TY - JOUR
T1 - X-linked elliptocytosis with impaired growth is related to mutated AMMECR1
AU - Basel-Vanagaite, Lina
AU - Pillar, Nir
AU - Isakov, Ofer
AU - Smirin-Yosef, Pola
AU - Lagovsky, Irina
AU - Orenstein, Naama
AU - Salmon-Divon, Mali
AU - Tamary, Hannah
AU - Zaft, Tami
AU - Bazak, Lily
AU - Meyerovitch, Joseph
AU - Pelli, Tal
AU - Botchan, Shay
AU - Farberov, Luba
AU - Weissglas-Volkov, Daphna
AU - Shomron, Noam
N1 - Publisher Copyright:
© 2017
PY - 2017/3/30
Y1 - 2017/3/30
N2 - In this study, we report a family with X-linked recessive syndrome caused by mutated AMMECR1 and characterized by elliptocytosis with or without anemia, midface hypoplasia, proportionate short stature and hearing loss. Recently, mutations in AMMECR1 were reported in two maternal half-brothers, presenting with nephrocalcinosis, midface hypoplasia and, in one of the siblings, deafness and elliptocytosis. AMMECR1 gene is localized in the critical region of contiguous deletion syndrome on Xq22.3 implicated in Alport syndrome, mental retardation, midface hypoplasia, and elliptocytosis (AMME complex). Interestingly, alternative splicing of exon 2, the same exon harboring the truncating mutation, was observed in the proband and in his unaffected mother. Alternative splicing of this exon is predicted to lead to an in-frame deletion. We provide further evidence that mutated AMMECR1 gene is responsible for this clinically recognizable X-linked condition with variable expressivity.
AB - In this study, we report a family with X-linked recessive syndrome caused by mutated AMMECR1 and characterized by elliptocytosis with or without anemia, midface hypoplasia, proportionate short stature and hearing loss. Recently, mutations in AMMECR1 were reported in two maternal half-brothers, presenting with nephrocalcinosis, midface hypoplasia and, in one of the siblings, deafness and elliptocytosis. AMMECR1 gene is localized in the critical region of contiguous deletion syndrome on Xq22.3 implicated in Alport syndrome, mental retardation, midface hypoplasia, and elliptocytosis (AMME complex). Interestingly, alternative splicing of exon 2, the same exon harboring the truncating mutation, was observed in the proband and in his unaffected mother. Alternative splicing of this exon is predicted to lead to an in-frame deletion. We provide further evidence that mutated AMMECR1 gene is responsible for this clinically recognizable X-linked condition with variable expressivity.
KW - AMMECR1 gene
KW - Elliptocytosis
KW - Genomics
KW - X-linked
UR - http://www.scopus.com/inward/record.url?scp=85009949445&partnerID=8YFLogxK
U2 - 10.1016/j.gene.2017.01.001
DO - 10.1016/j.gene.2017.01.001
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C2 - 28089922
AN - SCOPUS:85009949445
SN - 0378-1119
VL - 606
SP - 47
EP - 52
JO - Gene
JF - Gene
ER -