Germ cell tumors and associated hematologic malignancies evolve from a common shared precursor

Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the mediastinum have distinctly poor outcomes. One in every 17 patients with primary mediastinal nonseminomatous GCTs develop an incurable hematolo...

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Vydáno v:The Journal of clinical investigation Ročník 130; číslo 12; s. 6668 - 6676
Hlavní autoři: Taylor, Justin, Donoghue, Mark T.A., Ho, Caleb, Petrova-Drus, Kseniya, Al-Ahmadie, Hikmat A., Funt, Samuel A., Zhang, Yanming, Aypar, Umut, Rao, Pavitra, Chavan, Shweta S., Haddadin, Michael, Tamari, Roni, Giralt, Sergio, Tallman, Martin S., Rampal, Raajit K., Baez, Priscilla, Kappagantula, Rajya, Kosuri, Satyajit, Dogan, Ahmet, Tickoo, Satish K., Reuter, Victor E., Bosl, George J., Iacobuzio-Donahue, Christine A., Solit, David B., Taylor, Barry S., Feldman, Darren R., Abdel-Wahab, Omar
Médium: Journal Article
Jazyk:angličtina
Vydáno: United States American Society for Clinical Investigation 01.12.2020
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ISSN:0021-9738, 1558-8238, 1558-8238
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Abstract Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the mediastinum have distinctly poor outcomes. One in every 17 patients with primary mediastinal nonseminomatous GCTs develop an incurable hematologic malignancy and prior data intriguingly suggest a clonal relationship exists between hematologic malignancies and GCTs in these cases. To date, however, the precise clonal relationship between GCTs and the diverse additional somatic malignancies arising in such individuals have not been determined. Here, we traced the clonal evolution and characterized the genetic features of each neoplasm from a cohort of 15 patients with GCTs and associated hematologic malignancies. We discovered that GCTs and hematologic malignancies developing in such individuals evolved from a common shared precursor, nearly all of which harbored allelically imbalanced p53 and/or RAS pathway mutations. Hematologic malignancies arising in this setting genetically resembled mediastinal GCTs rather than de novo myeloid neoplasms. Our findings argue that this scenario represents a unique clinical syndrome, distinct from de novo GCTs or hematologic malignancies, initiated by an ancestral precursor that gives rise to the parallel evolution of GCTs and blood cancers in these patients.
AbstractList Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the mediastinum have distinctly poor outcomes. One in every 17 patients with primary mediastinal nonseminomatous GCTs develop an incurable hematologic malignancy and prior data intriguingly suggest a clonal relationship exists between hematologic malignancies and GCTs in these cases. To date, however, the precise clonal relationship between GCTs and the diverse additional somatic malignancies arising in such individuals have not been determined. Here, we traced the clonal evolution and characterized the genetic features of each neoplasm from a cohort of 15 patients with GCTs and associated hematologic malignancies. We discovered that GCTs and hematologic malignancies developing in such individuals evolved from a common shared precursor, nearly all of which harbored allelically imbalanced p53 and/or RAS pathway mutations. Hematologic malignancies arising in this setting genetically resembled mediastinal GCTs rather than de novo myeloid neoplasms. Our findings argue that this scenario represents a unique clinical syndrome, distinct from de novo GCTs or hematologic malignancies, initiated by an ancestral precursor that gives rise to the parallel evolution of GCTs and blood cancers in these patients.
Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the mediastinum have distinctly poor outcomes. One in every 17 patients with primary mediastinal nonseminomatous GCTs develop an incurable hematologic malignancy and prior data intriguingly suggest a clonal relationship exists between hematologic malignancies and GCTs in these cases. To date, however, the precise clonal relationship between GCTs and the diverse additional somatic malignancies arising in such individuals have not been determined. Here, we traced the clonal evolution and characterized the genetic features of each neoplasm from a cohort of 15 patients with GCTs and associated hematologic malignancies. We discovered that GCTs and hematologic malignancies developing in such individuals evolved from a common shared precursor, nearly all of which harbored allelically imbalanced p53 and/or RAS pathway mutations. Hematologic malignancies arising in this setting genetically resembled mediastinal GCTs rather than de novo myeloid neoplasms. Our findings argue that this scenario represents a unique clinical syndrome, distinct from de novo GCTs or hematologic malignancies, initiated by an ancestral precursor that gives rise to the parallel evolution of GCTs and blood cancers in these patients.Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the mediastinum have distinctly poor outcomes. One in every 17 patients with primary mediastinal nonseminomatous GCTs develop an incurable hematologic malignancy and prior data intriguingly suggest a clonal relationship exists between hematologic malignancies and GCTs in these cases. To date, however, the precise clonal relationship between GCTs and the diverse additional somatic malignancies arising in such individuals have not been determined. Here, we traced the clonal evolution and characterized the genetic features of each neoplasm from a cohort of 15 patients with GCTs and associated hematologic malignancies. We discovered that GCTs and hematologic malignancies developing in such individuals evolved from a common shared precursor, nearly all of which harbored allelically imbalanced p53 and/or RAS pathway mutations. Hematologic malignancies arising in this setting genetically resembled mediastinal GCTs rather than de novo myeloid neoplasms. Our findings argue that this scenario represents a unique clinical syndrome, distinct from de novo GCTs or hematologic malignancies, initiated by an ancestral precursor that gives rise to the parallel evolution of GCTs and blood cancers in these patients.
Audience Academic
Author Aypar, Umut
Reuter, Victor E.
Iacobuzio-Donahue, Christine A.
Tallman, Martin S.
Donoghue, Mark T.A.
Baez, Priscilla
Tickoo, Satish K.
Al-Ahmadie, Hikmat A.
Haddadin, Michael
Funt, Samuel A.
Rao, Pavitra
Zhang, Yanming
Chavan, Shweta S.
Giralt, Sergio
Solit, David B.
Kappagantula, Rajya
Dogan, Ahmet
Rampal, Raajit K.
Feldman, Darren R.
Abdel-Wahab, Omar
Bosl, George J.
Petrova-Drus, Kseniya
Kosuri, Satyajit
Taylor, Barry S.
Taylor, Justin
Ho, Caleb
Tamari, Roni
AuthorAffiliation 2 Division of Hematology, Department of Medicine, Sylvester Comprehensive Cancer Center at the University of Miami Miller School of Medicine, Miami, Florida, USA
3 Marie-Josée and Henry R. Kravis Center for Molecular Oncology
8 Deparment of Epidemiology and Biostatistics, Memorial Sloan Kettering Cancer Center, New York, New York, USA
6 Bone Marrow Transplant Service, Department of Medicine
1 Human Oncology and Pathogenesis Program, Memorial Sloan Kettering Cancer Center, New York, New York, USA
4 Department of Pathology
5 Genitourinary Oncology Service, Department of Medicine
7 Leukemia Service, Department of Medicine
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BackLink https://www.ncbi.nlm.nih.gov/pubmed/32897884$$D View this record in MEDLINE/PubMed
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Keywords Genetics
Hematology
Leukemias
Cancer
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Snippet Germ cell tumors (GCTs) are the most common cancer in men between the ages of 15 and 40. Although most patients are cured, those with disease arising in the...
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StartPage 6668
SubjectTerms Adolescent
Adult
Biomedical research
Blood cancer
Cancer therapies
Care and treatment
Chemotherapy
Cloning
Deoxyribonucleic acid
Development and progression
DNA
Evolution & development
Female
Gene expression
Gene mutation
Genetic aspects
Genotypes
Germinoma
Health aspects
Hematologic Neoplasms - genetics
Hematologic Neoplasms - metabolism
Hematologic Neoplasms - pathology
Hematological diseases
Histology
Humans
Identification and classification
Leukemia
Malignancy
Mediastinum
Medical prognosis
Middle Aged
Mutation
Neoplasms, Germ Cell and Embryonal - genetics
Neoplasms, Germ Cell and Embryonal - metabolism
Neoplasms, Germ Cell and Embryonal - pathology
Neoplasms, Second Primary - genetics
Neoplasms, Second Primary - metabolism
Neoplasms, Second Primary - pathology
p53 Protein
Patients
Signal Transduction - genetics
Tumor Suppressor Protein p53 - genetics
Tumor Suppressor Protein p53 - metabolism
Tumors
Title Germ cell tumors and associated hematologic malignancies evolve from a common shared precursor
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