Literature DB >> 19305025

[Variable clinical expression of familial Incontinentia Pigmenti syndrome - presentation of three cases].

Anna Kutkowska-Kaźmierczak1, Ewa Obersztyn, Jean-Paul Bonnefont, Danuta Rosińska-Borkowska, Tomasz Mazurczak, Agnieszka Sobczyńska-Tomaszewska, Tadeusz Mazurczak.   

Abstract

Incontinentia Pigmenti (IP, Bloch-Sulzberger syndrome, OMIM 308300) is a rare X-linked dominant genodermatosis, usually lethal in males in the prenatal period. Wide spectrum of clinical expression consists of skin hyperpigmented lines and swirling patterns, dysplastic teeth and nails, and in 30% central nervous system abnormalities including seizures, microcephaly and intellectual disability (10% of cases). In 80% of IP cases, the disease is caused by a large-scale deletion of exons 4 to 10 of the NEMO gene. Three cases of variable expression of Incontinentia Pigmenti are presented. In a one-year-old girl, her mother and grandmother molecular analysis revealed the same typical deletion of the NEMO gene. In the proband, characteristic skin lesions were detected located over the trunk and lower limbs. Characteristic evolution of the changes was observed. In the mother, expression of the disease was much milder, whereas in the grandmother lesions were restricted to the fingernails. Clinical characteristics and pedigree data are described.

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Year:  2008        PMID: 19305025

Source DB:  PubMed          Journal:  Med Wieku Rozwoj


  1 in total

1.  Incontinentia Pigmenti: A Case Report of a Complex Systemic Disease.

Authors:  Serena Gianfaldoni; Georgi Tchernev; Uwe Wollina; Torello Lotti
Journal:  Open Access Maced J Med Sci       Date:  2017-07-23
  1 in total

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