Baykal Can, Topkarci Zeynep, Yazganoglu K Didem, Azizlerli Gülsevim, Baykan Betül
Department of Dermatology, Istanbul Medical Faculty, Istanbul University, Istanbul, Turkey.
Int J Dermatol. 2007 Oct;46(10):1011-6. doi: 10.1111/j.1365-4632.2007.03115.x.
Lipoid proteinosis (LP) is a very rare genodermatosis. The literature on LP consists of case reports only. As we have observed 14 LP patients belonging to nine different families in the last 15 years in our practice, we decided to review all reported Turkish LP patients in this 15-year period, and noted 37 diagnosed cases. The reasons for this relatively large number of cases, the clinical features of the patients, and the associations of LP with other clinical conditions are described in this article.
Fourteen LP patients followed in our university clinic in Istanbul were scrutinized with regard to their demographic and clinical features. Diagnoses were established using clinical features, with histopathologic confirmation in 13 cases.
All but one of the patients had a history of consanguinity, or at least a marriage of parents from the same village. Typical cutaneous signs of LP and hoarseness of the voice were observed in all patients. Two patients of the same pedigree had insulin-dependent diabetes mellitus (IDDM), two patients from two different pedigrees had short stature, one patient had multinodular toxic goiter, and one patient had celiac disease.
LP is not rare in Turkey as consanguineous marriage is still a social problem, especially in some rural areas. The disease is not limited to a particular geographic region in Turkey. Short stature was observed in two cases from two different families, an association not reported previously; the association of LP with IDDM in one pedigree was thought to be coincidental.
类脂蛋白沉积症(LP)是一种非常罕见的遗传性皮肤病。关于LP的文献仅包含病例报告。由于在过去15年的临床实践中我们观察到14例LP患者,分属于9个不同家庭,我们决定回顾这15年间所有报道的土耳其LP患者,并记录到37例确诊病例。本文描述了这一相对大量病例的原因、患者的临床特征以及LP与其他临床病症的关联。
对在伊斯坦布尔我们大学诊所随访的14例LP患者的人口统计学和临床特征进行了详细检查。根据临床特征进行诊断,13例经组织病理学证实。
除1例患者外,所有患者都有近亲结婚史,或至少父母来自同一个村庄。所有患者均观察到LP的典型皮肤体征和声音嘶哑。同一家系的2例患者患有胰岛素依赖型糖尿病(IDDM),来自两个不同家系的2例患者身材矮小,1例患者患有多结节性毒性甲状腺肿,1例患者患有乳糜泻。
由于近亲结婚仍是一个社会问题,尤其是在一些农村地区,LP在土耳其并不罕见。该病在土耳其并不局限于特定地理区域。在来自两个不同家庭的2例患者中观察到身材矮小,这一关联此前未被报道;一个家系中LP与IDDM的关联被认为是偶然的。