Successful treatment with secukinumab of psoriasis-like dermatitis in a patient with holocarboxylase synthetase deficiency

被引:0
作者
Liu, Haifei [1 ,2 ]
Wei, Ruoqu [1 ,2 ]
Yang, Yijun [1 ,2 ]
Zhang, Zhen [1 ,2 ]
Yang, Yixuan [1 ,2 ]
Tang, Jue [1 ,2 ]
Chen, Jiawen [1 ,2 ]
Zhang, Jia [1 ,2 ]
Gu, Yan [1 ,2 ]
Yao, Zhirong [1 ,2 ]
机构
[1] Shanghai Jiao Tong Univ, Xinhua Hosp, Sch Med, Dept Dermatol, Shanghai, Peoples R China
[2] Shanghai Jiao Tong Univ, Sch Med, Inst Dermatol, Shanghai, Peoples R China
关键词
holocarboxylase synthetase deficiency; IL-17A; psoriasis-like dermatitis; secukinumab; treatment;
D O I
10.1111/1346-8138.16625
中图分类号
R75 [皮肤病学与性病学];
学科分类号
100206 ;
摘要
Holocarboxylase synthetase deficiency (HSD) is a rare autosomal recessive disorder of biotin metabolism. Typical manifestations include irreversible metabolic disorders and erythroderma-like dermatitis. Most patients respond well to biotin supplementation. Psoriasis-like phenotype associated with this disease has been rarely reported in the literature and experiences with the use of biologics in patients with HSD are still lacking. We reported a rare case of recurrent psoriasis-like skin lesions in a 6-year-old child with HSD. The patient did not respond to initial therapy with high-dose oral biotin. Immunofluorescence staining showed an increased number of interleukin (IL)-17A+ cells in his skin lesions. Based on this finding, the patient was successfully treated with human anti-IL-17A monoclonal antibody (secukinumab). He did not report any side effects and remained healthy during the 2-year follow-up. We provide a comprehensive review of the reported cases of HSD with psoriasis-like dermatitis to date. The psoriasis-like phenotype of HSD is controversial in treatment and IL-17A inhibitor is an alternative therapeutic option.
引用
收藏
页码:401 / 406
页数:6
相关论文
共 11 条
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Zheng, Zhenzhu ;
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BMC MEDICAL GENETICS, 2020, 21 (01)