Abstract
The aim of this study was to evaluate strategies to halt the progression of severe dystrophic calcification in a patient with progressive systemic sclerosis (PSS) and to monitor serial changes in biochemical markers of bone resorption and indices of calcium (Ca) metabolism in response to therapy. The relationship of bone turnover to the extent of dystrophic calcification was also investigated in a number of additional patients with varying degrees of calcinosis. Serial markers of bone turnover and indices of Ca metabolism were measured over a 3-year period in one patient with PSS and severe dystrophic calcification. Bone mineral density (BMD) was assessed by dual-energy x-ray absorptiometry (DXA). Bone turnover in this patient and two additional patients with PSS or dermatomyositis (DM) and severe dystrophic calcification (Group A, n = 3) was compared with that in patients with connective tissue disease with little or no evidence of dystrophic calcification (Group B, n = 13). Serial data on one patient with severe progressive calcinosis showed increased bone resorption markers, which remained high over a 3-year period despite antiresorptive therapy. BMD was low. Patients with PSS/DM with severe dystrophic calcification had higher bone resorption than those with minimal or no evidence of calcification. Mean serum ionized Ca and urinary Ca excretion were both lower in those with severe calcinosis. Bone resorption was increased in patients with connective tissue disease and severe dystrophic calcification. Several antiresorptive agents were shown to be ineffective in limiting either bone turnover or clinical progression in one patient.
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References
D McCarty (1993) Systemic sclerosis (scleroderma), localized forms of scleroderma and calcinosis. DJ McCarty WJ Koopman (Eds) Arthritis and allied conditions, 12th ed. Lea & Febiger Pennsylvania 1253–1592
M Nishikai K Itoh A Sato (1992) ArticleTitleCalcinosis and the anticentromere antibody: its clinical, radiological and immunogenetic aspects. Br J Rheumatol 31 9–12 Occurrence Handle1:STN:280:By2C3M%2Fms1I%3D Occurrence Handle1730111
JL Leroux F Pernot P Fedou P Poubelle F Bonnel P Baldet F Blotman L Simon (1983) ArticleTitleUltrastructural and crystallographic study of calcifications from a patient with CREST syndrome. J Rheumatol 10 242–246 Occurrence Handle1:STN:280:BiyB2c7ltFY%3D Occurrence Handle6864678
T Kawakami C Nakamura H Hasegawa S Eda S Akahane T Yamazaki N Takasu (1986) ArticleTitleUltrastructural study of calcinosis universalis with dermatomyositis. J Cutan Pathol (Denmark) 13 135–143 Occurrence Handle1:STN:280:BimB2M7mtFU%3D
JB Lian M Skinner MJ Glimcher P Gallop (1976) ArticleTitleThe presence of gamma-carboxyglutamic acid in the proteins associated with ectopic calcification. Biochem Biophys Res Commun 73 349–355 Occurrence Handle1:CAS:528:DyaE2sXhsVyjtg%3D%3D Occurrence Handle999715
JB Lian LM Pachman CM Gundberg RE Partridge MC Maryjowski (1982) ArticleTitleGamma-carboxyglutamate excretion and calcinosis in juvenile dermatomyositis. Arthritis Rheum 25 1094–1100 Occurrence Handle1:STN:280:BiyD383lt1U%3D Occurrence Handle6982044
KG Saag R Emkey TJ Schnitzer et al. (1998) ArticleTitleAlendronate for the prevention and treatment of glucocorticoid-induced osteoporosis. N Engl J Med 339 IssueID5 292–299 Occurrence Handle10.1056/NEJM199807303390502 Occurrence Handle1:CAS:528:DyaK1cXlsVOitr4%3D Occurrence Handle9682041
. Geigy (1968) Documenta Geigy Scientific Tables. 6th ed. Geigy, Cheshire UK 516
MJ McKenna R Freaney (1998) ArticleTitleSecondary hyperparathyroidism in the elderly: means to defining hypovitaminosis D. Osteoporosis Int 8(suppl 2) S3–S6
EF Eriksen K Brixen P Charles (1995) ArticleTitleNew markers of bone metabolism: clinical use in metabolic bone disease. Eur J Endocrinol 132 IssueID3 251–263 Occurrence Handle1:CAS:528:DyaK2MXksVKju7k%3D Occurrence Handle7889170
SP Robins (1995) ArticleTitleCollagen crosslinks in metabolic bone disease. Acta Orthop Scand (suppl) 266 171–175 Occurrence Handle1:STN:280:BymC2cvitlU%3D
J Melkko S Kauppila S Niemi L Risteli K Haukipuro A Jukkola J Risteli (1996) ArticleTitleImmunoassay for intact amino-terminal propeptide of human type 1 procollagen. Clin Chem 42 947–954 Occurrence Handle1:CAS:528:DyaK28XjslKisbo%3D Occurrence Handle8665688
P Garnero PD Delmas (1993) ArticleTitleAssessment of the serum levels of bone alkaline phosphatase with a new immunoradiometric assay in patients with metabolic bone disease. J Clin Endocrinol Metab 77 1046–1053
P Garnero WJ Shih E Gineyts DB Karpf PD Delmas (1994) ArticleTitleComparison of new biochemical markers of bone turnover in late postmenopausal osteoporotic women in response to alendronate treatment. J Clin Endocrinol Metab 79 IssueID6 1693–1700 Occurrence Handle1:CAS:528:DyaK2MXislWiuro%3D Occurrence Handle7989477
HS Ju S Leung B Brown MA Stringer S Leigh C Scherrer K Shepard D Jenkins J Knudsen R Cannon (1997) ArticleTitleComparison of analytical performance and biological variability of three bone resorption assays. Clin Chem 43 IssueID9 1570–1576 Occurrence Handle1:CAS:528:DyaK2sXmtVyit7o%3D Occurrence Handle9299935
HN Rosen AC Moses J Garber DS Ross SL Lee SL Greenspan (1998) ArticleTitleUtility of biochemical markers of bone turnover in the follow-up of patients treated with bisphosphonates. Calcif Tissue Int 63 IssueID5 363–368 Occurrence Handle10.1007/s002239900541 Occurrence Handle1:CAS:528:DyaK1cXntV2lsbk%3D Occurrence Handle9799818
PA Kyd KD Vooght F Kerkhoff E Thomas A Fairney (1999) ArticleTitleClinical usefulness of biochemical resorption markers in osteoporosis. Ann Clin Biochem 36 483–491 Occurrence Handle10456211
P Ravn D Hosking D Thompson G Cizza RD Wasnich M McClung AJ Yates NH Bjarnason C Christiansen (1999) ArticleTitleMonitoring of alendronate treatment and prediction of effect on bone mass by biochemical markers in the early postmenopausal intervention cohort study. J Clin Endocrinol Metab 84 IssueID7 2363–2368 Occurrence Handle1:CAS:528:DyaK1MXks1agtb8%3D Occurrence Handle10404804
Y Aihara M Mori M Ibe T Kuriyama Y Takahashi C Shimizu H Shike T Mitsuda S Yokota (1994) ArticleTitleA case of juvenile dermatomyositis with calcinosis universalis — remarkable improvement with aluminium hydroxide therapy. Ryumachi 34 879–884 Occurrence Handle1:STN:280:ByqD1MbktVI%3D Occurrence Handle7801200
RG Berger GL Featherstone RH Raasch WH McCartney NM Hadler (1987) ArticleTitleTreatment of calcinosis universalis with low - dose warfarin. Am J Med 83 72–76 Occurrence Handle1:STN:280:BiiB28jns1U%3D
S Yoshida K Torikai (1993) ArticleTitleThe effects of warfarin on calcinosis in a patient with systemic sclerosis. J Rheumatol 20 1233–1235 Occurrence Handle1:STN:280:ByyA1c7msVQ%3D Occurrence Handle8371227
SE Moore AA Jump JD Smiley (1986) ArticleTitleEffect of warfarin sodium therapy on excretion of 4-carboxy-L-glutamic acid in scleroderma, dermatomyositis and myositis ossificans progressiva. Arthritis Rheum 29 344–351 Occurrence Handle1:STN:280:BimC1cnpsF0%3D Occurrence Handle3008764
AL Dolan D Kassimos T Gibson GH Kingsley (1995) ArticleTitleDiltiazem induces remission of calcinosis in scleroderma. Br J Rheumatol 34 576–578 Occurrence Handle1:STN:280:ByqA28%2Fkt1A%3D Occurrence Handle7633802
MJ Farah GM Palmieri JI Sebes MA Cremer JD Massie RS Pinals (1990) ArticleTitleThe effect of diltiazem on calcinosis in a patient with the CREST syndrome. Arthritis Rheum 33 1287–1293 Occurrence Handle1:STN:280:By%2BA2MfptF0%3D Occurrence Handle2390132
RL Cram R Barmada WB Geho RD Ray (1971) ArticleTitleDiphosphonate treatment of calcinosis universalis. N Engl J Med 285 1012–1013 Occurrence Handle1:STN:280:CS2D3cvlsFw%3D Occurrence Handle5094812
MJ McKenna (1992) ArticleTitleDifferences in vitamin D status between countries in young adults and the elderly. Am J Med 93 69–77 Occurrence Handle1:STN:280:By2A38fgsFI%3D Occurrence Handle1385673
R Freaney Y McBrinn MJ McKenna (1993) ArticleTitleSecondary hyperparathyroidism in elderly people: combined effect of renal insufficiency and vitamin D deficiency. Am J Clin Nutr 58 187–191 Occurrence Handle1:STN:280:ByyA3s7pvFI%3D Occurrence Handle8338046
SL Bowyer CE Blane DB Sullivan JT Cassidy (1983) ArticleTitleChildhood dematomyositis: factors predicting functional outcome and development of dystrophic calcification. J Pediatr 103 882–888 Occurrence Handle1:STN:280:BiuD28vgt1c%3D Occurrence Handle6644423
PG Hazen AE Walker JF Carney JJ Stewart (1982) ArticleTitleCutaneous calcinosis of scleroderma. Successful treatment with intralesional adrenal steroids. Arch Dermatol 118 366–367 Occurrence Handle10.1001/archderm.118.5.366 Occurrence Handle1:STN:280:Bi2B3crhs1c%3D Occurrence Handle7082035
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Dr. Eithne Murphy is the recipient of the Rohan Newman Scholarship, University College Dublin.
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Murphy, E., Freaney, R., Bresnihan, B. et al. Increased Bone Resorption and Failure to Respond to Antiresorptive Therapy in Progressive Dystrophic Calcification . Calcif Tissue Int 73, 433–440 (2003). https://doi.org/10.1007/s00223-002-2127-x
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DOI: https://doi.org/10.1007/s00223-002-2127-x