Jóźwiak S, Schwartz R A, Janniger C K, Michałowicz R, Chmielik J
Neurology, The Children's Memorial Health Institute, Warsaw, Poland.
Int J Dermatol. 1998 Dec;37(12):911-7. doi: 10.1046/j.1365-4362.1998.00495.x.
Tuberous sclerosis complex (TSC) is an autosomal dominant disorder characterized by widespread cutaneous and visceral hamartomas.
The prevalence of cutaneous lesions in 106 children with TSC (47 boys and 59 girls) aged 1 month-18 years was evaluated from 1984 to 1995. Assessing the diagnostic usefulness of each National Tuberous Sclerosis Association skin criterion was an aim of this study.
Hypopigmented macules were the most frequent finding, seen in 103 of 106 children (97.2%). In 66 children they were evident at birth, and in 20 others their presentation was delayed until the first months of age. Facial angiofibromas were seen next most often (79 of 103, 74.5%), followed by a shagreen patch in 51 of 103 (48.1%), "cafe-au-lait" macules in 30 of 103 (28.3%), molluscum fibrosum pendulum (24 of 103, 22.6%), a forehead fibrous plaque (20 of 103, 18.9%), periungual fibromas (16 of 103, 15.1%) and "confetti-like" macules (3 of 103, 2.8%). The hypomelanotic macules were seen within the first 2 years of life in 95 children, as were café-au-lait spots in 24, facial angiofibromas in eight, shagreen patches in six, and forehead fibrous plaques in six, whereas molluscum pendulum and periungual fibromas were not evident. Seizures were seen in 102 of 106 children (98%), with 80 (75%) occurring during the first year of life.
Hypomelanotic macules were the overwhelmingly most common early finding in TSC. Infants with seizures or other possible stigmata of TSC should be carefully evaluated for these hypomelanotic macules, as well as for other associated findings.
结节性硬化症(TSC)是一种常染色体显性疾病,其特征为广泛的皮肤和内脏错构瘤。
1984年至1995年期间,对106例年龄在1个月至18岁的结节性硬化症患儿(47名男孩和59名女孩)的皮肤损害患病率进行了评估。本研究的目的是评估美国结节性硬化症协会的每项皮肤诊断标准的实用性。
色素减退斑是最常见的表现,106例患儿中有103例出现(97.2%)。其中66例在出生时即明显可见,另外20例直到出生后的头几个月才出现。面部血管纤维瘤次之(103例中有79例,74.5%),其次是鲨革斑,103例中有51例(48.1%),“牛奶咖啡”斑103例中有30例(28.3%),纤维软瘤(103例中有24例,22.6%),前额纤维斑块(103例中有20例,18.9%),甲周纤维瘤(103例中有16例,15.1%)和“纸屑样”斑(103例中有3例,2.8%)。95例患儿在出生后的头2年内出现色素减退斑,24例出现牛奶咖啡斑,8例出现面部血管纤维瘤,6例出现鲨革斑,6例出现前额纤维斑块,而纤维软瘤和甲周纤维瘤在出生后的头2年内不明显。106例患儿中有102例出现癫痫发作(98%),其中80例(75%)在出生后的第一年内出现。
色素减退斑是结节性硬化症早期最常见的表现。对于有癫痫发作或其他可能患有结节性硬化症体征的婴儿,应仔细评估是否存在这些色素减退斑以及其他相关表现。