نتایج جستجو برای: autosomal recessive primary microcephaly

تعداد نتایج: 682552  

2013
Matthew A Cottee Nadine Muschalik Yao Liang Wong Christopher M Johnson Steven Johnson Antonina Andreeva Karen Oegema Susan M Lea Jordan W Raff Mark van Breugel

Centrioles organise centrosomes and template cilia and flagella. Several centriole and centrosome proteins have been linked to microcephaly (MCPH), a neuro-developmental disease associated with small brain size. CPAP (MCPH6) and STIL (MCPH7) are required for centriole assembly, but it is unclear how mutations in them lead to microcephaly. We show that the TCP domain of CPAP constitutes a novel ...

Journal: :American journal of medical genetics. Part A 2007
Robert P Erickson John B Bodensteiner

We describe four patients, two pairs of siblings, with a somewhat unique oro-facial-digital syndrome. The siblings come from the Navajo population which has undergone several genetic "bottlenecks." Thus, as would be anticipated, this syndrome seems to show autosomal recessive inheritance. The combination of the presence of retinal colobomata and the paucity of digital findings in these patients...

2004
Dominique Smeets Corry Weemaes

Nijmegen breakage syndrome (NBS), a rare autosomal recessive condition also known as ataxia telangiectasia (AT) variants VI and V2, is characterised by microcephaly, typical facies, short stature, immunodeficiency, and chromosomal instability. We report the clinical, immunological, chromosomal, and cell biological findings in 42 patients who are included in the NBS Registry in Nijmegen. The imm...

Journal: :Human molecular genetics 2012
Anne Genin Julie Desir Nelle Lambert Martine Biervliet Nathalie Van Der Aa Genevieve Pierquin Audrey Killian Mario Tosi Montse Urbina Anne Lefort Frederick Libert Isabelle Pirson Marc Abramowicz

Several genes expressed at the centrosome or spindle pole have been reported to underlie autosomal recessive primary microcephaly (MCPH), a neurodevelopmental disorder consisting of an important brain size reduction present since birth, associated with mild-to-moderate mental handicap and no other neurological feature nor associated malformation. Here, we report a mutation of CASC5 (aka Blinkin...

2011
Maha S Zaki Ghada M H Abdel Salam Sahar N Saleem William B Dobyns Mahmoud Y Issa Shifteh Sattar Joseph G Gleeson

We identified a two-branch consanguineous family in which four affected members (three females and one male) presented with constitutive growth delay, severe psychomotor retardation, microcephaly, cerebellar hypoplasia, and second-degree heart block. They also shared distinct facial features and similar appearance of their hands and feet. Childhood-onset insulin-dependent diabetes mellitus deve...

Journal: :Turk patoloji dergisi 2017
Amandeep Kaur Vijayalaxmi Suranagi Kamal Patil Hema Bannur

The Neu-Laxova syndrome is a rare autosomal recessive condition associated with neuro-ectodermal abnormalities and other patterns of severe malformations leading to prenatal or early postnatal lethality. Association with kyphosis is an extremely rare finding. A fetus born from a 25-year-old gravida at 30 weeks gestation was diagnosed with Dandy Walker syndrome with severe intrauterine growth re...

Journal: :Journal of medical genetics 1998
A S Teebi S Miller H Ostrer P Eydoux C Colomb-Brockmann K Oudjhane G Watters

Two female sibs of first cousin Iranian parents were found to have the syndrome of spastic paraplegia, optic atrophy with poor vision, microcephaly, and normal cognitive development. Karyotype analysis showed a normal female constitution in one and a male constitution (46,XY) in the other. The XY female showed normal female external genitalia, normal uterus and tubes, and streak gonads. SRY gen...

Journal: :Archives of disease in childhood 1993
R D Milner K A Khallouf R Gibson A Hajianpour C G Mathew

A family in which three siblings born to related parents all manifested clinical abnormalities characteristic of Fanconi's anaemia (microcephaly, short stature, slow growth, beak nose, micrognathia, skin dyspigmentation and forearm and thumb dysplasia in 2/3) is reported. All five family members had normal spontaneous chromosome breakage, a normal response to diepoxybutane and mitomycin C, and ...

2018
Christian G. Bouwkamp Zaid Afawi Aviva Fattal-Valevski Inge E. Krabbendam Stefano Rivetti Rafik Masalha Marialuisa Quadri Guido J. Breedveld Hanna Mandel Muhammad Abu Tailakh H. Berna Beverloo Giovanni Stevanin Alexis Brice Wilfred F.J. van IJcken Meike W. Vernooij Amalia M. Dolga Femke M.S. de Vrij Vincenzo Bonifati Steven A. Kushner

Objective To identify the clinical characteristics and genetic etiology of a family affected with hereditary spastic paraplegia (HSP). Methods Clinical, genetic, and functional analyses involving genome-wide linkage coupled to whole-exome sequencing in a consanguineous family with complicated HSP. Results A homozygous missense mutation was identified in the ACO2 gene (c.1240T>G p.Phe414Val)...

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