Ranganath Lakshminarayan, Khedr Milad, Evans Leanne A, Bygott Helen, Luangrath Emily, West Elizabeth
Department of Clinical Biochemistry and Metabolic Medicine Royal Liverpool University Hospital Liverpool UK.
Department of Dermatology Royal Liverpool University Hospital Liverpool UK.
JIMD Rep. 2021 Jun 14;61(1):25-33. doi: 10.1002/jmd2.12225. eCollection 2021 Sep.
Four patients, from three families, with alkaptonuria receiving 4-hydroxyphenylpyruvate dioxygenase-inhibiting nitisinone therapy, which lowers homogentisic acid and increases tyrosine, developed vitiligo. Three of the four patients were receiving nitisinone 2 mg daily, while the fourth was on 10 mg daily. All four patients were either receiving or had received transiently proton-pump inhibitors as therapy for dyspepsia. The ages of the patients were 35, 42, 40, and 67 years, respectively. Three patients were men and one was a woman. All four patients were either taking a proton-pump inhibitor or had been taking one at some point. Three of the four were of South Asian and one of Caucasian background. The three patients with South Asian background also had either a personal or family history of autoimmune disease. Distressing vitiligo, initially in an acrofacial distribution, developed unexpectedly in these four patients, before then progressing to involve other parts of the body. Potential factors in the appearance of vitiligo in this setting, including nitisinone and other drug therapy, are explored and responses to the appearance of vitiligo are discussed.
来自三个家庭的四名患有黑尿症的患者接受了抑制4-羟基苯丙酮酸双加氧酶的尼替西农治疗，该治疗降低了尿黑酸并增加了酪氨酸，结果这四名患者都出现了白癜风。四名患者中有三名每天服用2毫克尼替西农，第四名患者每天服用10毫克。所有四名患者都正在接受或曾短暂接受过质子泵抑制剂治疗消化不良。患者年龄分别为35岁、42岁、40岁和67岁。三名患者为男性，一名为女性。所有四名患者都正在服用或曾在某个时候服用过质子泵抑制剂。四名患者中有三名具有南亚背景，一名具有白种人背景。三名具有南亚背景的患者也有自身免疫性疾病的个人史或家族史。这四名患者意外出现了令人苦恼的白癜风，最初表现为肢端面部分布，随后发展到身体其他部位。本文探讨了在这种情况下白癜风出现的潜在因素，包括尼替西农和其他药物治疗，并讨论了对白癜风出现的应对措施。
