Co-occurrence of Jalili syndrome and muscular overgrowth

被引:7
作者
Wawrocka, Anna [1 ]
Walczak-Sztulpa, Joanna [1 ]
Badura-Stronka, Magdalena [1 ,2 ]
Owecki, Michal [3 ]
Kopczynski, Przemyslaw [4 ]
Mrukwa-Kominek, Ewa [5 ]
Skorczyk-Werner, Anna [1 ]
Gasperowicz, Piotr [6 ,7 ]
Ploski, Rafal [6 ]
Krawczynski, Maciej R. [1 ,2 ]
机构
[1] Poznan Univ Med Sci, Dept Med Genet, Rokietnicka 8, PL-60806 Poznan, Poland
[2] Ctr Med Genet GENESIS, Poznan, Poland
[3] Poznan Univ Med Sci, Dept Hist Med Sci, Poznan, Poland
[4] Poznan Univ Med Sci, Dept Maxillofacial Orthopaed & Orthodont, Poznan, Poland
[5] Med Univ Silesia, Dept Ophthalmol, Katowice, Poland
[6] Warsaw Med Univ, Dept Med Genet, Warsaw, Poland
[7] Postgrad Sch Mol Med, Warsaw, Poland
关键词
amelogenesis imperfecta; CNNM4; gene; cone-rod dystrophy; Jalili syndrome; muscle hyperplasia; AMELOGENESIS IMPERFECTA;
D O I
10.1002/ajmg.a.38318
中图分类号
Q3 [遗传学];
学科分类号
071007 ; 090102 ;
摘要
Jalili syndrome is a rare disorder inherited in an autosomal recessive pattern manifesting as a combination of cone-rod dystrophy including progressive loss of visual acuity, color blindness, photophobia, and amelogenesis imperfecta with hypoplastic, immature, or hypocalcified dental enamel. It is caused by mutations in CNNM4, which encodes the ancient conserved domain protein 4. Here we report three brothers with Jalili syndrome and muscle overgrowth of the legs. Myopathic changes were found in needle electromyography. Mutational analysis showed in all three brothers a novel likely pathogenic homozygous missense substitution in exon 1 (c.1076T>C, p.(Leu359Pro)) of CNNM4. Both parents were carriers for the variant. In order to exclude other causative variants that could modify the patients' phenotype we performed exome sequencing and MLPA analysis of the DMD gene in Patient 1. These analyses did not identify any additional variants. Our results expand the mutational spectrum associated with Jalili syndrome and suggest that mild myopathy with muscle overgrowth of the legs could be a newly identified manifestation of the disorder.
引用
收藏
页码:2280 / 2283
页数:4
相关论文
共 8 条
[1]   Amelogenesis imperfecta: a classification and catalogue for the 21st century [J].
Aldred, MJ ;
Savarirayan, R ;
Crawford, PJM .
ORAL DISEASES, 2003, 9 (01) :19-23
[2]   Cone rod dystrophies [J].
Hamel, Christian P. .
ORPHANET JOURNAL OF RARE DISEASES, 2007, 2 (1)
[3]   A PROGRESSIVE CONE-ROD DYSTROPHY AND AMELOGENESIS IMPERFECTA - A NEW SYNDROME [J].
JALILI, IK ;
SMITH, NJD .
JOURNAL OF MEDICAL GENETICS, 1988, 25 (11) :738-740
[4]   The Human Phenotype Ontology in 2017 [J].
Koehler, Sebastian ;
Vasilevsky, Nicole A. ;
Engelstad, Mark ;
Foster, Erin ;
McMurry, Julie ;
Ayme, Segolene ;
Baynam, Gareth ;
Bello, Susan M. ;
Boerkoel, Cornelius F. ;
Boycott, Kym M. ;
Brudno, Michael ;
Buske, Orion J. ;
Chinnery, Patrick F. ;
Cipriani, Valentina ;
Connell, Laureen E. ;
Dawkins, Hugh J. S. ;
DeMare, Laura E. ;
Devereau, Andrew D. ;
de Vries, Bert B. A. ;
Firth, Helen V. ;
Freson, Kathleen ;
Greene, Daniel ;
Hamosh, Ada ;
Helbig, Ingo ;
Hum, Courtney ;
Jahn, Johaenna A. ;
James, Roger ;
Krause, Roland ;
Laulederkind, Stanley J. F. ;
Lochmuller, Hanns ;
Lyon, Gholson J. ;
Ogishima, Soichi ;
Olry, Annie ;
Ouwehand, Willem H. ;
Pontikos, Nikolas ;
Rath, Ana ;
Schaefer, Franz ;
Scott, Richard H. ;
Segal, Michael ;
Sergouniotis, Panagiotis I. ;
Sever, Richard ;
Smith, Cynthia L. ;
Straub, Volker ;
Thompson, Rachel ;
Turner, Catherine ;
Turro, Ernest ;
Veltman, Marijcke W. M. ;
Vulliamy, Tom ;
Yu, Jing ;
von Ziegenweidt, Julie .
NUCLEIC ACIDS RESEARCH, 2017, 45 (D1) :D865-D876
[5]  
Meyer TE, 2010, PLOS GENET, V5, P6
[6]   Does p. Q247X in TRIM63 Cause Human Hypertrophic Cardiomyopathy? [J].
Ploski, Rafal ;
Pollak, Agnieszka ;
Mueller, Sonja ;
Franaszczyk, Maria ;
Michalak, Ewa ;
Kosinska, Joanna ;
Stawinski, Piotr ;
Spiewak, Mateusz ;
Seggewiss, Hubert ;
Bilinska, Zofia T. .
CIRCULATION RESEARCH, 2014, 114 (02) :E2-E5
[7]  
Purwar P., 2015, ORAL SURG ORAL MED O, V120, P210
[8]   A new familial case of Jalili syndrome caused by a novel mutation in CNNM4 [J].
Topcu, Vehap ;
Alp, Muhammed Yunus ;
Alp, Cemile Kedici ;
Bakir, Abdullatif ;
Geylan, Dilay ;
Yilmazoglu, Meltem Ozgul .
OPHTHALMIC GENETICS, 2017, 38 (02) :161-166