LONG TERM PAMIDRONATE TREATMENT IN YOUNG ADULTS WITH POLYOSTOTIC FIBROUS DYSPLASIA OF BONE: CASE SERIES

PARISI MS,; OLIVERI B

CURRENT THERAPEUTIC RESEARCH-CLINICAL AND EXPERIMENTAL

ELSEVIER

Año: 2009 vol. 70 p. 161 - 172

0011-393X

Limited information is available about long-termpamidronate treatment in adults with fibrous dysplasia(FD)of bone.The aim of these case series stdy was to report the clinical outcome and the biochemical and densitometric findings in a group of young adult patients with polyostoticFD treated for >3 yearswith IV pamidronate. Pamidronate was administered every 6 months (60 mg/day for three days. Thereafter treatment was individualized. Pamidronate was administered shorter or longer based on response . Bone pain, radiography,serum bone alkaline phosphatase(BAP), and urine C-terminal cross-linking telopeptide of type I collagen(CTX-I)was assesed for a mean of 7 years.Bone mineral density(BMD) of FD areas (FDas) contralateral areas(CLa) werw measured at baseline , and at 24 and 48 months. Data were collected prospectevely. Seven patients(5w/2M) were included in the study (age 31+/-7.2 years)Pamidronate was associated with a reduction in bone pain and significant reduction in BAP in all patients at the end of the follow up (p<0.02)The mean reduction of CTX-I was 56% (3 patients-NS) Mean BMD of FDas was increased at 12 months(5.9% p<0.05) compared with baseline; but was not significantly increased at 2months(7.3%) probably reflecting a higher dispersion values due to individual response to treatment. No significant change was observed in CLa.  BMD of FDa was significantly decreased (-15.3%)compared to CLa at baseline, these decreases with pamidronate to 10.8 at 12 months and to 9.3% at 24 months (p<0.05). Refilling of the osteolytic lesions was not observed.CONCLUSIONS: These patients with FD of bone treated with IV pamidronate long term had improvement in bone pain and BMD, The effectiveness of individual pamidronate administration in the long term of FD adults should be investigated in blinded control trials