PURPOSE: (18)F-Fluoride PET/CT is a relatively undervalued diagnostic test to measure bone metabolism in bone diseases. Hyperostosis cranialis interna (HCI) is a (hereditary) bone disease characterised by endosteal hyperostosis and osteosclerosis of the skull and the skull base. Bone overgrowth causes entrapment and dysfunction of several cranial nerves. The aim of this study is to compare standardised uptake values (SUVs) at different sites in order to quantify bone metabolism in the affected anatomical regions in HCI patients. METHODS: Nine affected family members, seven non-affected family members and nine non-HCI non-family members underwent (18)F-fluoride PET/CT scans. SUVs were systematically measured in the different regions of interest: frontal bone, sphenoid bone, petrous bone and clivus. Moreover, the average (18)F-fluoride uptake in the entire skull was measured by assessing the uptake in axial slides. Visual assessment of the PET scans of affected individuals was performed to discover the process of disturbed bone metabolism in HCI. RESULTS: (18)F-Fluoride uptake is statistically significantly higher in the sphenoid bone and clivus regions of affected family members. Visual assessment of the scans of HCI patients is relevant in detecting disease severity and the pattern of disturbed bone metabolism throughout life. CONCLUSION: (18)F-Fluoride PET/CT is useful in quantifying the metabolic activity in HCI and provides information about the process of disturbed bone metabolism in this specific disorder. Limitations are a narrow window between normal and pathological activity and the influence of age. This study emphasises that (18)F-fluoride PET/CT may also be a promising diagnostic tool for other metabolic bone disorders, even those with an indolent course.
PURPOSE: (18)F-Fluoride PET/CT is a relatively undervalued diagnostic test to measure bone metabolism in bone diseases. Hyperostosis cranialis interna (HCI) is a (hereditary) bone disease characterised by endosteal hyperostosis and osteosclerosis of the skull and the skull base. Bone overgrowth causes entrapment and dysfunction of several cranial nerves. The aim of this study is to compare standardised uptake values (SUVs) at different sites in order to quantify bone metabolism in the affected anatomical regions in HCIpatients. METHODS: Nine affected family members, seven non-affected family members and nine non-HCI non-family members underwent (18)F-fluoride PET/CT scans. SUVs were systematically measured in the different regions of interest: frontal bone, sphenoid bone, petrous bone and clivus. Moreover, the average (18)F-fluoride uptake in the entire skull was measured by assessing the uptake in axial slides. Visual assessment of the PET scans of affected individuals was performed to discover the process of disturbed bone metabolism in HCI. RESULTS: (18)F-Fluoride uptake is statistically significantly higher in the sphenoid bone and clivus regions of affected family members. Visual assessment of the scans of HCIpatients is relevant in detecting disease severity and the pattern of disturbed bone metabolism throughout life. CONCLUSION: (18)F-Fluoride PET/CT is useful in quantifying the metabolic activity in HCI and provides information about the process of disturbed bone metabolism in this specific disorder. Limitations are a narrow window between normal and pathological activity and the influence of age. This study emphasises that (18)F-fluoride PET/CT may also be a promising diagnostic tool for other metabolic bone disorders, even those with an indolent course.
Authors: M Piert; T T Zittel; G A Becker; M Jahn; A Stahlschmidt; G Maier; H J Machulla; R Bares Journal: J Nucl Med Date: 2001-07 Impact factor: 10.057
Authors: R A Hawkins; Y Choi; S C Huang; C K Hoh; M Dahlbom; C Schiepers; N Satyamurthy; J R Barrio; M E Phelps Journal: J Nucl Med Date: 1992-05 Impact factor: 10.057
Authors: C Messa; W G Goodman; C K Hoh; Y Choi; A R Nissenson; I B Salusky; M E Phelps; R A Hawkins Journal: J Clin Endocrinol Metab Date: 1993-10 Impact factor: 5.958
Authors: Gretl Hendrickx; Vere M Borra; Ellen Steenackers; Timur A Yorgan; Christophe Hermans; Eveline Boudin; Jérôme J Waterval; Ineke D C Jansen; Tolunay Beker Aydemir; Niels Kamerling; Geert J Behets; Christine Plumeyer; Patrick C D'Haese; Björn Busse; Vincent Everts; Martin Lammens; Geert Mortier; Robert J Cousins; Thorsten Schinke; Robert J Stokroos; Johannes J Manni; Wim Van Hul Journal: PLoS Genet Date: 2018-04-05 Impact factor: 5.917