MYOD1 as a prognostic indicator in rhabdomyosarcoma.


Journal

Pediatric blood & cancer
ISSN: 1545-5017
Titre abrégé: Pediatr Blood Cancer
Pays: United States
ID NLM: 101186624

Informations de publication

Date de publication:
09 2021
Historique:
revised: 22 03 2021
received: 19 01 2021
accepted: 09 04 2021
pubmed: 30 4 2021
medline: 17 3 2022
entrez: 29 4 2021
Statut: ppublish

Résumé

Rhabdomyosarcoma (RMS) is characterized by the expression of the myogenic regulatory protein MYOD1. Histologic types include alveolar, embryonal (ERMS), and spindle cell sclerosing RMS (SRMS). SRMS harbors MYOD1 mutations in a subset of adult cases in association with poor prognosis. To study the level of MYOD1 protein expression and its clinical significance, we have analyzed variable numbers of pediatric (<18 years of age) and adult (age range ≥18 to 35 years) ERMS and SRMS cases for presence or absence of MYOD1 immunoreactivity in correlation with clinical outcome and MYOD1 L122R mutations. Lack of MYOD1 immunoreactivity, identified in 23.8% of nonalveolar RMS (non-ARMS) cases, was more prevalent in SRMS (44%) than ERMS (17.2%) and was significantly associated with low overall survival and unfavorable tumor sites (p < .05). Lack of MYOD1 immunoreactivity was not associated with MYOD1 L122R mutations, which were identified in 3/37 (8%) cases including only two of 31 (6.5%) pediatric cases, one of 11 or 9% pediatric SRMS, and one case of infant ERMS. These studies highlight the prognostic role of MYOD1 in non-ARMS. Lack of MYOD1 immunoreactivity is associated with poor prognosis in ERMS and SRMS. MYOD1 gene mutations are generally infrequent in pediatric RMS. Although mutations are predominant in SRMS, they may exceptionally occur in infantile ERMS.

Sections du résumé

BACKGROUND/OBJECTIVES
Rhabdomyosarcoma (RMS) is characterized by the expression of the myogenic regulatory protein MYOD1. Histologic types include alveolar, embryonal (ERMS), and spindle cell sclerosing RMS (SRMS). SRMS harbors MYOD1 mutations in a subset of adult cases in association with poor prognosis.
DESIGN/METHODS
To study the level of MYOD1 protein expression and its clinical significance, we have analyzed variable numbers of pediatric (<18 years of age) and adult (age range ≥18 to 35 years) ERMS and SRMS cases for presence or absence of MYOD1 immunoreactivity in correlation with clinical outcome and MYOD1 L122R mutations.
RESULTS
Lack of MYOD1 immunoreactivity, identified in 23.8% of nonalveolar RMS (non-ARMS) cases, was more prevalent in SRMS (44%) than ERMS (17.2%) and was significantly associated with low overall survival and unfavorable tumor sites (p < .05). Lack of MYOD1 immunoreactivity was not associated with MYOD1 L122R mutations, which were identified in 3/37 (8%) cases including only two of 31 (6.5%) pediatric cases, one of 11 or 9% pediatric SRMS, and one case of infant ERMS.
CONCLUSION
These studies highlight the prognostic role of MYOD1 in non-ARMS. Lack of MYOD1 immunoreactivity is associated with poor prognosis in ERMS and SRMS. MYOD1 gene mutations are generally infrequent in pediatric RMS. Although mutations are predominant in SRMS, they may exceptionally occur in infantile ERMS.

Identifiants

pubmed: 33913590
doi: 10.1002/pbc.29085
pmc: PMC9907363
mid: NIHMS1866377
doi:

Substances chimiques

MyoD Protein 0
MyoD1 myogenic differentiation protein 0

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

e29085

Subventions

Organisme : Intramural NIH HHS
ID : ZIA BC011387
Pays : United States

Informations de copyright

© 2021 Wiley Periodicals LLC.

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Auteurs

Atif A Ahmed (AA)

Department of Pathology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Sultan Habeebu (S)

Department of Pathology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Midhat S Farooqi (MS)

Department of Pathology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Alan S Gamis (AS)

Department of Pediatric Hematology Oncology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Elizabeth Gonzalez (E)

Department of Pediatric Hematology Oncology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Terrie Flatt (T)

Department of Pediatric Hematology Oncology, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Ashley Sherman (A)

Department of Health Services and Outcomes Research, Children's Mercy Hospital/University of Missouri, Kansas City, Missouri, USA.

Lea Surrey (L)

Department of Pathology, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

Michael A Arnold (MA)

Department of Pathology and Laboratory Medicine, Nationwide Children's Hospital, Columbus, Ohio, USA.
Department of Pathology, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.

Miriam Conces (M)

Department of Pathology and Laboratory Medicine, Nationwide Children's Hospital, Columbus, Ohio, USA.
Department of Pathology, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.

Selene Koo (S)

Department of Pathology and Laboratory Medicine, Nationwide Children's Hospital, Columbus, Ohio, USA.
Department of Pathology, The Ohio State University Wexner Medical Center, Columbus, Ohio, USA.

Nikolina Dioufa (N)

Department of Medicine, Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA.

Frederic G Barr (FG)

Laboratory of Pathology, National Cancer Institute, Bethesda, Maryland, USA.

Maria G Tsokos (MG)

Department of Medicine, Beth Israel Deaconess Medical Center, Boston, Massachusetts, USA.

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Classifications MeSH