# Core D - IDD Models Core

> **NIH NIH U54** · UNIVERSITY OF WISCONSIN-MADISON · 2020 · $201,218

## Abstract

Waisman Center investigators study Intellectual and Developmental Disabilities (IDD) through elucidation
of molecular pathways that are altered in genetic and epigenetic conditions affecting nervous system function.
Compared with other genetically tractable model organisms, rodents afford the highest similarity to humans in
anatomy, physiology, and genetics. Thus, study of IDD has relied on development of rodent models of various
disorders. In recent years, complementary development of human stem cell-based models to study similar
processes has greatly expanded the scope and relevance of studies of IDD. Investigators of the Waisman
Center have spearheaded research in a number of IDD conditions, and many investigators rely on
complementary mouse and human stem cell models to study different facets of these disorders. Use of these
models provides the means to evaluate candidate therapeutics, as well as develop novel screens to identify
promising compounds. The resources of the IDD Models Core, almost all housed within the Waisman Center
itself, provide the tools for development of mouse and human stem-cell derived models of IDD. Therefore, we
propose two specific aims for the next project period. Aim 1 is to create and analyze rodent models of IDD.
The objectives of this aim are to: 1) provide advanced technical services in the generation, cryopreservation,
and maintenance of mutant or genetically engineered strains of mice and rats; 2) provide advanced behavioral
testing services and facilities for the phenotypic characterization of novel strains of rodents; 3) provide training
and consultation to investigators and laboratory personnel; and 4) facilitate maintenance and exchange of
unique rodent models between institutions. Aim 2 is to to develop human stem cell models of IDD and facilitate
phenotyping of neurodevelopmental defects. The objectives of this aim are to: (1) support the generation of
induced pluripotent stem cell (iPSC) lines directly from individuals diagnosed with IDD; (2) support
development of IDD-specific PSC lines through genome editing; (3) provide high quality, cost effective, cellular
and molecular neuroscience technology and expertise to IDDRC investigators to facilitate phenotyping of
neurodevelopmental defects in human PSC derived cells and (4) provide training and technical support in
human stem cell culture, phenotyping and gene editing to promote usage of new approaches by our
investigators. These initiatives are further served by state-of-the-art imaging and molecular biology analysis
tools housed within the Waisman Center. With support from the Administrative Core, linkages are provided to
complementary capabilities elsewhere on campus, such as those offered by the UW Biotechnology Center,
UW Comprehensive Cancer Center, and other partnerships. These resources and partnerships are leveraged
to facilitate usage of a large repertoire of techniques to enhance multidimensional analysis of IDD models, and
to promote the de...

## Key facts

- **NIH application ID:** 10005051
- **Project number:** 5U54HD090256-05
- **Recipient organization:** UNIVERSITY OF WISCONSIN-MADISON
- **Principal Investigator:** John P Svaren
- **Activity code:** U54 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2020
- **Award amount:** $201,218
- **Award type:** 5
- **Project period:** — → —

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10005051

## Citation

> US National Institutes of Health, RePORTER application 10005051, Core D - IDD Models Core (5U54HD090256-05). Retrieved via AI Analytics 2026-06-01 from https://api.ai-analytics.org/grant/nih/10005051. Licensed CC0.

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