NUT carcinoma in children, adolescents and young adults.

Bromodomain and Extra-Terminal (BET) protein Children Histone deacetylase Midline carcinoma NUT carcinoma Rare tumour Targeted therapies

Journal

Bulletin du cancer
ISSN: 1769-6917
Titre abrégé: Bull Cancer
Pays: France
ID NLM: 0072416

Informations de publication

Date de publication:
Apr 2022
Historique:
received: 08 06 2021
revised: 22 01 2022
accepted: 31 01 2022
pubmed: 17 4 2022
medline: 7 5 2022
entrez: 16 4 2022
Statut: ppublish

Résumé

NUT carcinoma (NC), defined by the presence of the NUTM1 rearrangement, is an aggressive tumour associated with poor prognosis. This rare cancer is underdiagnosed and difficult to treat. The primary objective of this review is to describe the clinical, radiological and laboratory features of NC in young patients. The secondary objective is to propose a consensual strategy for the French very Rare Tumour group (FRACTURE group). NUT-specific antibody immunostaining in cases of undifferentiated or poorly differentiated carcinoma may demonstrate the specific NUT gene rearrangement. NCs are frequently advanced stage at diagnosis and the outcome remains poor despite a global strategy that generally includes conventional combination chemotherapy with wide local therapy (surgery, radiotherapy). Chemosensitivity is frequently only transient. Recent data have shown that new targeted drugs (histone deacetylase and bromodomain and extra-terminal protein inhibitors) are promising, but their role has yet to be evaluated in NC. Centralized data review is necessary to improve our knowledge of paediatric NC. We propose a multimodal strategy based on published data and their personal experience.

Sections du résumé

BACKGROUND BACKGROUND
NUT carcinoma (NC), defined by the presence of the NUTM1 rearrangement, is an aggressive tumour associated with poor prognosis. This rare cancer is underdiagnosed and difficult to treat.
OBJECTIVE AND METHODS OBJECTIVE
The primary objective of this review is to describe the clinical, radiological and laboratory features of NC in young patients. The secondary objective is to propose a consensual strategy for the French very Rare Tumour group (FRACTURE group).
RESULTS RESULTS
NUT-specific antibody immunostaining in cases of undifferentiated or poorly differentiated carcinoma may demonstrate the specific NUT gene rearrangement. NCs are frequently advanced stage at diagnosis and the outcome remains poor despite a global strategy that generally includes conventional combination chemotherapy with wide local therapy (surgery, radiotherapy). Chemosensitivity is frequently only transient.
CONCLUSION CONCLUSIONS
Recent data have shown that new targeted drugs (histone deacetylase and bromodomain and extra-terminal protein inhibitors) are promising, but their role has yet to be evaluated in NC. Centralized data review is necessary to improve our knowledge of paediatric NC. We propose a multimodal strategy based on published data and their personal experience.

Identifiants

pubmed: 35428456
pii: S0007-4551(22)00074-1
doi: 10.1016/j.bulcan.2022.01.015
pii:
doi:

Substances chimiques

Nuclear Proteins 0
Transcription Factors 0

Types de publication

Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

491-504

Informations de copyright

Copyright © 2022 Société Française du Cancer. Published by Elsevier Masson SAS. All rights reserved.

Auteurs

Lauriane Lemelle (L)

PSL University, Institut Curie, SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer), 75005 Paris, France; University of Paris, Paris, France. Electronic address: lauriane.lemelle@curie.fr.

Antoine Moya-Plana (A)

Paris-Saclay University, Gustave Roussy, Surgical and Interventional Department, 94805 Villejuif, France.

Benoît Dumont (B)

Institute for Paediatric Haematology and Oncology, Leon Bérard Cancer Centre, 69008 Lyon, France.

Brice Fresneau (B)

Université Paris-Saclay, Gustave Roussy, Department of Pediatric oncology, 94805 Villejuif, France.

Anne Laprie (A)

University Institute of Cancer Toulouse-Oncopôle, Department of Radiation Oncology, 31100 Toulouse, France.

Line Claude (L)

Léon Bérard Cancer Center, Radiation Oncology department, 69008 Lyon, France.

Sophie Deneuve (S)

Centre Léon Bérard, Oncologic Surgery Department, 69008 Lyon, France.

Camille Cordero (C)

PSL University, Institut Curie, SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer), 75005 Paris, France.

Gaelle Pierron (G)

Institut Curie, PSL Research University, Genetics Unit, Department of Tumor Biology, 75005 Paris, France.

Vincent Couloigner (V)

Assistance Publique-Hôpitaux de Paris, hôpital Necker Enfants-Malades, département de chirurgie de la tête et du cou, 75015 Paris, France.

Sophie Bernard (S)

Assistance Publique-Hôpitaux de Paris, hôpital Robert Debré, département de chirurgie de la tête et du cou, 75019 Paris, France.

Liesbeth Cardoen (L)

Institut Curie, département d'imagerie, Paris, France.

Hervé J Brisse (HJ)

Institut Curie, département d'imagerie, Paris, France.

Nina Jehanno (N)

Institut Curie, département de médecine nucléaire, Paris, France.

Lucy Metayer (L)

PSL University, Institut Curie, SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer), 75005 Paris, France.

Paul Fréneaux (P)

Institut Curie, département de médecine diagnostique et théranostique, 75005 Paris, France.

Sylvie Helfre (S)

Institut Curie, département de radiothérapie, 75005 Paris, France.

Fréderic Kolb (F)

University of California, Division of Plastic Surgery, Department of Surgery, San Diego California, USA.

Juliette Thariat (J)

Baclesse Cancer Center, Radiation Oncology Department, 14000 Caen, France.

Yves Réguerre (Y)

CHU de Saint Denis, service d'oncologie et d'hématologie pédiatrique, 97400 Saint-Denis, Reunion.

Daniel Orbach (D)

PSL University, Institut Curie, SIREDO Oncology Center (Care, Innovation and Research for Children, Adolescents and Young Adults with Cancer), 75005 Paris, France.

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Classifications MeSH