfunction of oxidative mechanism in phagocytic cells. The disease is due to a mutation in one of four genes that encode subunits of the nicotinamide adenine dinucleotide phosphate (NADPH) oxidase complex. Affected patients experience severe infections and granuloma formation due to exuberant inflammatory responses. Some evidence suggests that eosinophilic cystitis (EC) is included in the spectrum of inflammatory manifestations. EC is an inflammatory disease, rare in childhood, which may require different, nonstandardized therapeutic approaches, ranging from antihistamines to cyclosporine.
Case-Diagnosis/Treatment:
Herein we describe the cases of two CGD patients with CGD who experienced EC during hospitalization for a severe infection.
Conclusions:
EC in immunocompetent children seems to have a self-limiting course, unlike in CGD patients, in whom it presents a prolonged and recurrent course. We focus on the effective therapy administered to our patients with CGD and review the corresponding literature.
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