This study aimed to describe and molecularly characterize a rare case of early-onset multiple primary tumors associated with a novel germline protection of telomeres 1 (POT1) gene pathogenic variant, associated with POT1 tumor predisposition syndrome (POT1-TPD), a rare autosomal dominant disorder characterized by an increased risk for various tumors, including gliomas. A custom clinical exome panel for genes associated with Cancer Predisposition Syndromes was performed on DNA extracted from blood. RNA sequencing and genome-wide DNA methylation profiling were conducted on tumors. We report the case of a female patient diagnosed at age 12 with a diffuse glioma harboring a ROS1 fusion. At age 18, she developed a renal cell carcinoma. Genetic germline testing revealed a heterozygous germline POT1 variant, c.910dupG (p.Asp304fs*8), classified as pathogenic. Segregation analysis demonstrated paternal inheritance of the variant. The same POT1 variant was identified in both tumor tissues in a heterozygous state. This is the first reported case of a young adult carrying a new pathogenic POT1 variant who developed a pediatric high-grade glioma and a renal tumor in early adulthood. This report expands the clinical spectrum of POT1-associated tumors with early-onset and underscores the relevance of genetic testing for patient management and family counseling.

Cipri, S., Cacchione, A., Serra, A., Del Baldo, G., Miele, E., Pedace, L., Patrizi, S., D'Antonio, F., Agolini, E., Novelli, A., Crocoli, A., Bertocchini, A., Carai, A., Barresi, S., Rossi, S., Alaggio, R., Diomedi Camassei, F., Colafati, G. S., Boccuto, L., Mastronuzzi, A., Case Report: The revelation of a new pathogenic variant in the POT1 gene in a patient with a pediatric high-grade glioma and a renal cell carcinoma, <<FRONTIERS IN ONCOLOGY>>, 2026; 16 (16): 1-7. [doi:10.3389/fonc.2026.1778785] [https://hdl.handle.net/10807/342243]

Case Report: The revelation of a new pathogenic variant in the POT1 gene in a patient with a pediatric high-grade glioma and a renal cell carcinoma

Carai, Andrea
Writing – Review & Editing
;
Mastronuzzi, Angela
Ultimo
Conceptualization
2026

Abstract

This study aimed to describe and molecularly characterize a rare case of early-onset multiple primary tumors associated with a novel germline protection of telomeres 1 (POT1) gene pathogenic variant, associated with POT1 tumor predisposition syndrome (POT1-TPD), a rare autosomal dominant disorder characterized by an increased risk for various tumors, including gliomas. A custom clinical exome panel for genes associated with Cancer Predisposition Syndromes was performed on DNA extracted from blood. RNA sequencing and genome-wide DNA methylation profiling were conducted on tumors. We report the case of a female patient diagnosed at age 12 with a diffuse glioma harboring a ROS1 fusion. At age 18, she developed a renal cell carcinoma. Genetic germline testing revealed a heterozygous germline POT1 variant, c.910dupG (p.Asp304fs*8), classified as pathogenic. Segregation analysis demonstrated paternal inheritance of the variant. The same POT1 variant was identified in both tumor tissues in a heterozygous state. This is the first reported case of a young adult carrying a new pathogenic POT1 variant who developed a pediatric high-grade glioma and a renal tumor in early adulthood. This report expands the clinical spectrum of POT1-associated tumors with early-onset and underscores the relevance of genetic testing for patient management and family counseling.
2026
Inglese
Cipri, S., Cacchione, A., Serra, A., Del Baldo, G., Miele, E., Pedace, L., Patrizi, S., D'Antonio, F., Agolini, E., Novelli, A., Crocoli, A., Bertocchini, A., Carai, A., Barresi, S., Rossi, S., Alaggio, R., Diomedi Camassei, F., Colafati, G. S., Boccuto, L., Mastronuzzi, A., Case Report: The revelation of a new pathogenic variant in the POT1 gene in a patient with a pediatric high-grade glioma and a renal cell carcinoma, <<FRONTIERS IN ONCOLOGY>>, 2026; 16 (16): 1-7. [doi:10.3389/fonc.2026.1778785] [https://hdl.handle.net/10807/342243]
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