Kumble Smitha, Levy Amanda M, Punetha Jaya, Gao Hua, Ah Mew Nicholas, Anyane-Yeboa Kwame, Benke Paul J, Berger Sara M, Bjerglund Lise, Campos-Xavier Belinda, Ciliberto Michael, Cohen Julie S, Comi Anne M, Curry Cynthia, Damaj Lena, Denommé-Pichon Anne-Sophie, Emrick Lisa, Faivre Laurence, Fasano Mary Beth, Fiévet Alice, Finkel Richard S, García-Miñaúr Sixto, Gerard Amanda, Gomez-Puertas Paulino, Guillen Sacoto Maria J, Hoffman Trevor L, Howard Lillian, Iglesias Alejandro D, Izumi Kosuke, Larson Austin, Leiber Anja, Lozano Reymundo, Marcos-Alcalde Iñigo, Mintz Cassie S, Mullegama Sureni V, Møller Rikke S, Odent Sylvie, Oppermann Henry, Ostergaard Elsebet, Pacio-Míguez Marta, Palomares-Bralo Maria, Parikh Sumit, Paulson Anna M, Platzer Konrad, Posey Jennifer E, Potocki Lorraine, Revah-Politi Anya, Rio Marlene, Ritter Alyssa L, Robinson Scott, Rosenfeld Jill A, Santos-Simarro Fernando, Sousa Sérgio B, Wéber Mathys, Xie Yili, Chung Wendy K, Brown Natasha J, Tümer Zeynep
Victorian Clinical Genetics Services, Murdoch Children's Research Institute, Melbourne, Australia.
Department of Clinical Genetics, Kennedy Center, Copenhagen University Hospital, Rigshospitalet, Copenhagen, Denmark.
Hum Mutat. 2022 Feb;43(2):266-282. doi: 10.1002/humu.24308. Epub 2021 Dec 11.
De novo variants in QRICH1 (Glutamine-rich protein 1) has recently been reported in 11 individuals with intellectual disability (ID). The function of QRICH1 is largely unknown but it is likely to play a key role in the unfolded response of endoplasmic reticulum stress through transcriptional control of proteostasis. In this study, we present 27 additional individuals and delineate the clinical and molecular spectrum of the individuals (n = 38) with QRICH1 variants. The main clinical features were mild to moderate developmental delay/ID (71%), nonspecific facial dysmorphism (92%) and hypotonia (39%). Additional findings included poor weight gain (29%), short stature (29%), autism spectrum disorder (29%), seizures (24%) and scoliosis (18%). Minor structural brain abnormalities were reported in 52% of the individuals with brain imaging. Truncating or splice variants were found in 28 individuals and 10 had missense variants. Four variants were inherited from mildly affected parents. This study confirms that heterozygous QRICH1 variants cause a neurodevelopmental disorder including short stature and expands the phenotypic spectrum to include poor weight gain, scoliosis, hypotonia, minor structural brain anomalies, and seizures. Inherited variants from mildly affected parents are reported for the first time, suggesting variable expressivity.
最近有报道称,11名智力残疾(ID)患者存在QRICH1(富含谷氨酰胺蛋白1)的新生变异。QRICH1的功能在很大程度上尚不清楚,但它可能通过蛋白质稳态的转录控制在内质网应激的未折叠反应中发挥关键作用。在本研究中,我们又纳入了27名患者,并描述了携带QRICH1变异的38名患者的临床和分子谱系。主要临床特征为轻度至中度发育迟缓/智力残疾(71%)、非特异性面部畸形(92%)和肌张力减退(39%)。其他发现包括体重增加缓慢(29%)、身材矮小(29%)、自闭症谱系障碍(29%)、癫痫发作(24%)和脊柱侧弯(18%)。52%进行脑成像检查的患者报告有轻微的脑结构异常。28名患者发现有截短或剪接变异,10名有错义变异。4个变异是从症状较轻的父母那里遗传而来的。本研究证实,杂合QRICH1变异会导致包括身材矮小在内的神经发育障碍,并将表型谱扩展至包括体重增加缓慢、脊柱侧弯、肌张力减退、轻微脑结构异常和癫痫发作。首次报告了从症状较轻的父母那里遗传而来的变异,提示存在可变表达。