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Megalencephalic leukoencephalopathy with cysts in twelve Egyptian patients: novel mutations in MLC1 and HEPACAM and a founder effect.
Abdel-Salam, Ghada M H; Abdel-Hamid, Mohamed S; Ismail, Samira I; Hosny, Heba; Omar, Tarek; Effat, Laila; Aglan, Mona S; Temtamy, Samia A; Zaki, Maha S.
Affiliation
  • Abdel-Salam GM; Clinical Genetics Department, Human Genetics and Genome Research Division, National Research Centre, Tahrir street, Dokki, Cairo, Egypt. ghada.abdelsalam@yahoo.com.
  • Abdel-Hamid MS; Centre of Excellence for Human Genetics, National Research Centre, Cairo, Egypt. ghada.abdelsalam@yahoo.com.
  • Ismail SI; Centre of Excellence for Human Genetics, National Research Centre, Cairo, Egypt.
  • Hosny H; Medical Molecular Genetics Department, Human Genetics and Genome Research Division, National Research Centre, Cairo, Egypt.
  • Omar T; Clinical Genetics Department, Human Genetics and Genome Research Division, National Research Centre, Tahrir street, Dokki, Cairo, Egypt.
  • Effat L; Centre of Excellence for Human Genetics, National Research Centre, Cairo, Egypt.
  • Aglan MS; National Institute of Neuromotor system, Cairo, Egypt.
  • Temtamy SA; Department of Pediatrics, Alexandria University, Alexandria, Egypt.
  • Zaki MS; Centre of Excellence for Human Genetics, National Research Centre, Cairo, Egypt.
Metab Brain Dis ; 31(5): 1171-9, 2016 10.
Article in En | MEDLINE | ID: mdl-27389245
Two genes causing megalencephalic leukoencephalopathy with subcortical cysts (MLC) have been discovered so far. Here, we identified MLC1 and HEPACAM mutations in ten and two patients, respectively. The molecular results included an unreported inframe duplication mutation (c.929_930dupCTGCTG; p.L309dup) of MLC1 and a novel missense mutation c.293G>A (p.R98H) of HEPACAM. Further, the previously reported missense (c.278C>T; p.S93L) and the deletion/insertion (c.908_918delinsGCA; p.V303Gfs*96) were found in one and 8 patients (75 %), respectively. The 8 patients carrying the p.V303Gfs*96 shared a similar haplotype suggesting a founder effect. All mutations were in the homozygous state proving the autosomal recessive mode of inheritance. The core phenotype of macrocephaly, subcortical cysts and white matter appeared homogeneous although the patients differed in the onset, clinical course, disease severity and brain imaging findings. Our study expands the spectrum of mutations in MLC1 and HEPACAM and supports the genetic and clinical heterogeneity. Further, It confirms c.908_918delinsGCA (p.V303Gfs*96) as a founder mutation among Egyptian patients. This finding will contribute to provide targeted testing for this mutation in MLC patients in our population.
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Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Proteins / Founder Effect / Hereditary Central Nervous System Demyelinating Diseases / Cysts / Membrane Proteins / Mutation Type of study: Etiology_studies / Incidence_studies / Observational_studies / Prognostic_studies / Risk_factors_studies Limits: Adolescent / Adult / Child / Child, preschool / Female / Humans / Infant / Male Country/Region as subject: Africa Language: En Journal: Metab Brain Dis Journal subject: CEREBRO / METABOLISMO Year: 2016 Type: Article Affiliation country: Egypt

Full text: 1 Collection: 01-internacional Database: MEDLINE Main subject: Proteins / Founder Effect / Hereditary Central Nervous System Demyelinating Diseases / Cysts / Membrane Proteins / Mutation Type of study: Etiology_studies / Incidence_studies / Observational_studies / Prognostic_studies / Risk_factors_studies Limits: Adolescent / Adult / Child / Child, preschool / Female / Humans / Infant / Male Country/Region as subject: Africa Language: En Journal: Metab Brain Dis Journal subject: CEREBRO / METABOLISMO Year: 2016 Type: Article Affiliation country: Egypt