Department of Pediatrics, Instituto da Criança e do Adolescente, Hospital das Clínicas, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.
Department of Dermatology, Hospital das Clínicas, Faculdade de Medicina, Universidade de São Paulo, São Paulo, SP, Brazil.
Einstein (Sao Paulo). 2023 May 29;21:eRC0256. doi: 10.31744/einstein_journal/2023RC0256. eCollection 2023.
A male infant presented with progressive jaundice immediately after birth. Fecal acholia and choluria associated with extensive bullous skin lesions in his trunk, abdomen, and upper and lower limbs developed during phototherapy. Several diagnostic hypotheses were presented, including neonatal porphyria, hemochromatosis, Alagille syndrome, and neonatal lupus. A 24-hour urine sample for the dosage of urinary porphyrins was collected, showing high results (1823.6µg in 100mL). At 50 days of life, fluorescence spectroscopy using a Wood's lamp revealed simultaneous bright red fluorescence of urine-stained diapers and sample blood. A definitive diagnosis of congenital erythropoietic porphyria was made following identification of a mutation of the uroporphyrinogen synthetases III gene on genetic testing. The patient was subsequently maintained in a low light environment since then, resulting in improvement of the lesions. Congenital erythropoietic porphyria is a disease of the group of porphyrias that presents shortly after birth with blistering occurring in regions exposed to the sun or other ultraviolet light. Atrophic scars, mutilated fingers, and bright red fluorescence of the urine and teeth may also be observed. There is no specific treatment, and prophylaxis comprising a total avoidance of sunlight is generally recommended. A high degree of suspicion is required for diagnosis. An early diagnosis can lead to less damage. Here, we present the case of a newborn with congenital erythropoietic porphyria diagnosed after presenting with bullous lesions secondary to phototherapy.
一名男婴出生后即出现进行性黄疸。在光疗期间,他的躯干、腹部、上下肢出现广泛大疱性皮肤损伤,并伴有粪便色胆素缺失和尿胆素缺失。提出了几种诊断假设,包括新生儿卟啉症、血色病、Alagille 综合征和新生儿狼疮。采集了 24 小时尿液样本以测定尿卟啉的含量,结果显示含量很高(100ml 中有 1823.6μg)。在出生后 50 天时,使用伍德灯进行荧光光谱检查显示,尿染尿布和样本血液同时呈现明亮的红色荧光。在基因检测中发现尿卟啉原 III 合成酶基因突变后,明确诊断为先天性红细胞生成性卟啉症。此后,患者一直生活在低光照环境中,病变得到改善。先天性红细胞生成性卟啉症是一组卟啉症中的一种疾病,在出生后不久即出现,在暴露于阳光或其他紫外线的区域出现水疱。萎缩性瘢痕、残缺的手指以及尿液和牙齿的明亮红色荧光也可能被观察到。目前尚无特定的治疗方法,通常建议进行全面避免阳光照射的预防。诊断需要高度怀疑。早期诊断可以减少损伤。在这里,我们报告了一例新生儿病例,该新生儿因光疗后出现大疱性病变而被诊断为先天性红细胞生成性卟啉症。