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Inhibition of mediator release in systemic mastocytosis is associated with reversal of bone changes
Author(s) -
Graves Leland,
Stechschulte Daniel J.,
Morris David C.,
Lukert Barbara P.
Publication year - 1990
Publication title -
journal of bone and mineral research
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 1.882
H-Index - 241
eISSN - 1523-4681
pISSN - 0884-0431
DOI - 10.1002/jbmr.5650051104
Subject(s) - systemic mastocytosis , medicine , endocrinology , histamine , osteocalcin , mast cell , bone resorption , bone remodeling , alkaline phosphatase , ketotifen , chemistry , bone marrow , immunology , biochemistry , enzyme , asthma
A 59‐year‐old male presented with systemic mastocytosis with extensive skeletal involvement resulting in vertebral compression fractures and bone pain. Histomorphometric analysis of bone revealed increased mast cells, elevated static parameters of bone resorption, and low bone formation. Serum calcium, phosphorus, and alkaline phosphatase were normal; however, serum 1,25‐dihydroxyvitamin D 3 and osteocalcin levels were low. Histamine levels in plasma and urine were elevated. Following therapy with ketotifen, the patient had resolution of bone pain along with decreased flushing and pruritus. Elevated plasma and urine histamine levels normalized, as did 1,25‐dihydroxy vitamin D 3 and osteocalcin levels. Indices of low bone formation improved on therapy. Eroded surfaces improved but remained elevated. This case is the first demonstration that bone symptoms and histomorphometric change in systemic mastocytosis are reversed with inhibition of mast cell degranulation. The role of mast cells and their products in bone metabolism is poorly understood, but the therapy of bone disease in systemic mastocytosis should include inhibition of the release of mast cell products along with the use of histamine antagonist.