نتایج جستجو برای: harlequin ichthyosis

تعداد نتایج: 2442  

Journal: :International Journal of Pregnancy & Child Birth 2021

Journal: :Journal of Clinical Investigation 2005

Journal: :CMAJ : Canadian Medical Association journal = journal de l'Association medicale canadienne 2010
Judy Tang James Bergman Joseph M Lam

CMAJ • NOVEMBER 23, 2010 • 182(17) © 2010 Canadian Medical Association or its licensors E801 Ahealthy girl was born at term after an uncomplicated pregnancy and delivery. When she was five days old, unilateral erythema with contralateral pallor developed that was strikingly demarcated at midline (Figure 1). This phenomenon occurred after the infant had been placed on her right side, with the er...

Journal: :The Journal of clinical investigation 2005
Alain Hovnanian

Harlequin ichthyosis (HI)--the most severe form of keratinizing disorders, often lethal in the neonatal period--is characterized by a profound thickening of the keratin skin layer, a dense "armor"-like scale that covers the body, and contraction abnormalities of the eyes, ears, and mouth. In this issue of the JCI, Akiyama et al. report that mutations in ABCA12 caused defective lipid transport t...

2013
Deren Özcan Murat Derbent Deniz Seçkin Yunus Emre Bikmaz Muhteşem Ağildere Annachiara De Sandre-Giovannoli Nicolas Lévy Berkan Gürakan

Neu-Laxova syndrome is a rare, lethal, autosomal recessive disorder characterized by intrauterine growth retardation, central nervous system anomalies, skin findings, such as ichthyosis, edema, collodion baby and harlequin fetus, facial dysmorphic features, limb anomalies and genital hypoplasia. Although it is generally a lethal condition, cases of such patients who lived beyond 6 months and 10...

Journal: :Human molecular genetics 2015
Denny L Cottle Gloria M A Ursino Sally Chi Ieng Ip Lynelle K Jones Tia Ditommaso Douglas F Hacking Niamh E Mangan Natalie A Mellett Katya J Henley Dmitri Sviridov Claudia A Nold-Petry Marcel F Nold Peter J Meikle Benjamin T Kile Ian M Smyth

Harlequin ichthyosis (HI) is a severe skin disease which leads to neonatal death in ∼50% of cases. It is the result of mutations in ABCA12, a protein that transports lipids required to establish the protective skin barrier needed after birth. To better understand the life-threatening newborn HI phenotype, we analysed the developing epidermis for consequences of lipid dysregulation in mouse mode...

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