University of Wisconsin–Madison Medical College of Wisconsin

The University of Wisconsin Undiagnosed Disease Program: Unveiling Rare Neurodevelopmental Disorders in Exome-Negative Patients

Jadin M. Heilmann; April L. Hall, PhD, MS, CGC; Janet M. Legare, MD; Gregory M. Rice, MD; Kristin A. Seaborg, MD; Derek M. Pavelec, PhD; Xiangqiang Shao, PhD; Vanessa Horner, PhD; M. Stephen Meyn, MD, PhD; Bryn D. Webb, MD

WMJ. 2024;123(6):619-624.

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ABSTRACT

Introduction: The University of Wisconsin Undiagnosed Disease Program employs a “beyond the exome” approach to diagnose rare disease patients.

Case Presentations: We present 2 cases of rare neurodevelopmental disorders identified by whole genome sequencing. The first is a 12-year-old boy with global developmental delay/intellectual disability (GDD/ID) and congenital hypotonia who was diagnosed with CAPZA2-related disorder. The second is a 13-year-old boy with microcephaly, GDD/ID, and seizures who was diagnosed with neurodevelopmental disorder with language delay and behavioral abnormalities, with or without seizures (NEDLAS).

Discussion: Our use of whole genome sequencing identified the fifth reported case of CAPZA2-related neurodevelopmental disorder. Fewer than 40 patients have been reported with NEDLAS, and we identified the fourth patient with the AGO1 in-frame deletion p.Glu376del.

Conclusions: Whole genome sequencing can be effective in diagnosing patients with suspected genetic disorders despite negative standard of care clinical genetic testing and enables the practice of precision medicine.


Author Affiliations: Center for Human Genomics and Precision Medicine, University of Wisconsin School of Medicine and Public Health (UW SMPH); Madison, Wisconsin (Heilmann, Hall, Meyn, Webb); Department of Pediatrics, UW SMPH, Madison, Wisconsin (Hall, Legare, Rice, Shao, Meyn, Webb); Gunderson Health System, LaCrosse Clinic, LaCrosse, Wisconsin (Rice); Department of Neurology, UW SMPH, Madison, Wisconsin (Seaborg); Biotechnology Center, UW – Madison; Madison, Wisconsin (Pavelec); Wisconsin State Laboratory of Hygiene; Madison, Wisconsin (Shao, Horner); Department of Pathology, UW SMPH; Madison, Wisconsin (Horner).
Corresponding Author: Bryn D. Webb, MD, 1111 Highland Ave, Office 2792, Madison, WI 53705; phone 608.262.2559; email bdwebb@wisc.edu; ORCID ID 0000-0001-6174-4677
Funding/Support: None declared.
Financial Disclosures: None declared.
Acknowledgements: The authors extend their gratitude to the patients and their families who participated in this study.
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