An Instructive Case Suggesting Cyclical Primary Hyperparathyroidism

Makita, Noriko; Iiri, Taroh; Sato, Junichiro; Fukumoto, Seiji; Okazaki, Taro; Yamazaki, Kimie; Obara, Takao; Fujita, Toshiro

doi:10.1507/endocrj.k05-097

Cited by 5 publications

(2 citation statements)

References 20 publications

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“…In parallel, over the last two decades the number of reported cases that do not follow this pattern has increased [ 3 ]. The first case of cyclic PHPT was recently reported [ 4 ] and sporadic cases of spontaneous resolution due to apoplexy of parathyroid adenomas have been reported more frequently [ 3 , 5 , 6 ]. In the latter situation most patients were submitted to early surgery or were followed for as long as 30 months but this did not include bone mass evaluation.…”

Section: Introductionmentioning

confidence: 99%

Parathyroid adenoma apoplexy as a temporary solution of primary hyperparathyroidism: a case report

et al. 2007

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IntroductionThe natural history of patients with spontaneous parathyroid necrosis is unknown. In this case report we describe the clinical course, laboratory, radiographic, bone densitometry tests, parathyroid ultrasonography and scintigraphy examinations of a patient performed over a period of eight years after she first presented with a sudden episode of spontaneous resolution of primary hyperparathyroidism (PHPT).Case presentationA 24-year-old woman with a clinical history and laboratory and radiographic tests compatible with PHPT suffered a sudden episode of cervical pain and presented with clinical evidence of hypocalcemia. Biopsy of a cervical nodule revealed necrotic material compatible with ischemia of the parathyroid. The follow-up of the patient presented four distinct phases: the first, which lasted two years, was compatible with a period of bone hunger during which it was necessary to introduce calcitriol and calcium carbonate. During this period, the patient showed bone mass gain. The second phase was characterized by normalization of calcium and parathyroid hormone levels and its end was difficult to define. During the third phase there was a recurrence of hypercalcemia associated with elevated parathyroid hormone (PTH) levels and loss of bone mass. The last phase corresponded to the interval after parathyroidectomy, which was characterized by normalization of serum levels of calcium and PTH, as well as bone mass gain.ConclusionThis case report indicates that spontaneous resolution of PHPT by adenoma necrosis is potentially temporary. Thus, in cases in which a conservative approach is chosen, clinical and laboratory follow-up is indispensable. Bone mass measurement is a useful tool in the follow-up of these cases. However, this option exposes the patient to a potential roller-coaster ride of bone mass gain and loss, whose long term consequences are still unknown.

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Section: Introductionmentioning

confidence: 99%

Parathyroid adenoma apoplexy as a temporary solution of primary hyperparathyroidism: a case report

et al. 2007

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“…However, other authors have reported that cyclical secretion of PTH by the parathyroid adenoma causes hypercalcemic crises, followed by a spontaneous decrease in PTH levels. However, the patient had renal insufficiency; hence, the cyclic PTH fluctuation may have been caused by pending chronic renal failure (10). In cases reported by Kwak et al, the PTH level rebounded after 6 months; hence, surgical treatment was provided; the parathyroid lesion in the permanent biopsy sample was confirmed to be a parathyroid adenoma (11).…”

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confidence: 99%

Unexpected remission of hyperparathyroidism caused by hemorrhage due to the use of fine-needle aspiration biopsy: two cases report

Kim

Lee

et al. 2021

Gland Surg

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Hyperparathyroidism is not a rare disease; if a parathyroid adenoma is confirmed, the treatment of choice is the surgical resection. Diagnostic use of fine-needle aspiration biopsy (FNAB) for histological confirmation in patients with hyperparathyroidism is controversial. And spontaneous remission of hyperparathyroidism caused by bleeding or infarction of the adenoma rarely occurs. Here we have reported two cases of hyperparathyroidism in which spontaneous remission occurred due to the use of FNAB for diagnosis. The remission was confirmed after surgical removal and pathological review of the adenoma.The first patient diagnosed with primary hyperparathyroidism (PHPT) had neck pain and severe swelling 4 days after FNAB, and spontaneous remission due to intracapsular hemorrhage was confirmed after surgery.In the second patient receiving hemodialysis treatment for end-stage renal disease, hyperparathyroidism spontaneously resolved after FNAB and the parathyroid hormone (PTH) levels normalized after surgery.The first patient maintained a normal level of PTH for 6 years, and the second patient received kidney transplantation 6 years after surgery, and the normal level of PTH was confirmed for 13 years. Caution is needed while performing FNAB for diagnosis of hyperparathyroidism and during decision making regarding whether to observe the patient or perform surgery after spontaneous remission due to bleeding or infarction.

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An unusual association of neuroendocrine tumors in MEN 1A

et al. 2011

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Multiple Endocrine Neoplasia type 1 is an autonomic dominant disease with a high degree of penetrance. It is characterized by combinations of over 20 different endocrine and nonendocrine tumors. A 25-year-old woman was referred for 1 year-evolution amenorrhea and bilateral galactorrhea. She also had fasting hypoglycaemia and hypercalcemia, and she was diagnosed of Multiple Endocrine Neoplasia type 1A. Resection of three parathyroid glands was performed showing hyperplasia of principal cells. Post-parathyroidectomy serum levels of calcium and intact PTH were normal but 3 years later serum calcium levels rose again. A 99mTc-sestamibi scan showed increased uptake in the low right area compatible with adenoma. After biochemical test showing probable insulinoma, somatostatin receptor scintigraphy showed a focal captation in head and body of pancreas. MRI found two nodules in the same localization. An antral gastrectomy, total pancreatoduodenectomy, colecistectomy and truncal vagotomy was performed and histopathologic examination revealed a combination of neuroendocrine tumors: gastrinomas, somastotinomas, glucagonomas and insulinomas. After surgery she started with tingling in fingers, toes and lips, and calcium levels was 5.9 mg/dl and PTH intact 3 pg/ml. A new 99m Tc-sestamibi scan showed no captation and cervical ultrasonography was normal. Now, 2 years later, she continues with normal calcium and i-PTH levels. This report represents an unusual case of MEN 1A with association of insulinomas, gastrinomas glucagonomas and somatostatinomas in the same patient.

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An Instructive Case Suggesting Cyclical Primary Hyperparathyroidism

Cited by 5 publications

References 20 publications

Parathyroid adenoma apoplexy as a temporary solution of primary hyperparathyroidism: a case report

Parathyroid adenoma apoplexy as a temporary solution of primary hyperparathyroidism: a case report

Unexpected remission of hyperparathyroidism caused by hemorrhage due to the use of fine-needle aspiration biopsy: two cases report

An unusual association of neuroendocrine tumors in MEN 1A

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