Abstract
BackgroundThyrotropin-producing pituitary tumor is relatively rare. In particular, concurrent cases associated with Graves’ disease are extremely rare and only nine cases have been reported so far. We describe a case of a thyrotropin-producing pituitary adenoma concomitant with Graves’ disease, which was successfully treated.Case presentationA 40-year-old Japanese woman presented with mild signs of hyperthyroidism. She had positive anti-thyroid-stimulating hormone receptor antibody, anti-thyroglobulin antibody, and anti-thyroid peroxidase antibody. Her levels of serum thyroid-stimulating hormone, which ranged from low to normal in the presence of high levels of serum free thyroid hormones, were considered to be close to a state of syndrome of inappropriate secretion of thyroid-stimulating hormone. Magnetic resonance imaging showed a macropituitary tumor. The coexistence of thyrotropin-producing pituitary adenoma and Graves’ disease was suspected. Initial therapy included anti-thyroid medication, which was immediately discontinued due to worsening symptoms. Subsequently, surgical therapy for the pituitary tumor was conducted, and her levels of free thyroid hormones, including the thyroid-stimulating hormone, became normal. On postoperative examination, her anti-thyroid-stimulating hormone receptor antibody levels decreased, and the anti-thyroglobulin antibody became negative. The coexistence of thyrotropin-producing pituitary adenoma and Graves’ disease is rarely reported. The diagnosis of this condition is complicated, and the appropriate treatment strategy has not been clearly established.ConclusionsThis case suggests that physicians should consider the coexistence of thyrotropin-producing pituitary adenoma with Graves’ disease in cases in which thyroid-stimulating hormone values range from low to normal in the presence of thyrotoxicosis, and the surgical treatment of thyrotropin-producing pituitary adenoma could be the first-line therapy in patients with both thyrotropin-producing pituitary adenoma and Graves’ disease.
Highlights
This case suggests that physicians should consider the coexistence of thyrotropin-producing pituitary adenoma with Graves’ disease in cases in which thyroid-stimulating hormone values range from low to normal in the presence of thyrotoxicosis, and the surgical treatment of thyrotropin-producing pituitary adenoma could be the first-line therapy in patients with both thyrotropin-producing pituitary adenoma and Graves’ disease
Due to the increase in her Free triiodothyronine (FT3) and Free thyroxine (FT4) levels, and the decrease in her thyroid-stimulating hormone (TSH) level and TSH receptor antibody (TRAb) positivity, Graves’ disease was diagnosed, and anti-thyroid drug therapy was started in April 2010
Thyrotropin-producing pituitary adenoma (TSHoma) are a rare cause of hyperthyroidism, and they account for less than 1% of all pituitary adenomas [1, 2, 10]
Summary
The present case demonstrated that trans-sphenoidal surgery, which is the standard therapy for TSHoma, should be considered first-line therapy in patients with both TSHoma and Graves’ disease. When Graves’ disease becomes active, anti-thyroid therapy should be started as second-line therapy. A definitive operation is needed to prevent the subsequent treatment from becoming complicated. Abbreviations FT3: Free triiodothyronine; FT4: Free thyroxine; MRI: Magnetic resonance image; RTH: Resistance to thyroid hormone; SITSH: Syndrome of inappropriate secretion of TSH; TgAb: Anti-thyroglobulin antibody; TPOAb: Anti-thyroid peroxidase antibody; TR: Thyroid hormone receptor; TRAb: Anti-thyroid-stimulating hormone receptor antibody; TRH: Thyroidstimulating hormone-releasing hormone; TSH: Thyroid-stimulating hormone; TSHoma: Thyrotropin-producing pituitary adenoma
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