Ezgi Karatas, Canan Asli Utine, Banu Lebe, Mahmut Kaya
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引用次数: 0
摘要
我们介绍了一例推测为色素性红斑狼疮(XP)并伴有眼窝发育不全的病例。一名 50 岁的男性患者患有广泛的双侧对称性假性翼状胬肉,因视力康复而被转诊。经过皮肤科会诊和眼科检查,诊断为假性翼状胬肉。光学相干断层扫描显示该患者的眼窝发育不良程度为 2 级。由于同时患有 XP 和眼窝发育不全的情况非常罕见,患者被转诊接受基因检测。基因检测结果显示一些基因发生了突变,包括血色沉着病基因HFE、COL1A2、溶酶体运输调节器(LYST)、NF1和HMBS。已知 LYST 基因与眼窝发育不全有关。由于文献中曾报道过另一例眼窝发育不全与 XP 相关的病例,因此我们介绍了我们的病例,与大家分享这一罕见的关联。
Foveal Hypoplasia in Presumed Xeroderma Pigmentosum: A Case Report.
We present a case of presumed xeroderma pigmentosum (XP) with concomitant foveal hypoplasia. A 50-year-old male patient with extensive bilateral symblepharon-like pseudopterygia was referred for visual rehabilitation. After dermatology consultation and ophthalmologic examination, presumed XP was diagnosed. Optical coherence tomography revealed grade 2 foveal hypoplasia. The patient was referred for genetic testing because concomitant XP and foveal hypoplasia are rare. The genetic test results revealed mutations in some genes, including the hemochromatosis genes HFE, COL1A2, Lysosome Trafficking Regulator (LYST), NF1, and HMBS. The LYST gene is known to be associated with foveal hypoplasia. Since the association of foveal hypoplasia and XP has been reported in another case in the literature, we present our case to share this rare association.