Riffe R Madison, Downes Gerald B
Neuroscience and Behavior Graduate Program, University of Massachusetts Amherst, Amherst, MA, 01003, USA.
Biology Department, University of Massachusetts Amherst, Amherst, MA, 01003, USA.
Biol Open. 2025 Mar 15;14(3). doi: 10.1242/bio.061808. Epub 2025 Mar 10.
The five-subunit endosomal Rab5 and RNA/ribose intermediary (FERRY) complex is a newly described protein complex consisting of TBCK, PPP1R21, FERRY3 (previously C12orf4), CRYZL1, and GATD1. The FERRY complex is proposed to function as a Rab5 effector to shuttle mRNA to the cell periphery for local translation, a process especially important in cells with far reaching processes. Interestingly, three members of the FERRY complex are associated with ultra-rare neurogenetic disorders. Mutation of TBCK causes TBCK syndrome, mutation of PPP1R21 is associated with PPP1R21-related intellectual disability, and mutation of FERRY3 results in an autosomal recessive intellectual disability. Neurologic disorders have yet to be associated with mutation of GATD1 or CRYZL1. Here, we provide a review of each FERRY complex-related neurologic disorder and draw clinical comparisons between the disease states. We also discuss data from the current cellular and animal models available to study these disorders, which is notably disparate and scattered across different cell types and systems. Taken together, we explore the possibility that these three diseases may represent one shared disease class, which could be further understood by combining and comparing known information about each individual disease. If true, this could have substantial implications on our understanding of the cellular role of the FERRY complex and on treatment strategies for affected individuals, allowing researchers, clinicians, and patient organizations to maximize the utility of research efforts and resources to support patients with these disorders.
由五个亚基组成的内体Rab5与RNA/核糖中介体(FERRY)复合物是一种新描述的蛋白质复合物,由TBCK、PPP1R21、FERRY3(以前称为C12orf4)、CRYZL1和GATD1组成。FERRY复合物被认为作为一种Rab5效应物,将mRNA转运到细胞周边进行局部翻译,这一过程在具有长距离延伸过程的细胞中尤为重要。有趣的是,FERRY复合物的三个成员与极罕见的神经遗传性疾病相关。TBCK突变导致TBCK综合征,PPP1R21突变与PPP1R21相关的智力残疾有关,FERRY3突变导致常染色体隐性智力残疾。神经系统疾病尚未与GATD1或CRYZL1突变相关联。在这里,我们对每种与FERRY复合物相关的神经系统疾病进行综述,并对疾病状态进行临床比较。我们还讨论了目前可用于研究这些疾病的细胞和动物模型的数据,这些数据明显分散且分布在不同的细胞类型和系统中。综上所述,我们探讨了这三种疾病可能代表一种共同疾病类型的可能性,通过结合和比较关于每种个体疾病的已知信息可以进一步理解这一点。如果属实,这可能对我们理解FERRY复合物的细胞作用以及对受影响个体的治疗策略产生重大影响,使研究人员、临床医生和患者组织能够最大限度地利用研究工作和资源来支持患有这些疾病的患者。