American journal of medical genetics. Part A · 2009
Majewski osteodysplastic primordial dwarfism type II (MOPD II) syndrome previously diagnosed as Seckel syndrome: report of a novel mutation of the PCNT gene.
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Piane Maria, Della Monica Matteo, Piatelli Gianluca, Lulli Patrizia, Lonardo Fortunato, Chessa Luciana, Scarano Gioacchino
Аннотация
We report on a 3-year-old boy with prenatal onset of proportionate dwarfism, postnatal severe microcephaly, high forehead with receded hairline, sparse scalp hair, beaked nose, mild retrognathia and hypotonia diagnosed at birth as Seckel syndrome. At age 3 years, he became paralyzed due to a cerebrovascular malformation. Based on the clinical and radiological features showing evidence of skeletal dysplasia, the diagnosis was revised to Majewski osteodysplastic primordial dwarfism type II (MOPD II) syndrome. Western blot analysis of the patient's lymphoblastoid cell line lysate showed the absence of the protein pericentrin. Subsequent molecular analysis identified a novel homozygous single base insertion (c.1527_1528insA) in exon 10 of the PCNT gene, which leads to a frameshift (Treo510fs) and to premature protein truncation. PCNT mutations must be considered diagnostic of MOPD II syndrome. A possible role of pericentrin in the development of cerebral vessels is suggested.
Предметные рубрики MeSH
AdultAntigensBase SequenceBlotting, WesternBone and BonesBrainCase-Control StudiesChild, PreschoolDNA Mutational AnalysisDiagnosis, DifferentialDwarfismFemaleHumansInfantInfant, NewbornMagnetic Resonance AngiographyMaleMolecular Sequence DataMutationPregnancyRadiographyShort Rib-Polydactyly SyndromeSyndrome