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• W3021482960 abstract "This 59-year-old post-menopausal woman was referred to the oncology clinic in May 1999 to be investigated for a malignant tumour because of right hip pain and a suspicion of pathologic fractures on Technetium 99m diphosphonate bone scan. The scan showed hot spots at the distal part of the right sacroiliac joint, the 10th rib, the right hip region on the lower acetabulum and the right superior and inferior pubic arch (left side).Her history retained a pre-symptomatic primary biliary cirrhosis (PBC), diagnosed in 1985 by liver biopsy. AMA was absent at diagnosis in 1985, and ANA became positive during follow-up with a speckled pattern at a titer of 1/640. At presentation in 1999 she was taking cholestyramine 8 g/day and 25000 IU 25(OH)D3 monthly during winter. Standard X-rays were negative. CT scan and MR showed fractures at the right ileopubic arch, sacrum and acetabulum. A trephine bone marrow biopsy from the right iliac crest and an open biopsy of the right ileopubic arch showed absence of malignant cells. An extensive diagnostic procedure, including a negative total body fluorodeoxyglucose PET scan, showed absence of malignant disease. Bone densitometry showed a T score of −5.21 SD and −5.23 SD at the lumbar vertebra and the right femoral neck, respectively. Serum total calcium was 8.7 mg/dl (8.9–10.5), AIc Phosph 831 U/I (90–260), 25(OH)D3 6.1 μg/l (7–60), PTH 11.7 ng/l (3–40) and osteocalcin 7.6 μg/l (11–43), compatible with hepatic osteodystrophy. The patient underwent liver transplantation in January 2000 because of rising cholestasis with a total bilirubin level at 5.7 mg/dl. She remained AMA negative and pANCA negative, but IgM was 3.54 g/l. The explant liver showed end-stage vanishing bile duct syndrome, presumably due to PBC, since lymphoid aggregates were still present. She is currently well under FK 506 based immunosuppression. In addition she takes 500 mg elementary calcium, 10 mg alendronate and 0.50 μg 1,25(OH2)D3 per day. A follow-up bone scan shows resolution of all fractures 1 year after transplantation (right side). Bone densitometry 18 months post-transplantation at the lumbar vertebra and right femoral neck has improved to a T score of −4.33 SD and −3.49 SD, respectively.Pathologic fractures may be a presenting sign of hepatic osteodystrophy. This condition is potentially reversible after liver transplantation and the start of bone specific therapy [1Eastell R. Dickson E.R. Hodgson S.F. Wiesner R.H. Porayko M.K. Wahner H.W. et al.Rates of vertebral bone loss before and after liver transplantation in women with primary biliary cirrhosis.Hepatology. 1991; 14: 296-300Crossref PubMed Scopus (272) Google Scholar, 2Collier J.D. Ninkovic M. Compston J.E. Guidelines on the management of osteoporosis associated with chronic liver disease.Gut. 2002; 50: i1-i9Crossref PubMed Google Scholar]. This 59-year-old post-menopausal woman was referred to the oncology clinic in May 1999 to be investigated for a malignant tumour because of right hip pain and a suspicion of pathologic fractures on Technetium 99m diphosphonate bone scan. The scan showed hot spots at the distal part of the right sacroiliac joint, the 10th rib, the right hip region on the lower acetabulum and the right superior and inferior pubic arch (left side). Her history retained a pre-symptomatic primary biliary cirrhosis (PBC), diagnosed in 1985 by liver biopsy. AMA was absent at diagnosis in 1985, and ANA became positive during follow-up with a speckled pattern at a titer of 1/640. At presentation in 1999 she was taking cholestyramine 8 g/day and 25000 IU 25(OH)D3 monthly during winter. Standard X-rays were negative. CT scan and MR showed fractures at the right ileopubic arch, sacrum and acetabulum. A trephine bone marrow biopsy from the right iliac crest and an open biopsy of the right ileopubic arch showed absence of malignant cells. An extensive diagnostic procedure, including a negative total body fluorodeoxyglucose PET scan, showed absence of malignant disease. Bone densitometry showed a T score of −5.21 SD and −5.23 SD at the lumbar vertebra and the right femoral neck, respectively. Serum total calcium was 8.7 mg/dl (8.9–10.5), AIc Phosph 831 U/I (90–260), 25(OH)D3 6.1 μg/l (7–60), PTH 11.7 ng/l (3–40) and osteocalcin 7.6 μg/l (11–43), compatible with hepatic osteodystrophy. The patient underwent liver transplantation in January 2000 because of rising cholestasis with a total bilirubin level at 5.7 mg/dl. She remained AMA negative and pANCA negative, but IgM was 3.54 g/l. The explant liver showed end-stage vanishing bile duct syndrome, presumably due to PBC, since lymphoid aggregates were still present. She is currently well under FK 506 based immunosuppression. In addition she takes 500 mg elementary calcium, 10 mg alendronate and 0.50 μg 1,25(OH2)D3 per day. A follow-up bone scan shows resolution of all fractures 1 year after transplantation (right side). Bone densitometry 18 months post-transplantation at the lumbar vertebra and right femoral neck has improved to a T score of −4.33 SD and −3.49 SD, respectively. Pathologic fractures may be a presenting sign of hepatic osteodystrophy. This condition is potentially reversible after liver transplantation and the start of bone specific therapy [1Eastell R. Dickson E.R. Hodgson S.F. Wiesner R.H. Porayko M.K. Wahner H.W. et al.Rates of vertebral bone loss before and after liver transplantation in women with primary biliary cirrhosis.Hepatology. 1991; 14: 296-300Crossref PubMed Scopus (272) Google Scholar, 2Collier J.D. Ninkovic M. Compston J.E. Guidelines on the management of osteoporosis associated with chronic liver disease.Gut. 2002; 50: i1-i9Crossref PubMed Google Scholar]." @default.
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• W3021482960 title "Hepatic osteodystrophy" @default.
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• W3021482960 doi "https://doi.org/10.1016/s0168-8278(02)00381-1" @default.
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