Sahn E E, Salinas C F, Sens M A, Key J, Swiger F K, Holbrook K A
Department of Dermatology, Medical University of South Carolina, Charleston 29425-2215.
Pediatr Dermatol. 1994 Mar;11(1):52-60. doi: 10.1111/j.1525-1470.1994.tb00076.x.
A black girl was born with flexion contractures and experienced pain on movement by 1 week of age. She subsequently developed perioral papules, gingival hyperplasia, perianal nodules, torticollis, diarrhea, rectal prolapse, and inability to open her mouth. Her skin became increasingly sclerodermatous, and velvety, hyperpigmented plaques arose over bony prominences. A skin biopsy specimen showed hyaline material in the papillary dermis with lack of elastic fibers. Ultrastructural examination revealed fibrillogranular material around fibroblasts and blood vessels. This child had the clinical, histologic, and ultrastructural features of infantile systemic hyalinosis. This disorder has not been described in a black infant. Previous case reports of infantile systemic hyalinosis are reviewed and unusual features of our case are discussed.
一名黑人女孩出生时即患有屈曲挛缩,在1周龄时出现活动时疼痛。随后她出现了口周丘疹、牙龈增生、肛周结节、斜颈、腹泻、直肠脱垂以及无法张口。她的皮肤逐渐变得硬皮病样,骨隆突处出现天鹅绒样、色素沉着过度的斑块。皮肤活检标本显示乳头真皮中有透明物质且缺乏弹性纤维。超微结构检查显示成纤维细胞和血管周围有纤维颗粒物质。该患儿具有婴儿系统性透明变性的临床、组织学和超微结构特征。这种疾病在黑人婴儿中尚未有过描述。回顾了既往婴儿系统性透明变性的病例报告并讨论了本病例的不寻常特征。