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Sarcoma arising in Paget's disease of bone: Declining incidence and increasing age at presentation
Bone , 12/31/08

Mangham DC et al. - In a study to determine whether sarcomatous change exhibits increasing age at presentation, whether the trend to monostotic Paget's disease exists, whether male predominance in sarcoma still continues, and explore factors that might affect survival, it was found that the proportion of Paget's disease pts with sarcoma has fallen steadily and is now about 0.3%. Sarcoma is not necessarily associated with very high serum total alkaline phosphatase (ALP). It is present amongst polyostotic cases in the expected proportion suggesting that more widespread skeletal involvement by Paget's disease is not a significant risk factor for malignant transformation.

Methods

Notes of all pts from the primary malignant bone tumour registry were reviewed and all cases of Pagetic sarcoma since 1975 extracted.
In addition to basic demographic data, mode of presentation, skeletal involvement by Paget's disease, history of treatment, presence of pulmonary metastases, serum total ALP levels and survival were obtained.

Results

Unequivocal Pagetic sarcoma was identified in 32 pts (23 M, 9 F).
Age at presentation was 73.8 yrs with no sex difference and with known pre-existing Paget's disease in 42%.
Only 15% had received any specific Paget's disease treatment.
Serum total ALP was not invariably markedly elevated and was related to number of skeletal sites, but not to the sarcoma histological subtype.
Paget's disease was monostotic in 46%.
Pagetic sarcoma fell from 23% to 8% of primary bone sarcoma referrals in patients aged over 50 yrs between the decades 1986–1995 and 1996–2005.
Median survival remained poor at 0.66 yrs.
Survival of greater than 2 yrs occurred in 4 pts, 1 of whom with a low-grade Pagetic sarcoma being alive at 12 yrs follow up.

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