We present the case of a 70-year-old woman who was examined due to osteoporosis and her analyses showed hypercalcemia and increased PTH. A cervical echography was conducted, showing bilateral parathyroid adenomas versus nodular parathyroid hyperplasia. These findings were confirmed by a cervical CAT. The study was completed with a gammagraphy which showed bilateral parathyroid hyperfunction in both lower poles, more obviously in the left one. The case was approached as a calcidiol deficit and strontium ranelate and vitamin D were prescribed.
Due to clinical persistence, treatment with calcimimetics was started in 20 months later and a new gammagraphy showed parathyroid hyperfunction in the left lower pole, compatible with a parathyroid adenoma in the left lobe, with the disappearance of capture in the right lower pole. Therefore, it was considered as a case of primary hyperparathyroidism secondary to a single lower left adenoma and left lower parathyroidectomy was carried out. The pathological examination reported findings compatible with parathyroid adenoma, with no cells suggesting malignancy.
Despite PTH immediate postoperative vàlues were normal, 1 week after surgery the patient continued to have high PTH levels. A new gammagraphy was taken, which found capture in the upper right parathyroid region, suggesting incipient parathyroid hyperfunction. The case was approached as a case of persistent primary hyperparathyroidism, secondary to an upper right adenoma. The patient refused surgery and calcium mimetics were started, with no improvement, so surgical treatment was proposed again. An upper right parathyroidectomy was performed 10 months later. The pathological examination reported a parathyroid adenoma again, with no signs of malignancy. However, after second adenoma was removed, the patient developed hypoparathyroidism of obscure origin because all the tests were repeated (US, CT scan and scintigraphy) and no abnormal findings were reported.
Sporadic multiglandular disease is a pathology for which little scientific evidence is available. A prevalence of 8-33% is described in the literature. Hence, two types of hyperparathyroidism after surgery are described: the persistent type and the recurring type. There is persistent hyperparathyroidism when PTH levels are not normalized in the first six months after the procedure. By contrast, recurrent hyperparathyroidism is defined as the type of parathyroidism which after surgery presents with a normalization of PTH levels in the first six months after surgery, and later presents with a new increase. Diagnosis is established by means of analysis when the patient presents with high PTH levels, and secondary hypercalcemia. This is due to the presence of a single benign adenoma in 85% of cases, to hyperplasia or multiple adenomas in 10-15%, and to parathyroid carcinoma in less than 1% of cases, associated with inherited genetic syndromes. As a differential diagnosis, treatment with lithium or thiazides should be discarded, as well as cases of familial hypocalciuric hypercalcemia. It should also be distinguished from secondary hyperparathyroidism resulting from kidney failure, kidney calcium loss, gastrointestinal disorders, or vitamin D deficit.
To support the analytic diagnosis, which defines this pathology, there are multiple imaging tests that can help. Non-invasive tests include cervical ultrasound and CAT marked with 9mTC sestamibi. Invasive tests include echo-guided percutaneous PTH aspiration and angiographic procedures.
It should be borne in mind that they all have high false positive and negative rates in patients previously operated on. As regards pathological results, several findings distinguish adenomas from parathyroid hyperplasia.
Finally, we will briefly review treatment options. In cases with mild symptoms, dietary measures can be taken: adequate hydration and monitoring. For moderate or serious cases, the first therapeutic option is surgery. If operating is not possible, there are various alternatives such as pharmacological treatment or the ablation of the anomalous parathyroid tissue.
Thus, we highlight the importance of making diagnostic-therapeutic decisions in a multidisciplinary committee comprising endocrinologists, surgeons, radiologists, and pathologists For these patients requiring new surgery, with the surgical complexity this entails, an expert surgeon must be available to obtain an optimal outcome. Long-term close monitoring is necessary to control potential complications.