Poster Presentation 43rd Lorne Genome Conference 2022

NT2/D1-cas9: a novel in vitro manipulatable model for male sex development (#265)

Brittany Vining 1 2 , Alejandra Reyes 2 , Aleisha Symon 2 , Zhenhua Ming 1 2 , Janelle Ryan 2 , Vincent Harley 1 2
  1. Monash University, Melbourne, Victoria, Australia
  2. Sex Development Laboratory, Hudson Institute of Medical Research, Melbourne, Victoria, Australia

Sex is determined by the onset of SRY expression around week 6 in the human XY embryo.  Differences of Sex Development (DSD) are a spectrum of conditions in which gonadal, chromosomal or anatomical sex is atypical 1. SRY mutations account for 15% of 46,XY DSDs, and rare mutations in about 30 other genes are involved, but the underlying molecular basis in many forms of DSD remains unknown, due in part to the lack of manipulatable models that recapitulate human sex determination.

 

Mouse models using knockout/transgenic approaches have been useful in studying the function of critical sex determination genes. Differences in gene expression thresholds and genetic robustness between humans and mice are becoming apparent2. Limitations include functional redundancy, gene dosage or genetic buffering, such that mutations causing DSD in humans produces no phenotype consequence in mice3.

 

An in vitro model that can be used to model Sertoli cell function in the testis is NT2/D1, a human pluripotent clonal cell line derived from a testicular tumour4. NT2/D1 cells can be used to model a variety of human developmental processes: undifferentiated they can model human Sertoli cell development, and when differentiated they can model neuronal development5 or smooth muscle development6.

 

We have established an NT2/D1-cas9 cell line, and characterised these via a suite of cell phenotyping assays including xCELLigence® RTCA and germ cell maturation assays. This model can be used in conjunction with siRNA knockdown investigations of genes of interest, to assess their individual contributions to characteristics such as cell adhesion, proliferation and capacity to nurture germ cells.

  1. León NY, Reyes AP, Harley VR. A clinical algorithm to diagnose differences of sex development. Lancet Diabetes Endocrinol. 2019 Jul;7(7):560-574. doi: 10.1016/S2213-8587(18)30339-5
  2. Gonen N, Quinn A, O’Neill HC, Koopman P, Lovell-Badge R (2017) Normal Levels of Sox9 Expression in the Developing Mouse Testis Depend on the TES/TESCO Enhancer, but This Does Not Act Alone. PLOS Genetics 13(1): e1006520. https://doi.org/10.1371/journal.pgen.1006520
  3. Barbaric I, Miller G, Dear TN (2017) Appearances can be deceiving: phenotypes of knockout mice. Brief Funct Genomic Proteomic 6(2):91-103. doi: 10.1093/bfgp/elm008.
  4. Knower KC, Kelly S, Ludbrook LM, Bagheri-Fam S, Sim H, et al. (2011) Failure of SOX9 Regulation in 46XY Disorders of Sex Development with SRY, SOX9 and SF1 Mutations. PLOS ONE 6(3): e17751. https://doi.org/10.1371/journal.pone.0017751
  5. Pleasure SJ, Lee VM-J (1993) Ntera 2 cells: a human cell line which displays characteristics expected of a human committed neuronal progenitor cell. J. Neurosci. Res 35(6):585-602
  6. Chadalavada, R, Houldsworth, J, Olshen, A, Bosl, G, Studer, L, Chaganti, R (2005) Transcriptional program of bone morphogenetic protein-2-induced epithelial and smooth muscle differentiation of pluripotent human embryonal carcinoma cells. Funct Integr Genomics 5(2):59-69. doi: 10.1007/s10142-005-0132-7