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- Nelson, A E, et al.
(författare)
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Diagnosis of a patient with oncogenic osteomalacia using a phosphate uptake bioassay of serum and magnetic resonance imaging
- 2001
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Ingår i: European Journal of Endocrinology. - : Oxford University Press (OUP). - 0804-4643 .- 1479-683X. ; 145:4, s. 469-476
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Tidskriftsartikel (refereegranskat)abstract
- A previously healthy man with no family history of fractures presented with muscle pain, back pain and height loss. Investigations revealed hypophosphataemia, phosphaturia, undetectable serum 1,25-dihydroxyvitamin D and severe osteomalacia on bone biopsy, suggestive of a diagnosis of oncogenic osteomalacia. Thorough physical examination did not locate a tumour. Support for the diagnosis was obtained by detection of phosphate uptake inhibitory activity in a blinded sample of the patient's serum using a renal cell bioassay. On the basis of detection of this bioactivity, a total body magnetic resonance (MR) examination was performed. A small tumour was located in the right leg. Removal of the tumour resulted in the rapid reversal of symptoms and the abnormal biochemistry typical of oncogenic osteomalacia. Inhibitory activity was also demonstrated using the bioassay in serum from two other patients with confirmed or presumptive oncogenic osteomalacia, but not in serum from two patients with hypophosphataemia of other origin. This is the first case to be reported in which the diagnosis of oncogenic osteomalacia was assisted by demonstration of inhibitory activity of the patient's serum in a renal cell phosphate bioassay that provided an impetus for total body MR imaging.
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2. |
- Wikström, Johan, et al.
(författare)
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Gadobenate dimeglumine-enhanced magnetic resonance angiography of the pelvic arteries
- 2003
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Ingår i: Investigative Radiology. - : Ovid Technologies (Wolters Kluwer Health). - 0020-9996 .- 1536-0210. ; 38:8, s. 504-515
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Tidskriftsartikel (refereegranskat)abstract
- RATIONALE AND OBJECTIVES: To evaluate 4 doses of gadobenate dimeglumine (Gd-BOPTA) for contrast-enhanced magnetic resonance angiography (CE-MRA) of the pelvic arteries and to compare CE-MRA with unenhanced time-of-flight MRA (2D-TOF-MRA). METHODS: A multicenter Phase II dose-finding study was performed in 136 patients with Gd-BOPTA doses of 0.025, 0.05, 0.1, and 0.2 mmol/kg bodyweight. Evaluation of CE-MRA images and comparison with 2D-TOF-MRA images was performed onsite and by 2 blinded offsite reviewers in terms of subjective image quality, number of lesions detected, and confidence in lesion characterization. RESULTS: Significant (P < 0.05) improvements over unenhanced findings were observed for CE-MRA at all dose levels. For reviewer 1 and the onsite investigators, the overall image quality increased up to a dose of 0.1 mmol/kg and then plateaued. For reviewer 2, increased image quality was noted up to a dose of 0.2 mmol/kg. Significant (P < 0.005) increases in diagnostic confidence on CE-MRA versus unenhanced MRA was observed for all dose groups by reviewer 1 and the onsite investigators and for the 0.1 and 0.2 mmol/kg dose groups by reviewer 2. No serious adverse events were recorded that were attributable to the study drug and no trends in laboratory parameters, vital signs, or electrocardiogram recordings were observed. CONCLUSIONS: Gadobenate dimeglumine-enhanced MRA is safe and significantly more effective than unenhanced 2D-TOF-MRA for imaging the pelvic arteries. A dose of 0.1 mmol/kg appears the most appropriate dose for subsequent Phase III clinical evaluation.
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