CHU de Québec, Centre Hospitalier de l'Université Laval, Department of Pediatrics, Université Laval, Québec City, QC, Canada.
CHU de Québec, Centre Hospitalier de l'Université Laval, Division of Dermatology, Department of Medicine, Université Laval, Québec City, QC, Canada.
Clin Pediatr (Phila). 2023 Jun;62(5):399-403. doi: 10.1177/00099228221128661. Epub 2022 Oct 11.
Congenital erythropoietic porphyria (CEP), a rare form of porphyria, is caused by a defect in the heme biosynthesis pathway of the enzyme uroporphyrinogen III synthase (UROS). Uroporphyrinogen III synthase deficiency leads to an accumulation of nonphysiological porphyrins in bone marrow, red blood cells, skin, bones, teeth, and spleen. Consequently, the exposure to sunlight causes severe photosensitivity, long-term intravascular hemolysis, and eventually, irreversible mutilating deformities. Several supportive therapies such as strict sun avoidance, physical sunblocks, red blood cells transfusions, hydroxyurea, and splenectomy are commonly used in the management of CEP. Currently, the only available curative treatment of CEP is hematopoietic stem cell transplantation (HSCT). In this article, we present a young girl in which precocious genetic testing enabled early diagnosis and allowed curative treatment with HSCT for CEP at the age of 3 months of age, that is, the youngest reported case thus far.
先天性红细胞生成性卟啉症(CEP)是一种罕见的卟啉症,由尿卟啉原 III 合酶(UROS)酶的血红素生物合成途径缺陷引起。尿卟啉原 III 合酶缺乏导致非生理卟啉在骨髓、红细胞、皮肤、骨骼、牙齿和脾脏中积累。因此,暴露在阳光下会导致严重的光敏性、长期的血管内溶血,最终导致不可逆转的致残畸形。在 CEP 的治疗中,通常采用严格的防晒、物理防晒霜、红细胞输注、羟基脲和脾切除术等支持性治疗。目前,CEP 唯一可用的治愈性治疗方法是造血干细胞移植(HSCT)。在本文中,我们介绍了一名年轻女孩,早期基因检测使她能够在 3 个月大时就被早期诊断,并通过 HSCT 进行治愈性治疗,这是迄今为止报告的最小年龄。