Deletion at an 1q24 locus reveals a critical role of long noncoding RNA DNM3OS in skeletal development

Ting ting Yu, Qiu fan Xu, Si Yang Li, Hui jie Huang, Sarah Dugan, Lei Shao, Jennifer A. Roggenbuck, Xiao tong Liu, Huai ze Liu, Betsy A. Hirsch, Shen Yue, Chen Liu, Steven Y. Cheng

Research output: Contribution to journalArticlepeer-review

Abstract

Background: Skeletal development and maintenance are complex processes known to be coordinated by multiple genetic and epigenetic signaling pathways. However, the role of long non-coding RNAs (lncRNAs), a class of crucial epigenetic regulatory molecules, has been under explored in skeletal biology. Results: Here we report a young patient with short stature, hypothalamic dysfunction and mild macrocephaly, who carries a maternally inherited 690 kb deletion at Chr.1q24.2 encompassing a noncoding RNA gene, DNM3OS, embedded on the opposite strand in an intron of the DYNAMIN 3 (DNM3) gene. We show that lncRNA DNM3OS sustains the proliferation of chondrocytes independent of two co-cistronic microRNAs miR-199a and miR-214. We further show that nerve growth factor (NGF), a known factor of chondrocyte growth, is a key target of DNM3OS-mediated control of chondrocyte proliferation. Conclusions: This work demonstrates that DNM3OS is essential for preventing premature differentiation of chondrocytes required for bone growth through endochondral ossification.

Original languageEnglish (US)
Article number47
JournalCell and Bioscience
Volume11
Issue number1
DOIs
StatePublished - Mar 2 2021

Bibliographical note

Publisher Copyright:
© 2021, The Author(s).

Copyright:
Copyright 2021 Elsevier B.V., All rights reserved.

Keywords

  • 1q24
  • DNM3OS
  • lncRNA
  • Nerve growth factor
  • Skeletal abnormalities

PubMed: MeSH publication types

  • Journal Article

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