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迟发性皮肤卟啉症:与复合杂合性粪卟啉原氧化酶(CPOX)突变相关的终身光敏性病例。

Harderoporphyria: Case of lifelong photosensitivity associated with compound heterozygous coproporphyrinogen oxidase (CPOX) mutations.

作者信息

Moghe A, Ramanujam V M S, Phillips J D, Desnick R J, Anderson K E

机构信息

Department of Preventive Medicine and Community Health, University of Texas Medical Branch, Galveston, TX, USA.

Department of Medicine, University of Utah, Salt Lake City, UT, USA.

出版信息

Mol Genet Metab Rep. 2019 Feb 19;19:100457. doi: 10.1016/j.ymgmr.2019.100457. eCollection 2019 Jun.

Abstract

A 78-year-old man with a history of neonatal anemia and jaundice and life-long photosensitivity was found to have harderoporphyria, as evidenced by increased porphyrins in urine, plasma, erythrocytes and feces including large amounts of harderoporphyrin in feces and erythrocytes. Two previously undescribed coproporphyrinogen oxidase (CPOX) mutations were identified, including a deletion of four amino acids in a region of the enzyme mutated in 7 of the 8 previously reported cases. This case increases the molecular heterogeneity of this rare porphyria, and illustrates that it should be considered as a cause of chronic photosensitivity and porphyrin elevation at any age.

摘要

一名78岁男性,有新生儿贫血和黄疸病史,且终生对光敏感,被诊断为硬卟啉病,尿液、血浆、红细胞和粪便中的卟啉增加可证明,粪便和红细胞中含有大量硬卟啉。鉴定出两个以前未描述的粪卟啉原氧化酶(CPOX)突变,其中一个突变区域有四个氨基酸缺失,在之前报道的8个病例中有7个发生了该区域的突变。该病例增加了这种罕见卟啉病的分子异质性,并表明应将其视为任何年龄慢性光敏性和卟啉升高的一个病因。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/c6cb/6383327/0c161c52d764/gr1.jpg

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