Genetic Heterogeneity Underlying Familial Short Stature

<b>Background and Clinical Significance:</b> Familial short stature is a common reason for referral in clinical genetics. While often attributed to a single genetic cause, genetic heterogeneity can complicate diagnosis and management. This report describes a family in which three distinct pathogenic variants in <i>SHOX</i>, <i>PDE4D</i> and <i>ACAN</i> caused overlapping phenotypes of familial short stature. <b>Case Presentation:</b> Clinical, radiological and molecular data were collected retrospectively at the Reference Centre for Constitutional Bone Diseases at Montpellier University Hospital. Targeted gene panels, whole genome sequencing and Sanger sequencing were employed to identify pathogenic variants. Variant interpretation followed the guidelines of the American College of Medical Genetics. A pathogenic <i>SHOX</i> variant (c.452G>A; p.Ser151Asn) was identified in the proband and her mother, which is consistent with dyschondrosteosis. A de novo <i>PDE4D</i> variant (c.671C>T; p.Thr224Ile) was identified in a cousin presenting with syndromic acrodysostosis. An <i>ACAN</i> splice variant (c.6833-1G>A) was detected in several family members and is associated with short stature and skeletal anomalies. An individual carrying both the <i>SHOX</i> and <i>ACAN</i> variants exhibited a more severe phenotype, suggesting an additive effect. <b>Conclusions:</b> This case study highlights the importance of systematic molecular investigations in families with overlapping yet heterogeneous phenotypes. Comprehensive genetic familial analysis enables personalized care and accurate genetic counselling, particularly when multiple diagnoses coexist. A family history should not preclude molecular testing, since similar phenotypes can result from different genetic causes.

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Journal
Diagnostics
Published
2025-12-09
DOI
https://doi.org/10.3390/diagnostics15243127
Primary Topic
Genomics and Rare Diseases
Type
article
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article

Genetic Heterogeneity Underlying Familial Short Stature

O. Prodhomme, Margot Comel, Mouna Barat‐Houari, Sophie Rondeau et al.
Diagnostics
Genomics and Rare Diseases
article

Genetic Heterogeneity Underlying Familial Short Stature

O. Prodhomme, Margot Comel, Mouna Barat‐Houari, Sophie Rondeau, Yves‐Marie Pers, Marjolaine Willems, Cyril Amouroux, Constance Wells, David Geneviève, Fanny Alkar, Nathalie Ruiz
article en

Abstract

<b>Background and Clinical Significance:</b> Familial short stature is a common reason for referral in clinical genetics. While often attributed to a single genetic cause, genetic heterogeneity can complicate diagnosis and management. This report describes a family in which three distinct pathogenic variants in <i>SHOX</i>, <i>PDE4D</i> and <i>ACAN</i> caused overlapping phenotypes of familial short stature. <b>Case Presentation:</b> Clinical, radiological and molecular data were collected retrospectively at the Reference Centre for Constitutional Bone Diseases at Montpellier University Hospital. Targeted gene panels, whole genome sequencing and Sanger sequencing were employed to identify pathogenic variants. Variant interpretation followed the guidelines of the American College of Medical Genetics. A pathogenic <i>SHOX</i> variant (c.452G>A; p.Ser151Asn) was identified in the proband and her mother, which is consistent with dyschondrosteosis. A de novo <i>PDE4D</i> variant (c.671C>T; p.Thr224Ile) was identified in a cousin presenting with syndromic acrodysostosis. An <i>ACAN</i> splice variant (c.6833-1G>A) was detected in several family members and is associated with short stature and skeletal anomalies. An individual carrying both the <i>SHOX</i> and <i>ACAN</i> variants exhibited a more severe phenotype, suggesting an additive effect. <b>Conclusions:</b> This case study highlights the importance of systematic molecular investigations in families with overlapping yet heterogeneous phenotypes. Comprehensive genetic familial analysis enables personalized care and accurate genetic counselling, particularly when multiple diagnoses coexist. A family history should not preclude molecular testing, since similar phenotypes can result from different genetic causes.

DiagnosticsVol. 15(24)
Hôpital Necker-Enfants Malades (FR), Inserm (FR), Université de Montpellier (FR), Centre Hospitalier Universitaire de Montpellier (FR), Institute for Regenerative Medicine & Biotherapy (FR), Assistance Publique – Hôpitaux de Paris (FR), Institut Universitaire de Recherche Clinique (FR), Hôpital Arnaud de Villeneuve (FR), Institut des Maladies Génétiques Imagine (FR)
Openalex Percentile: Top 59%
Genomics and Rare Diseases
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