Rubinstein-Taybi综合征伴严重湿疹、复发性感染和高IgE对Dupilumab治疗的反应

IF 1.2 4区 医学 Q3 DERMATOLOGY Pediatric Dermatology Pub Date : 2025-07-01 Epub Date: 2025-02-05 DOI:10.1111/pde.15874
Arti Nanda, Maysoun Al Rushood, Waguih W Tawfeek, Atlal AlLafi
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引用次数: 0

摘要

鲁宾斯坦-泰比综合征(RTS)是一种罕见的常染色体显性遗传病,其特征为出生后生长迟缓、面部畸形、大拇指和幻觉以及智力缺陷。RTS具有广泛的表型谱,包括心脏、泌尿生殖系统、消化系统、耳、鼻、喉和皮肤表现。患者发生严重感染、良性肿瘤和提示原发性免疫缺陷的免疫异常的风险也增加。我们报告了一例RTS患者,伴有严重的顽固性湿疹、复发性皮肤和胸部感染、与高ige相关的食物过敏和其他免疫异常,他对dupilumab有显著的反应。
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Rubinstein-Taybi Syndrome With Severe Eczema, Recurrent Infections, and Hyper IgE Profile Responsive to Dupilumab Treatment.

Rubinstein-Taybi syndrome (RTS) is a rare autosomal dominant disorder characterized by post-natal growth retardation, facial dysmorphism, large thumbs and halluces, and intellectual deficits. RTS has a broad phenotypic spectrum including cardiac, genitourinary, digestive, ear, nose and throat, and skin manifestations. Patients also have an increased risk of severe infections, developing benign tumors, and immunological abnormalities suggesting primary immunodeficiencies. We report a case of RTS with severe recalcitrant eczema, recurrent skin and chest infections, food allergies associated with a hyper-IgE profile, and other immunological abnormalities, who had a significant response to dupilumab.

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来源期刊
Pediatric Dermatology
Pediatric Dermatology 医学-皮肤病学
CiteScore
3.20
自引率
6.70%
发文量
269
审稿时长
1 months
期刊介绍: Pediatric Dermatology answers the need for new ideas and strategies for today''s pediatrician or dermatologist. As a teaching vehicle, the Journal is still unsurpassed and it will continue to present the latest on topics such as hemangiomas, atopic dermatitis, rare and unusual presentations of childhood diseases, neonatal medicine, and therapeutic advances. As important progress is made in any area involving infants and children, Pediatric Dermatology is there to publish the findings.
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