Literature DB >> 27389245

Megalencephalic leukoencephalopathy with cysts in twelve Egyptian patients: novel mutations in MLC1 and HEPACAM and a founder effect.

Ghada M H Abdel-Salam1,2, Mohamed S Abdel-Hamid3,4, Samira I Ismail5,3, Heba Hosny6, Tarek Omar7, Laila Effat3,4, Mona S Aglan5,3, Samia A Temtamy5,3, Maha S Zaki5,3.   

Abstract

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.

Entities:  

Keywords:  Founder effect; HEPACAM; Intrafamilial variability; Leukoencephalopathy; MLC1; Megalencephaly; Portwine facial hemangioma with facial hemihypertrophy; Subcortical cysts

Mesh:

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Year:  2016        PMID: 27389245     DOI: 10.1007/s11011-016-9861-7

Source DB:  PubMed          Journal:  Metab Brain Dis        ISSN: 0885-7490            Impact factor:   3.584


  18 in total

1.  Molecular genetic studies in Indian patients with megalencephalic leukoencephalopathy.

Authors:  Pallavi Shukla; Neerja Gupta; Manju Ghosh; Suman Vasisht; Sheffali Gulati; Prahlad Balakrishnan; Raju Sharma; Arun K Gupta; Mahesh Kamate; Veena Kalra; Madhulika Kabra
Journal:  Pediatr Neurol       Date:  2011-06       Impact factor: 3.372

2.  Megalencephalic leukoencephalopathy with subcortical cysts caused by compound heterozygous mutations in MLC1, in patients with and without subcortical cysts in the brain.

Authors:  Teruaki Masuda; Mitsuharu Ueda; Hidetsugu Ueyama; Shino Shimada; Masatoshi Ishizaki; Shigehiro Imamura; Toshiyuki Yamamoto; Yukio Ando
Journal:  J Neurol Sci       Date:  2015-03-12       Impact factor: 3.181

3.  An unusually mild presentation of megalencephalic leukoencephalopathy with subcortical cysts.

Authors:  Gulsen Kocaman; Gozde Eryigit; Truus E M Abbink; Rukiye Kılıcarslan; Talip Asil; Alpay Alkan; Marjo S Van der Knaap; Abdulkadir Kocer
Journal:  Clin Neurol Neurosurg       Date:  2013-02-26       Impact factor: 1.876

4.  A Japanese adult case of megalencephalic leukoencephalopathy with subcortical cysts with a good long-term prognosis.

Authors:  Shingo Koyama; Toru Kawanami; Shigeki Arawaka; Manabu Wada; Takeo Kato
Journal:  Intern Med       Date:  2012-03-01       Impact factor: 1.271

5.  Molecular mechanisms of MLC1 and GLIALCAM mutations in megalencephalic leukoencephalopathy with subcortical cysts.

Authors:  Tania López-Hernández; Sònia Sirisi; Xavier Capdevila-Nortes; Marisol Montolio; Victor Fernández-Dueñas; Gert C Scheper; Marjo S van der Knaap; Pilar Casquero; Francisco Ciruela; Isidre Ferrer; Virginia Nunes; Raúl Estévez
Journal:  Hum Mol Genet       Date:  2011-05-30       Impact factor: 6.150

6.  Clinical, neuroimaging, and genetic characteristics of megalencephalic leukoencephalopathy with subcortical cysts in Egyptian patients.

Authors:  Iman G Mahmoud; Marwa Mahmoud; Miral Refaat; Marian Girgis; Nevin Waked; Ameera El Badawy; Laila Selim; Sawsan Hassan; Alice K Abdel Aleem
Journal:  Pediatr Neurol       Date:  2013-10-24       Impact factor: 3.372

7.  Genetic heterogeneity of megalencephalic leukoencephalopathy and subcortical cysts.

Authors:  C Patrono; G Di Giacinto; E Eymard-Pierre; F M Santorelli; D Rodriguez; N De Stefano; A Federico; R Gatti; V Benigno; A Megarbané; B Tabarki; O Boespflug-Tanguy; E Bertini
Journal:  Neurology       Date:  2003-08-26       Impact factor: 9.910

8.  Leukoencephalopathy with swelling and a discrepantly mild clinical course in eight children.

Authors:  M S van der Knaap; P G Barth; H Stroink; O van Nieuwenhuizen; W F Arts; F Hoogenraad; J Valk
Journal:  Ann Neurol       Date:  1995-03       Impact factor: 10.422

9.  Megalencephalic leukoencephalopathy with subcortical cysts; a founder effect in Israeli patients and a higher than expected carrier rate among Libyan Jews.

Authors:  Bruria Ben-Zeev; Etgar Levy-Nissenbaum; Hadas Lahat; Yair Anikster; Yael Shinar; Nathan Brand; Varda Gross-Tzur; Daune MacGregor; Roy Sidi; Robert Kleta; Moshe Frydman; Elon Pras
Journal:  Hum Genet       Date:  2002-07-16       Impact factor: 4.132

10.  Megalencephalic leukoencephalopathy with subcortical cysts protein-1 regulates epidermal growth factor receptor signaling in astrocytes.

Authors:  Angela Lanciotti; Maria Stefania Brignone; Sergio Visentin; Chiara De Nuccio; Luigi Catacuzzeno; Cinzia Mallozzi; Stefania Petrini; Martino Caramia; Caterina Veroni; Gaetana Minnone; Antonietta Bernardo; Fabio Franciolini; Mauro Pessia; Enrico Bertini; Tamara Corinna Petrucci; Elena Ambrosini
Journal:  Hum Mol Genet       Date:  2016-02-09       Impact factor: 6.150

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  2 in total

1.  The use of targeted genomic capture and massively parallel sequencing in diagnosis of Chinese Leukoencephalopathies.

Authors:  Xiaole Wang; Fang He; Fei Yin; Chao Chen; Liwen Wu; Lifen Yang; Jing Peng
Journal:  Sci Rep       Date:  2016-10-25       Impact factor: 4.379

Review 2.  Astrocyte-Oligodendrocyte-Microglia Crosstalk in Astrocytopathies.

Authors:  Dieuwke Maria de Waard; Marianna Bugiani
Journal:  Front Cell Neurosci       Date:  2020-11-19       Impact factor: 5.505

  2 in total

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