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新生儿 X 连锁原卟啉症光疗后出现严重反应。

A Severe Reaction After Phototherapy in a Neonate With X-Linked Protoporphyria.

机构信息

Divisions of Neonatology.

Medical Genetics and Genomics.

出版信息

Pediatrics. 2024 Aug 1;154(2). doi: 10.1542/peds.2023-064724.

Abstract

Protoporphyria is a subtype of porphyria characterized primarily by painful phototoxic skin reactions after light exposure at specific wavelengths. Historically, phototherapy is not contraindicated in patients with protoporphyria since there have not been any reports of phototoxic reactions. However, patients with protoporphyria are advised to avoid direct sunlight. In this case report, we describe a neonate not known to have X-linked protoporphyria who received phototherapy for 1 to 2 hours. Within hours after initiation of phototherapy, this neonate developed a life-threatening reaction consisting of rash over the distribution of phototherapy, acute liver failure with coagulopathy, diffuse hypotonia with diaphragmatic failure, and subsequent acute respiratory failure that required mechanical ventilation. As in this case, patients with protoporphyria-related acute liver failure can have signs and symptoms similar to that of an acute hepatic porphyria attack. Neither neonatal reactions to phototherapy nor liver failure temporally associated with phototherapy have been reported in patients with X-linked protoporphyria. Early recognition of this entity is crucial in light of potential life-threatening complications. Therefore, providers must react quickly when neonates have abnormal reactions to phototherapy and consider protoporphyria in the differential diagnosis.

摘要

原卟啉症是一种卟啉症亚型,主要特征是在特定波长的光照射后出现疼痛性光毒性皮肤反应。历史上,由于没有光毒性反应的报道,光疗法在原卟啉症患者中并不被禁忌。然而,建议原卟啉症患者避免阳光直射。在本病例报告中,我们描述了一名新生儿,其并不患有 X 连锁原卟啉症,但接受了 1 至 2 小时的光疗。在开始光疗后数小时内,该新生儿出现了危及生命的反应,包括光疗分布区域的皮疹、伴有凝血功能障碍的急性肝衰竭、弥漫性低张力伴膈肌衰竭,随后发生急性呼吸衰竭,需要机械通气。正如本例中,与原卟啉症相关的急性肝衰竭患者可能具有类似于急性肝卟啉症发作的体征和症状。在 X 连锁原卟啉症患者中,尚未报道与光疗相关的新生儿反应或与光疗同时发生的肝衰竭。鉴于潜在的危及生命的并发症,早期识别该病症至关重要。因此,当新生儿对光疗出现异常反应时,提供者必须迅速做出反应,并考虑在鉴别诊断中包括原卟啉症。

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