Parathyromatosis: a very rare cause of recurrent primary hyperparathyroidism - case report and review of the literature

Abstract

Parathyromatosis is a rare entity and usually appears as a consequence of the seeding on previous parathyroid surgery which was applied for the secondary hyperparathyroidism. A 63-year-old woman presented with a history of subtotal thyroidectomy 20 years ago and parathyroidectomy due to primary hyperparathyroidism (PHPT) four years ago. Imaging methods revealed multiple parathyromatosis foci on subcutaneous tissue of the neck. En-bloc resection was performed and pathological examination confirmed the diagnosis of parathyromatosis. After an uneventful 10 months, biochemical and radiological tests revealed recurrence on bilateral thyroid lodges. En-bloc resection was performed. The patient has remained well for 24 months after the second operation and has been followed-up with normal parathormone and serum calcium values. To the best of our knowledge, this report describes the twenty-first case of parathyromatosis in PHPT setting in the literature. It should be kept in mind that parathyromatosis may recur at different sites in the neck even in patients with PHPT.

Keywords: Parathyroidectomy; Parathyromatosis; Primary hyperparathyroidism; Recurrent parathyromatosis.

Figure 1

The parathyromatosis foci with ecchymotic appearance due to preoperative nodule aspiration, located at the edge of the old Kocher incision scar.

Figure 2

a) Subcutaneous localised parathyromatosis foci (arrows) with dimensions of 16 mm, 7 mm and 8 mm, respectively, on ultrasound. b) Images of Tc99m sestamibi scintigraphy at 0th and 20th minutes. At the 20th-minute, the initial physiological involvement is seen to be disappeared. c) Contrast-enhanced cervical tomography revealed contrast enhanced multiple parathyromatosis foci (arrows) on the anterior and medial sides of the right sternocleidomastoid muscle.

Figure 3

a) Incision parallel to the sternocleidomastoid muscle. b, c) En-bloc excision with adjacent skin and subcutaneous tissue of the parathyromatosis foci over the right sternocleidomastoid muscle.

Figure 4

a) Excised tissue, dimensions 5 × 2 cm. b) Multiple parathyromatosis foci in a dirty white colour on the macroscopic section of the specimen. c) Parathyroid tissue (star) in subcutaneous adipose tissue (haematoxylin and eosin × 4). d) Parathyromatosis foci (stars) separated with trabeculae (haematoxylin and eosin × 40).

Figure 5

Computed tomography of the neck revealed contrast-enhanced lesions (arrows) on both sides of the trachea. The radiologists described the lesions as the residual thyroid tissue on the right and as parathyroid tissue on the left.

Figure 6

a) Macroscopic appearance of the parathyromatosis foci excised from the right thyroid lodge (inferior) and left thyroid lodge (superior). b) Multiple cell mitoses (circles) were detected in parathyroid tissue (haematoxylin and eosin × 40). c) Presence of necrosis (arrow) was obtained on microscopic examination (haematoxylin and eosin × 40). d) A suspected vascular invasion image (arrow) was detected in one section (haematoxylin and eosin × 40).