Recurrence hyperparathyroidism caused by synchronous parathyroid carcinoma and parathyromatosis in a patient with long-term hemodialysis

Abstract

Background Parathyroid carcinoma and parathyromatosis are very rare diseases in patients on hemodialysis. Its pathogenesis, clinical features, preoperative diagnosis, and surgery are challenging. We describe a rare case of recurrent hyperparathyroidism due to synchronous parathyroid carcinoma and parathyromatosis. Case presentation A 46-year-old Chinese woman was diagnosed with end-stage renal disease and received regular hemodialysis. Four years later, she experienced discomfort due to itching and was diagnosed with drug-resistant secondary hyperparathyroidism. Parathyroidectomy was performed, and her parathyroid hormone (PTH) levels were reduced. The pathology also revealed that the four nodules were parathyroid nodular hyperplasia without evidence of malignancy. Five years after surgery, the right subcutaneous nodule and left inferior nodule were detected by multiple imaging modalities, and the nodules were accompanied by recurrence itching and elevation of PHT. A complete resection of two nodules was performed, and the patient was diagnosed with parathyroid carcinoma and parathyromatosis. At 8 months postsurgery, her PHT and serum calcium levels were stable, and there were no signs of recurrence. Conclusions This is a rare case of synchronous parathyroid carcinoma and parathyromatosis in a patient with secondary hyperparathyroidism after parathyroidectomy. We suggest meticulous handling of parathyroid hyperplasia to avoid rupture and spillage during surgery, and precise pro-operation location by multiple imaging modalities is crucial for successful parathyroidectomy.

Coincidence or Causality: Parathyroid Carcinoma in Chronic Kidney Disease—Case Report and Literature Review

“…The diagnosis of PC is usually made after surgery on histopathological analysis. This is not ideal, as inappropriate resection and manipulation of the tumor may lead to parathyromatosis (loco-regional spillage and seeding of malignant parathyroid tissue) and recurrence, as reported in some cases [ 20 ]. Thus, precise pre-operation diagnosis and localization are desired, especially as complementary treatment options are limited; further, the prognosis in PC is poor if positive surgical margins or distant metastases are present [ 1 ].…”

“…Moreover, features that help identify PC in PHPT mimic the natural changes undergone by the parathyroid glands in SHPT (large dimensions, very high parathormone (PTH), high 99mTc-methoxyisobuthylisonitrile (99mTc-MIBI) scintigraphy uptake on scintigraphy, and low expression of vitamin D or calcium-sensing receptor) [ 11 , 12 , 16 , 18 , 19 ]. The management of these patients after diagnosis remains a dilemma, as reintervention poses the risk of parathyromatosis [ 20 ]. Nevertheless, reported outcomes differ significantly, even in the absence of “gold standard” surgery [ 14 , 15 ].…”