A Cohort Study of CNS Tumors in Multiple ...
Type de document :
Compte-rendu et recension critique d'ouvrage
PMID :
Titre :
A Cohort Study of CNS Tumors in Multiple Endocrine Neoplasia Type 1
Auteur(s) :
Graillon, Thomas [Auteur correspondant]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Institut Marseille Maladies Rares [MarMaRa]
Romanet, Pauline [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Camilla, Clara [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Gélin, Camille [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Appay, Romain [Auteur]
Institut de neurophysiopathologie [INP]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Roche, Catherine [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Lagarde, Arnaud [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Mougel, Grégory [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Farah, Kaissar [Auteur]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Le Bras, Maëlle [Auteur]
PHU 2 - Institut du Thorax et du Sytème Nerveux [CHU Nantes] [ITSN]
Engelhardt, Julien [Auteur]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
CHU Bordeaux
Kalamarides, Michel [Auteur]
CHU Pitié-Salpêtrière [AP-HP]
Peyre, Matthieu [Auteur]
CHU Pitié-Salpêtrière [AP-HP]
Amelot, Aymeric [Auteur]
Hôpital Bretonneau
Emery, Evelyne [Auteur]
CHU Caen
Physiopathologie et imagerie des troubles neurologiques [PhIND]
GIP Cyceron [Cyceron]
Institut Blood and Brain @ Caen-Normandie [Caen] [BB@C]
Magro, Elsa [Auteur]
Centre Hospitalier Régional Universitaire de Brest [CHRU Brest]
Cebula, Hélène [Auteur]
Hôpital de Hautepierre [Strasbourg]
Aboukais, Rabih [Auteur]
Thérapies Assistées par Lasers et Immunothérapies pour l'Oncologie - U 1189 [OncoThAI]
Bauters, Catherine [Auteur]
Hôpital Claude Huriez [Lille]
Jouanneau, Emmanuel [Auteur]
Hospices Civils de Lyon [HCL]
Université Claude Bernard Lyon 1 [UCBL]
Centre de Recherche en Cancérologie de Lyon [UNICANCER/CRCL]
Berhouma, Moncef [Auteur]
Hospices Civils de Lyon [HCL]
Centre de Recherche en Acquisition et Traitement de l'Image pour la Santé [CREATIS]
Cuny, Thomas [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Dufour, Henry [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Institut de Neurosciences de la Timone [INT]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
Loiseau, Hugues [Auteur]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
CHU Bordeaux
Figarella‑branger, Dominique [Auteur]
Institut de neurophysiopathologie [INP]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Bauchet, Luc [Auteur]
Neurochirurgie [Hôpital Gui de Chauliac]
Institut de Génomique Fonctionnelle [IGF]
Binquet, Christine [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Barlier, Anne [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Goudet, Pierre [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Institut Marseille Maladies Rares [MarMaRa]
Romanet, Pauline [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Camilla, Clara [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Gélin, Camille [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Appay, Romain [Auteur]
Institut de neurophysiopathologie [INP]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Roche, Catherine [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Lagarde, Arnaud [Auteur]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Mougel, Grégory [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Farah, Kaissar [Auteur]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Le Bras, Maëlle [Auteur]
PHU 2 - Institut du Thorax et du Sytème Nerveux [CHU Nantes] [ITSN]
Engelhardt, Julien [Auteur]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
CHU Bordeaux
Kalamarides, Michel [Auteur]
CHU Pitié-Salpêtrière [AP-HP]
Peyre, Matthieu [Auteur]
CHU Pitié-Salpêtrière [AP-HP]
Amelot, Aymeric [Auteur]
Hôpital Bretonneau
Emery, Evelyne [Auteur]
CHU Caen
Physiopathologie et imagerie des troubles neurologiques [PhIND]
GIP Cyceron [Cyceron]
Institut Blood and Brain @ Caen-Normandie [Caen] [BB@C]
Magro, Elsa [Auteur]
Centre Hospitalier Régional Universitaire de Brest [CHRU Brest]
Cebula, Hélène [Auteur]
Hôpital de Hautepierre [Strasbourg]
Aboukais, Rabih [Auteur]
Thérapies Assistées par Lasers et Immunothérapies pour l'Oncologie - U 1189 [OncoThAI]
Bauters, Catherine [Auteur]
Hôpital Claude Huriez [Lille]
Jouanneau, Emmanuel [Auteur]
Hospices Civils de Lyon [HCL]
Université Claude Bernard Lyon 1 [UCBL]
Centre de Recherche en Cancérologie de Lyon [UNICANCER/CRCL]
Berhouma, Moncef [Auteur]
Hospices Civils de Lyon [HCL]
Centre de Recherche en Acquisition et Traitement de l'Image pour la Santé [CREATIS]
Cuny, Thomas [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Dufour, Henry [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Institut de Neurosciences de la Timone [INT]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
Loiseau, Hugues [Auteur]
Institut des Maladies Neurodégénératives [Bordeaux] [IMN]
CHU Bordeaux
Figarella‑branger, Dominique [Auteur]
Institut de neurophysiopathologie [INP]
Hôpital de la Timone [CHU - APHM] [TIMONE]
Bauchet, Luc [Auteur]
Neurochirurgie [Hôpital Gui de Chauliac]
Institut de Génomique Fonctionnelle [IGF]
Binquet, Christine [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Barlier, Anne [Auteur]
Marseille medical genetics - Centre de génétique médicale de Marseille [MMG]
Institut Marseille Maladies Rares [MarMaRa]
Hôpital de la Conception [CHU - APHM] [LA CONCEPTION]
Goudet, Pierre [Auteur]
Physiopathologie des Dyslipidémies (CTM UMR 1231) [PADYS]
Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques [CIC-EC]
Titre de la revue :
CLINICAL CANCER RESEARCH
Pagination :
2835-2845
Éditeur :
American Association for Cancer Research
Date de publication :
2024-07
ISSN :
1078-0432
Discipline(s) HAL :
Sciences du Vivant [q-bio]/Médecine humaine et pathologie
Sciences cognitives/Neurosciences
Sciences du Vivant [q-bio]/Cancer
Sciences cognitives/Neurosciences
Sciences du Vivant [q-bio]/Cancer
Résumé en anglais : [en]
Purpose: Multiple endocrine neoplasia type 1 (MEN1) is thought to increase the risk of meningioma and ependymoma. Thus, we aimed to describe the frequency, incidence, and specific clinical and histological features of ...
Lire la suite >Purpose: Multiple endocrine neoplasia type 1 (MEN1) is thought to increase the risk of meningioma and ependymoma. Thus, we aimed to describe the frequency, incidence, and specific clinical and histological features of central nervous system (CNS) tumors in the MEN1 population (except pituitary tumors).Experimental design: The study population included patients harboring CNS tumors diagnosed with MEN1 syndrome after 1990 and followed up in the French MEN1 national cohort. The standardized incidence ratio (SIR) was calculated based on the French Gironde CNS Tumor Registry. Genomic analyses were performed on somatic DNA from seven CNS tumors, including meningiomas and ependymomas from patients with MEN1, and then on 50 sporadic meningiomas and ependymomas.Results: A total of 29 CNS tumors were found among the 1,498 symptomatic patients (2%; incidence = 47.4/100,000 person-years; SIR = 4.5), including 12 meningiomas (0.8%; incidence = 16.2/100,000; SIR = 2.5), 8 ependymomas (0.5%; incidence = 10.8/100,000; SIR = 17.6), 5 astrocytomas (0.3%; incidence = 6.7/100,000; SIR = 5.8), and 4 schwannomas (0.3%; incidence = 5.4/100,000; SIR = 12.7). Meningiomas in patients with MEN1 were benign, mostly meningothelial, with 11 years earlier onset compared with the sporadic population and an F/M ratio of 1/1. Spinal and cranial ependymomas were mostly classified as World Health Organization grade 2. A biallelic MEN1 inactivation was observed in 4/5 ependymomas and 1/2 meningiomas from patients with MEN1, whereas MEN1 deletion in one allele was present in 3/41 and 0/9 sporadic meningiomas and ependymomas, respectively.Conclusions: The incidence of each CNS tumor was higher in the MEN1 population than in the French general population. Meningiomas and ependymomas should be considered part of the MEN1 syndrome, but somatic molecular data are missing to conclude for astrocytomas and schwannomas.Lire moins >
Lire la suite >Purpose: Multiple endocrine neoplasia type 1 (MEN1) is thought to increase the risk of meningioma and ependymoma. Thus, we aimed to describe the frequency, incidence, and specific clinical and histological features of central nervous system (CNS) tumors in the MEN1 population (except pituitary tumors).Experimental design: The study population included patients harboring CNS tumors diagnosed with MEN1 syndrome after 1990 and followed up in the French MEN1 national cohort. The standardized incidence ratio (SIR) was calculated based on the French Gironde CNS Tumor Registry. Genomic analyses were performed on somatic DNA from seven CNS tumors, including meningiomas and ependymomas from patients with MEN1, and then on 50 sporadic meningiomas and ependymomas.Results: A total of 29 CNS tumors were found among the 1,498 symptomatic patients (2%; incidence = 47.4/100,000 person-years; SIR = 4.5), including 12 meningiomas (0.8%; incidence = 16.2/100,000; SIR = 2.5), 8 ependymomas (0.5%; incidence = 10.8/100,000; SIR = 17.6), 5 astrocytomas (0.3%; incidence = 6.7/100,000; SIR = 5.8), and 4 schwannomas (0.3%; incidence = 5.4/100,000; SIR = 12.7). Meningiomas in patients with MEN1 were benign, mostly meningothelial, with 11 years earlier onset compared with the sporadic population and an F/M ratio of 1/1. Spinal and cranial ependymomas were mostly classified as World Health Organization grade 2. A biallelic MEN1 inactivation was observed in 4/5 ependymomas and 1/2 meningiomas from patients with MEN1, whereas MEN1 deletion in one allele was present in 3/41 and 0/9 sporadic meningiomas and ependymomas, respectively.Conclusions: The incidence of each CNS tumor was higher in the MEN1 population than in the French general population. Meningiomas and ependymomas should be considered part of the MEN1 syndrome, but somatic molecular data are missing to conclude for astrocytomas and schwannomas.Lire moins >
Langue :
Anglais
Vulgarisation :
Non
Source :