Gündüz Ozgür, Sahiner Neriman, Atasoy Pınar, Senyücel Cağrı
Department of Dermatology and Venerology, Suleyman Demirel Teaching Hospital, University of Kırıkkale, Fabrikalar Mahallesi, Saglık Sokak, Kırıkkale 71100, Turkey.
Case Rep Dermatol Med. 2012;2012:324506. doi: 10.1155/2012/324506. Epub 2012 Aug 9.
Lipoid proteinosis (LP) is a rare, autosomal-recessive disease characterized by the hoarseness and widespread cutaneous scarring, more prominent on sun-exposed areas. Yellow-white plaques can be seen on oral mucosa and on the skin among depressed scars. Histological evaluation of the affected sites shows accumulation of hyaline-like material in dermis and disruption of basement membrane. Although LP is compatible with normal life expectancy, involvement of upper respiratory tract may endanger patient's life, especially in the case of a respiratory tract infection. Involvement of central nervous system has also been reported, but its clinical importance is obscure. Due to the rarity of LP, a definite therapeutical approach is not established. In this paper we describe a 21-year-old LP patient who was treated with acitretin for six months. Although the outcome with cutaneous lesions was not satisfactory, her hoarseness was significantly improved.
类脂蛋白沉积症(LP)是一种罕见的常染色体隐性疾病,其特征为声音嘶哑和广泛的皮肤瘢痕形成,在暴露于阳光下的部位更为明显。在口腔黏膜和凹陷瘢痕处的皮肤上可见黄白色斑块。对受累部位的组织学评估显示真皮中透明样物质的积聚和基底膜的破坏。虽然LP患者的预期寿命正常,但上呼吸道受累可能危及患者生命,尤其是在呼吸道感染的情况下。也有中枢神经系统受累的报道,但其临床重要性尚不清楚。由于LP罕见,尚未确立明确的治疗方法。在本文中,我们描述了一名21岁的LP患者,她接受阿维A治疗6个月。虽然皮肤病变的治疗效果不尽人意,但她的声音嘶哑明显改善。