Parathyroid apoplexy, the explanation of spontaneous remission of primary hyperparathyroidism: a case report
KeywordsAdenoma Parathyroid Gland Primary Hyperparathyroidism Minimal Invasive Surgery Parathyroid Adenoma
Primary hyperparathyroidism due to parathyroid adenoma represents an endocrine disease that is usually treated by surgical intervention (parathyroidectomy). In a very few patients, primary hyperparathyroidism can be spontaneously remit either by infraction or hemorrhage of the adenoma, a fact that is almost certain that will not lead to complete and definite cure. We describe a similar case of a 59-year-old male patient who underwent surgery for a cystic degeneration of a parathyroid adenoma, with substantial preoperative reduction of parathyroid hormone and calcium serum levels, and the diagnostic and treatment modalities are discussed, with a brief review of the current literature.
Primary hyperparathyroidism (PHPT) is a disease that is characterized by elevated calcium and parathyroid hormone (PTH) levels. The disease affects almost all organs and it is presented with a variety of symptoms and signs, ranging from asymptomatic PHPT to a potentially lethal course. The dominant cause of PHPT is the parathyroid adenoma (â‰ˆ85% of cases) and the optimal treatment is the surgical removal of the lesion. However, in the literature there have been a few cases reported, that haemorrhage or infraction in the lesion led to spontaneous remission of hyperparathyroidism. We report a similar case, where a patient was operated on for PHPT, with reduced PTH values postoperatively, compared to the previous ones, and a cystic adenoma with necrotic regions was found and removed.
The treatment of choice for patients with primary hyperparathyroidism due to parathyroid adenoma is conventional bilateral neck exploration and surgical removal of the lesion. Nowadays, minimally invasive techniques for the management of parathyroid adenomas are gaining ground, although they are not yet widely accepted to become the golden standard for the disease.
Classification of parathyroid apoplexy proposed by Nylen et al 
Adenoma with necrosis without hemorrhage
Asymptomatic, GI discomfort/pain, joint pain, cervical pain
Initial hypercalcemia- hypocalcemia
Adenoma with intracapsular hemorrhage and necrosis
Asymptomatic, GI discomfort, cervical pain, tetany, convulsions
Initial hyper- or hypocalcemia
Adenoma with extracapsular hemorrhage
Acute pain (neck to substernal), dysphagia, dysphonia, hoarseness, stridor, hypercalcemic crisis, widened mediastinum, visible hematoma or ecchymoses
Stable postoperative course, normocalcemia
Parathyroid apoplexy can lead to spontaneous remission of hyperparathyroidism, although in many cases the course of the phenomenon can be milder and can result only in asymptomatic reduction of calcium and PTH levels . Most of the reported cases presented with acute symptoms of hypocalcemia with or without tetania, resulting from the adenomaâ€™s necrosis and the incompetence of the remaining parathyroid glands to produce the necessary amount of PTH or hypercalcemia due to excessive PTH release, followed by a period of normocalcemia at first and finally recurrence of the disease. In rare cases, patients can present with signs and symptoms of massive cervical or mediastinal hemorrhage due to extracapsular hemorrhage of the adenoma, a condition that requires emergency neck exploration surgery. In the majority of the cases reported in the literature, surgical treatment was ultimately performed, while only a few patients were treated conservatively, under periodical clinical and biochemical follow-up [7, 8].
Based on the data, in cases of PHPT with spontaneous reduction of PTH and calcium serum levels, the hypothesis of “parathyroid apoplexy” can be supported only by a histopathological report of the adenoma’s necrosis and degeneration.
In our case, the episode of cervical pain and the automatic reduction of calcium and PTH levels, although they did not reach normal values, seem to support the hypothesis of incomplete necrosis of the parathyroid adenoma. This suggestion is also supported by the fact that Sestamibi scan after the episode did not show any clear evidence of an adenoma, while the histopathology report of the surgical specimen documented the cystic degeneration of the adenoma.
Authors’ suggestion, which comes in agreement with the previously reported data, is to perform parathyroidectomy rather than conservative treatment in all cases of suspected “parathyroid apoplexy”, at any stage of the disease, in order to have a definite diagnosis and permanent solution of PHPT. Moreover, authors’ belief is that the preferred surgical approach should be conventional bilateral neck exploration by a specialist surgeon in the endocrine surgery field, rather than minimal invasive surgery, an approach which offers the advantage of identification and examination of the remaining parathyroid glands.
Written informed consent was obtained from the patient for publication of this case report. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
EIE conceived the case report, analyzed and interpreted the patient data. MSP collected the data and was a major contribution in writing the manuscript. EP participated in drafting the manuscript. KJM operated on the patient, reviewed the manuscript, and gave final approval. NL participated in the operation, conceived the case report, and drafted the manuscript. All authors read and approved the final manuscript.
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