Unusual recurrent renal secondary hyperparathyroidism caused by hyperplastic autograft with supernumerary parathyroid adenoma: A case report

Abstract

Rationale: Secondary hyperparathyroidism (SHPT) is often complicated with chronic renal failure. Though the total parathyroidectomy (TPTX) with forearm autotransplantation (FAT) has been commonly used to treatment refractory renal SHPT, the recurrence of SHPT is not infrequent, resulting from hyperplastic autograft, remnant parathyroid tissues, and supernumerary parathyroid gland (SPG).

Patient concerns: A 67-year-old man undergoing TPTX+FAT 4 years previously for renal SHPT, who received regular hemodialysis with active vitamin D supplements of Rocaltrol treatment postoperatively, was admitted to our hospital with progressively elevated serum intact parathyroid hormone (iPTH) from 176 to 1266 pg/mL for 8 months and bilateral ankle joints pain for 1 month. Tc-sestamibi dual-phase imaging with single positron emission tomography (SPECT)/computed tomography (CT) revealed a nodule in suprasternal fossa, besides a nodule in autografted site, accompanied with intense radioactivity.

Diagnosis: Recurrent SHPT was easily diagnosed based on previous medical history, painful joints, increased serum iPTH level and positive findings of Tc-sestamibi imaging. Routine postoperative pathology showed that the nodules were consistent with an adenomatoid hyperplasic autograft and a supernumerary parathyroid adenoma in suprasternal fossa, respectively.

Interventions: Reoperation for removing nodules in suprasternal fossa and autografted site was performed 1 month later. Then regular hemodialysis 3 times a week with Rocaltrol was continued.

Outcomes: During 12 months of follow-up, the joints pain improved obviously and the serum iPTH level ranged from 30.1 to 442 pg/mL.

Lessons: Although rare, recurrent renal SHPT may be caused by a coexistence of both hyperfunctional autograft and SPG after TPTX+FAT. The Tc-sestamibi parathyroid imaging with SPECT/CT is helpful to locate the culprits of recurrent renal SHPT before reoperation. To prevent recurrence of renal SHPT, the present initial surgical procedures should be further optimized in patient on permanent hemodialysis.

Figure 1

^99mTc-sestamibi parathyroid dual-phase scan (A, B) revealed intense radioactivity at the middle-lower region of the right thyroid lobe (black arrow) and cervical contrast-enhanced computed tomography (C) showed multiple enhanced nodules behind thyroid gland (right lower: 3.0 × 2.5 cm, white arrow) before initial surgery. Hematoxylin-eosin stain (D, ×200) confirmed the nodules as parathyroid hyperplasia.

Figure 2

^99mTc-sestamibi parathyroid (A, B) and autograft (C) dual-phase scan and SPECT/CT (CT images: D, G; SPECT images: E, H; fusion images: F, I) at 2 hours after administration indicated a nodule (1.5 × 1.2 cm) in suprasternal fossa and a nodule (2.5 × 1.0 cm) in autografted site of right forearm, accompanied with intense radioactivity (arrow). SPECT/CT = single positron emission tomography/computed tomography.

Figure 3

Hematoxylin-eosin stain (×200) showed that the nodules were consistent with a supernumerary parathyroid adenoma (A) in suprasternal fossa and an adenomatoid hyperplasic autograft (B) in right forearm after reoperation, respectively.