Alexandre Roux1,2,3, Nathalie Boddaert2,4,5, Jacques Grill6,7, David Castel6,7, Marc Zanello1,2,3, Gilles Zah-Bi1, Fabrice Chrétien2,8, Etienne Lefevre2, Volodia Dangouloff Ros2,4,5, Michel Zerah2,9, Stéphanie Puget2,9, Johan Pallud1,2,3, Pascale Varlet2,3,8. 1. Department of Neurosurgery, Sainte-Anne Hospital, Paris, France. 2. Université Paris Descartes, Sorbonne Paris Cité, Paris, France. 3. Inserm UMR 1266, IMA-Brain, Institut de Psychiatrie et Neurosciences de Paris, Paris, France. 4. Department of Paediatric Radiology, Necker Enfants-Malades Hospital, Paris, France. 5. Inserm UMR 1163, Institut Imagine, Inserm U1000, Paris, France. 6. Department of Pediatric Oncology, Gustave-Roussy, Université Paris-Sud, Université Paris-Saclay, Villejuif, France. 7. UMR8203 "Vectorologie et Thérapeutiques Anticancéreuses," CNRS, Gustave Roussy, Université Paris-Sud, Université Paris-Saclay, Villejuif, France. 8. Department of Neuropathology, Sainte-Anne Hospital, Paris, France. 9. Department of Pediatric Neurosurgery, Necker Enfants-Malades Hospital, Paris, France.
Abstract
BACKGROUND: No link has been demonstrated between diffuse intrinsic pontine glioma and developmental venous anomaly in pediatric patients. OBJECTIVE: To determine the prevalence of developmental venous anomaly in a pediatric cohort of diffuse intrinsic pontine glioma. METHODS: We performed a retrospective cohort study (1998-2017) of consecutive pediatric patients harboring a diffuse intrinsic pontine glioma (experimental set, n = 162) or a craniopharyngioma (control set, n = 142) in a tertiary pediatric neurosurgical center. The inclusion criteria were the following: age <18 yr at diagnosis; histopathological diagnosis of diffuse intrinsic pontine glioma or craniopharyngioma according to the 2016 World Health Organization classification of tumors of the central nervous system; no previous oncological treatment; and available preoperative magnetic resonance imaging performed with similar acquisition protocol. RESULTS: We found a significantly higher prevalence of developmental venous anomaly in the experimental set of 162 diffuse intrinsic pontine gliomas (24.1%) than in the control set of 142 craniopharyngiomas (10.6%; P = .001). The prevalence of developmental venous anomalies was not significantly impacted by demographic data (sex, age at diagnosis, and underlying pathological condition), biomolecular analysis (H3-K27M-mutant subgroup, H3.1-K27M-mutant subgroup, and H3.3-K27M-mutant subgroup), or imaging findings (anatomic location, anatomic extension, side, and obstructive hydrocephalus) of the studied diffuse intrinsic pontine gliomas. CONCLUSION: We report a higher prevalence of developmental venous anomaly in pediatric diffuse intrinsic pontine glioma patients than in control patients, which suggests a potential underlying common predisposition or a causal relationship that will require deeper investigations.
BACKGROUND: No link has been demonstrated between diffuse intrinsic pontine glioma and developmental venous anomaly in pediatric patients. OBJECTIVE: To determine the prevalence of developmental venous anomaly in a pediatric cohort of diffuse intrinsic pontine glioma. METHODS: We performed a retrospective cohort study (1998-2017) of consecutive pediatric patients harboring a diffuse intrinsic pontine glioma (experimental set, n = 162) or a craniopharyngioma (control set, n = 142) in a tertiary pediatric neurosurgical center. The inclusion criteria were the following: age <18 yr at diagnosis; histopathological diagnosis of diffuse intrinsic pontine glioma or craniopharyngioma according to the 2016 World Health Organization classification of tumors of the central nervous system; no previous oncological treatment; and available preoperative magnetic resonance imaging performed with similar acquisition protocol. RESULTS: We found a significantly higher prevalence of developmental venous anomaly in the experimental set of 162 diffuse intrinsic pontine gliomas (24.1%) than in the control set of 142 craniopharyngiomas (10.6%; P = .001). The prevalence of developmental venous anomalies was not significantly impacted by demographic data (sex, age at diagnosis, and underlying pathological condition), biomolecular analysis (H3-K27M-mutant subgroup, H3.1-K27M-mutant subgroup, and H3.3-K27M-mutant subgroup), or imaging findings (anatomic location, anatomic extension, side, and obstructive hydrocephalus) of the studied diffuse intrinsic pontine gliomas. CONCLUSION: We report a higher prevalence of developmental venous anomaly in pediatric diffuse intrinsic pontine gliomapatients than in control patients, which suggests a potential underlying common predisposition or a causal relationship that will require deeper investigations.
Authors: Daniel A Snellings; Romuald Girard; Rhonda Lightle; Abhinav Srinath; Sharbel Romanos; Ying Li; Chang Chen; Aileen A Ren; Mark L Kahn; Issam A Awad; Douglas A Marchuk Journal: Nat Cardiovasc Res Date: 2022-03-14