A Case of Marine-Lenhart Syndrome with a Negative TSH Receptor Antibody Titer Successfully Treated with a Fixed, Low Dose of I(131)

We herein describe a case of Marine-Lenhart syndrome with a negative TSH receptor antibody titer. A 75-year-old female presented to our hospital with malaise, palpitations, and mild fine tremors. She did not have any signs suggestive of Graves' ophthalmopathy, including conjunctival injection, periorbital edema, or proptosis. Her laboratory data were negative for thyroid autoantibodies, including anti-thyroid peroxidase antibodies, anti-thyroglobulin antibodies, and anti-TSH receptor antibodies (TRAb). Ultrasonography of the thyroid gland revealed a tumor in the right lobe. The remaining thyroid gland had an inhomogeneous and rough texture with a high color Doppler flow. I(123) scintigraphy disclosed a hot nodule in the right thyroid gland corresponding to the tumor detected on ultrasonography, suggesting Plummer disease. Furthermore, there was an increased uptake of radionuclide in the rest of the thyroid gland, despite the suppressed level of TSH and negative titer of TRAb, suggesting underlying Graves' disease. The present findings suggested a diagnosis of Marine-Lenhart syndrome with a negative TRAb titer. Treatment with 10 mCi of radioiodine was highly effective in treating hyperthyroidism in this case. A negative TSH receptor antibody titer does not necessarily rule out the existence of Graves' disease in patients with Plummer disease.