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.

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Journal
Diagnostics
Published
2026-10-15
DOI
https://doi.org/10.3390/diagnostics16203356
Primary Topic
Metabolism and Genetic Disorders
Type
article
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article

Multi-Locus Molecular Findings in Pediatric-Onset Inherited Metabolic Disorders: An Eight-Year Series of Outsourced Genomic Testing in Taiwan

Hsiang‐Yu Lin, Yuan-Rong Tu, Chung‐Lin Lee, Chih‐Kuang Chuang et al.
Diagnostics
Metabolism and Genetic Disorders
article

Multi-Locus Molecular Findings in Pediatric-Onset Inherited Metabolic Disorders: An Eight-Year Series of Outsourced Genomic Testing in Taiwan

Hsiang‐Yu Lin, Yuan-Rong Tu, Chung‐Lin Lee, Chih‐Kuang Chuang, Huei‐Ching Chiu, Yun‐Ting Lo, Huang-Ying Huang, Jun-Yi Wu, Shuan-Pei Lin, Ya-Hui Chang
article en

Abstract

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.

DiagnosticsVol. 16(20)
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)
Good health and well-being
Openalex Percentile: Top 15%
Metabolism and Genetic Disorders
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