Hyperprolactinemia and
prolactinoma in patients with long-term
primary hypothyroidism have been recognized for decades. We report a case of 57-year-old female patient with
lingual thyroid and
cretinism who had a high serum
prolactin level (greater than 200 ng/ml) and a
pituitary tumor with suprasellar and parasellar extension. The
tumor regressed to a size undetectable by CT scan after 2 years of
thyroid hormone replacement
therapy, but complete normalization of the
hyperprolactinemia required additional
bromocriptine therapy. This patient showed generalized short metacarpal and phalangeal bones, calcification of the basal ganglia and dentate nuclei bilaterally, and subcutaneous calcification at both gluteal regions, while serum
calcium,
phosphorus and c-PTH levels were all normal. Thus in addition to short stature,
brachydactyly, a round face, and
obesity, which are related to
hypothyroidism, she also presented features uniquely mimicking the Albright's hereditary osteodystrophy seen in patients with
pseudohypoparathyroidism and
pseudopseudohypoparathyroidism. Since she had no family history of
pseudohypoparathyroidism and had a normal level of Gs alpha
protein on the membrane of the red blood cells, there is no evidence of
pseudopseudohypoparathyroidism. The cause of the ectopic calcification remains unknown.