Multi-Locus Molecular Findings in Pediatric-Onset Inherited Metabolic Disorders: An Eight-Year Series of Outsourced Genomic Testing in Taiwan
Background/Objectives: Outsourced genomic testing can identify additional findings in patients with inherited metabolic disorders (IEMs), including lysosomal storage diseases. We examined these findings in a selected Taiwanese series. Methods: We retrospectively reviewed 13 patients referred during 2018–2025 with a reported or suspected metabolic diagnosis. Whole-exome sequencing (WES) was initially used mainly after inconclusive testing or for incompletely explained phenotypes and was used more broadly later. The original registry also records whole-genome sequencing (WGS) for two included patients. We distinguished reported gene-level findings from diagnoses supported by pathogenic or likely pathogenic (P/LP) variants at each locus. Results: The 13 patients had 18 reported gene-level findings comprising 27 variants. Five met the inclusive multi-locus definition (38.5%; exact 95% confidence interval, 13.9–68.4%). The proportion was 30.8% after excluding candidate gene–disease associations and 7.7% when P/LP variants in the expected zygosity were required at both loci using the later case-summary classifications. Several classifications differed between the original registry and later summaries. These estimates are not directly comparable with the published 4.9% benchmark because ascertainment and diagnostic definitions differ. Enzyme replacement therapy was documented in three patients, including one treated before WES. Conclusions: Unexplained features may warrant evaluation for an additional diagnosis. The selected sample and differing levels of diagnostic certainty limit estimates of confirmed multi-locus disease.
Authors
- Hsiang‐Yu Lin (ORCID: https://orcid.org/0000-0002-9619-0990)
- Yuan-Rong Tu
- Chung‐Lin Lee (ORCID: https://orcid.org/0000-0001-8178-6938)
- Chih‐Kuang Chuang (ORCID: https://orcid.org/0000-0001-9776-2092)
- Huei‐Ching Chiu
- Yun‐Ting Lo
- Huang-Ying Huang
- Jun-Yi Wu
- Shuan-Pei Lin
- Ya-Hui Chang
Institutions
- Fu Jen Catholic University (TW)
- National Yang Ming Chiao Tung University (TW)
- National Taipei University of Nursing and Health Science (TW)
- Mackay Memorial Hospital (TW)
- Mackay Medical University (TW)
- China Medical University Hospital (TW)
- Mackay Junior College of Medicine, Nursing and Management (TW)
Publication Details
- Journal
- Diagnostics
- Published
- 2026-10-15
- DOI
- https://doi.org/10.3390/diagnostics16203356
- Primary Topic
- Metabolism and Genetic Disorders
- Type
- article
- Field-Weighted Citation Impact
- 0.00