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Targeted demethylation at the CDKN1C/p57 locus induces human β cell replication
Kristy Ou, … , Dana Avrahami, Klaus H. Kaestner
Kristy Ou, … , Dana Avrahami, Klaus H. Kaestner
Published January 2, 2019; First published October 23, 2018
Citation Information: J Clin Invest. 2019;129(1):209-214. https://doi.org/10.1172/JCI99170.
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Categories: Concise Communication Endocrinology

Targeted demethylation at the CDKN1C/p57 locus induces human β cell replication

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Abstract

The loss of insulin-secreting β cells is characteristic among type I and type II diabetes. Stimulating proliferation to expand sources of β cells for transplantation remains a challenge because adult β cells do not proliferate readily. The cell cycle inhibitor p57 has been shown to control cell division in human β cells. Expression of p57 is regulated by the DNA methylation status of the imprinting control region 2 (ICR2), which is commonly hypomethylated in Beckwith-Wiedemann syndrome patients who exhibit massive β cell proliferation. We hypothesized that targeted demethylation of the ICR2 using a transcription activator–like effector protein fused to the catalytic domain of TET1 (ICR2-TET1) would repress p57 expression and promote cell proliferation. We report here that overexpression of ICR2-TET1 in human fibroblasts reduces p57 expression levels and increases proliferation. Furthermore, human islets overexpressing ICR2-TET1 exhibit repression of p57 with concomitant upregulation of Ki-67 while maintaining glucose-sensing functionality. When transplanted into diabetic, immunodeficient mice, the epigenetically edited islets show increased β cell replication compared with control islets. These findings demonstrate that epigenetic editing is a promising tool for inducing β cell proliferation, which may one day alleviate the scarcity of transplantable β cells for the treatment of diabetes.

Authors

Kristy Ou, Ming Yu, Nicholas G. Moss, Yue J. Wang, Amber W. Wang, Son C. Nguyen, Connie Jiang, Eseye Feleke, Vasumathi Kameswaran, Eric F. Joyce, Ali Naji, Benjamin Glaser, Dana Avrahami, Klaus H. Kaestner

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Figure 4

Targeted epimutation at the ICR2 induces human β cell replication.

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Targeted epimutation at the ICR2 induces human β cell replication.
(A) E...
(A) Expression of CDKN1C mRNA is significantly anticorrelated with mRNA expression levels of the proliferation marker MKI67 in islets transduced with Ad.ICR2-TET1. mRNA levels were normalized to HPRT1. Each data point represents a single donor. (B) Schematic of subcutaneous transplantation of transduced human islets in diabetic NSG mice. For each donor, 100 islets transduced with the ICR2-TET1dead or ICR2-TET1 adenovirus were transplanted in the left or right subcutaneous abdominal incision, respectively. Mice were administered BrdU via the drinking water for 3 weeks before the grafts were harvested. (C) Example of an islet transduced with Ad.ICR2-TET1 with 3 BrdU+ β cells. For each donor and treatment, 800 to 2,000 C-peptide+ cells were counted. Scale bars: 50 μm and 10 μm (insets). (D) Percentage of BrdU+ β cells in sectioned xenografts 3 weeks after transplant. Each pair of data points represents an individual donor. *P < 0.05 by ratio paired t test. (E) BrdU/C-peptide double-positive doublets, indicated by the white arrowheads, in Ad.ICR2-TET1–transduced xenografts. Scale bars: 50 μm.
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ISSN: 0021-9738 (print), 1558-8238 (online)

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