AIMS: Diffuse intrinsic pontine glioma (DIPG) is a fatal paediatric malignancy. Tumour resection is not possible without serious morbidity and biopsies are rarely performed. The resulting lack of primary DIPG material has made preclinical research practically impossible and has hindered the development of new therapies for this disease. The aim of the current study was to address the lack of primary DIPG material and preclinical models by developing a multi-institutional autopsy protocol. METHODS: An autopsy protocol was implemented in the Netherlands to obtain tumour material within a brief post mortem interval. A team of neuropathologists and researchers was available at any time to perform the autopsy and process the material harvested. Whole brain autopsy was performed and primary DIPG material and healthy tissue were collected from all affected brain areas. Finally, the study included systematic evaluation by parents. RESULTS: Five autopsies were performed. The mean time interval between death and time of autopsy was 3 h (range 2-4). All tumours were graded as glioblastoma. None of the parents regretted their choice to participate, and they all derived comfort in donating tissue of their child in the hope to help future DIPG patients. In addition, we developed and characterized one of the first DIPG cell cultures from post mortem material. CONCLUSION: Here we show that obtaining post mortem DIPG tumour tissue for research purposes is feasible with short delay, and that the autopsy procedure is satisfying for participating parents and can be suitable for the development of preclinical DIPG models.
AIMS: Diffuse intrinsic pontine glioma (DIPG) is a fatal paediatric malignancy. Tumour resection is not possible without serious morbidity and biopsies are rarely performed. The resulting lack of primary DIPG material has made preclinical research practically impossible and has hindered the development of new therapies for this disease. The aim of the current study was to address the lack of primary DIPG material and preclinical models by developing a multi-institutional autopsy protocol. METHODS: An autopsy protocol was implemented in the Netherlands to obtain tumour material within a brief post mortem interval. A team of neuropathologists and researchers was available at any time to perform the autopsy and process the material harvested. Whole brain autopsy was performed and primary DIPG material and healthy tissue were collected from all affected brain areas. Finally, the study included systematic evaluation by parents. RESULTS: Five autopsies were performed. The mean time interval between death and time of autopsy was 3 h (range 2-4). All tumours were graded as glioblastoma. None of the parents regretted their choice to participate, and they all derived comfort in donating tissue of their child in the hope to help future DIPGpatients. In addition, we developed and characterized one of the first DIPG cell cultures from post mortem material. CONCLUSION: Here we show that obtaining post mortem DIPGtumour tissue for research purposes is feasible with short delay, and that the autopsy procedure is satisfying for participating parents and can be suitable for the development of preclinical DIPG models.
Authors: Viola Caretti; A Charlotte P Sewing; Tonny Lagerweij; Pepijn Schellen; Marianna Bugiani; Marc H A Jansen; Dannis G van Vuurden; Anna C Navis; Ilona Horsman; W Peter Vandertop; David P Noske; Pieter Wesseling; Gertjan J L Kaspers; Javad Nazarian; Hannes Vogel; Esther Hulleman; Michelle Monje; Thomas Wurdinger Journal: Acta Neuropathol Date: 2014-04-29 Impact factor: 17.088
Authors: Briony F Hudson; Linda Jm Oostendorp; Bridget Candy; Victoria Vickerstaff; Louise Jones; Monica Lakhanpaul; Myra Bluebond-Langner; Paddy Stone Journal: Palliat Med Date: 2016-09-08 Impact factor: 4.762
Authors: Maria Tsoli; Han Shen; Chelsea Mayoh; Laura Franshaw; Anahid Ehteda; Danielle Upton; Diana Carvalho; Maria Vinci; Michael H Meel; Dannis van Vuurden; Alexander Plessier; David Castel; Rachid Drissi; Michael Farrell; Jane Cryan; Darach Crimmins; John Caird; Jane Pears; Stephanie Francis; Louise E A Ludlow; Andrea Carai; Angela Mastronuzzi; Bing Liu; Jordan Hansford; Nick Gottardo; Tim Hassall; Maria Kirby; Maryam Fouladi; Cynthia Hawkins; Michelle Monje; Jacques Grill; Chris Jones; Esther Hulleman; David S Ziegler Journal: J Neurooncol Date: 2018-11-16 Impact factor: 4.130
Authors: Marc H A Jansen; Reina W Kloet; Dannis G van Vuurden; Sophie Em Veldhuijzen van Zanten; Birgit I Witte; Serge Goldman; W Peter Vandertop; Emile Fi Comans; Otto S Hoekstra; Ronald Boellaard; Gert-Jan Jl Kaspers Journal: EJNMMI Res Date: 2014-01-28 Impact factor: 3.138