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Sexual precocity in hypomelanosis of Ito: mosaicism-associated case report and literature review.
MedLine Citation:
PMID:  21244380     Owner:  NLM     Status:  In-Data-Review    
Abstract/OtherAbstract:
Background  Hypomelanosis of Ito (HI), a neurocutaneous disorder characterized by hypopigmented skin lesions along Blaschko's line with multiple accompanying diseases, may occasionally present with sexual precocity. Methods  We reviewed cases of HI with sexual precocity reported in the literature and focused on collecting information on other associated organ abnormalities, chromosomal karyotype, and type of sexual precocity. We also present our own case report. Results  Five children with sexual precocity in HI were studied. All patients were full-term females without significant family history. Their ages ranged from three years to 11 years. Skin lesions involved the trunk and limbs. Developmental delay was present in all patients. Central nervous system involvement was found in four patients. Craniofacial abnormalities were present in four patients. Eye involvement was present in two patients. Limb abnormalities (brachydactyly and clinodactyly) were present in two patients. Skeletal organ involvement was present in three patients. Other skin lesions were present in three patients. Dental abnormalities occurred in one patient. Of the five patients, four demonstrated a peripheral type of sexual precocity, and one presented with central type sexual precocity. Vaginal bleeding was present in two patients. Chromosomal karyotype abnormalities were found in two patients, and mosaicism was present in one of these. Conclusions  Hypomelanosis of Ito is a syndrome characterized by cutaneous signs frequently associated with nervous, ocular, and musculoskeletal system abnormalities. Although it is rarely reported in conjunction with HI, physicians should recognize sexual precocity as a possible associated symptom.
Authors:
Jin Mo Park; Hee Jung Kim; Taegyun Kim; Hyun Wook Chae; Duk Hee Kim; Min-Geol Lee
Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  International journal of dermatology     Volume:  50     ISSN:  1365-4632     ISO Abbreviation:  Int. J. Dermatol.     Publication Date:  2011 Feb 
Date Detail:
Created Date:  2011-01-19     Completed Date:  -     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  0243704     Medline TA:  Int J Dermatol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  168-74     Citation Subset:  IM    
Copyright Information:
© 2011 The International Society of Dermatology.
Affiliation:
Department of Dermatology, Yonsei University College of Medicine, Seoul, South Korea Cutaneous Biology Research Institute, Brain Korea 21 Project for Medical Science, Yonsei University College of Medicine, Seoul, South Korea Department of Dermatology, Kangbuk Samsung Hospital, Sunkyunkwan University School of Medicine, Seoul, South Korea Department of Pediatrics, Severance Children's Hospital, Yonsei University College of Medicine, Seoul, South Korea.
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