Abstract
We report two adult cases of hypophosphatemic vitamin D resistant rickets with Ankylosing Spinal Hyperostois (ASH).
Case 1. A 52-year-old female had a short stature with bilateral bow legs and abnormalities of bone and joints since her childhood. At age 40, she manifested sensory disturbance in her lower extremities and gait disturbance. On admission, she had a peculiar short stature, 118cm in height, 35kg in weight. Laboratory examinations showed hypophosphatemia with normal serum calcium level, increased level of serum alkaliphosphatase and decreased renal reabsorption of phosphate. Radiological findings showed ossification in the pelvis, elbow, knee, especially, ASH with ossification of the posterior longitudial ligaments (OPLL) in the cervical.
Case 2. A 52-year-old female had essentially the same clinical history, physical characteristics and laboratory examinations. However, at age 30, she suffered from Filariasis and chyuria, so that, her laboratory examinations showed hypoprotenemia and hypocalcemia. Radiological examinations revealed ASH.
We guess that the cause of our cases with ASH may be in connection with hypophosphatemia, low level of (Ca×P) and high calcium retention rate for long periods. Our cases suggest that ASH may have a close relationship with abnormality of calcium and phosphate metabolism.
