A case of mediastinal parathyroid adenoma with primary hyperparathyroidism

A 21-year-old man with a history of recurrent urolithiasis was admitted. Hypercalcemia was found and a diagnosis of primary hyperparathyroidism was made. Neck exploration was performed without success. Localization studies were done. Repeated selective arteriography revealed a mediastinal parathyroid adenoma. CT scan as well showed the adenoma in the anterior mediastinum. Ultrasonotomography, 201Tl-chloride and 131I scintigraphy with subtraction image and two venous samplings were negative. Mediastinal exploration with partial sternotomy was performed and a parathyroid adenoma was subsequently removed. Serum calcium and phosphorous levels were normalized, several postoperative days.     

Primary hyperparathyroidism due to mediastinal parathyroid adenoma

Of the 90 cases of primary hyperparathyroidism surgically treated in our department over the last decade (1975-1985), ten cases had a mediastinal parathyroidal adenoma. In only two of these patients was a median sternotomy required for excision of the mediastinal adenoma. Three of the ten patients underwent the initial operation in other institutions, having undergone a previous neck exploration. There were seven males and three females, ages ranging from 41-68 years. Six patients had nephrolithiasis, four had both renal stones and bone disease and two had peptic ulcer disease. One of them was operated on as an emergency because of hyperparathyroidism crisis with calcium levels of 15/16 mg%. Four patients were asymptomatic and had hypercalcemia detected by SMA screening. The calcium level ranged from 11.5-16.2 mg%. The phosphorus ranged from 1.6-2.8 mg% with a mean of 2.0 mg%. All ten patients had plasma PTH determination by radioimmunoassay, the values ranged from 1.5-3 times normal. In seven of the ten cases, the mediastinal parathyroid adenoma was localized within the thymus, the other three were adjacent to the great vessels, two to the aortic arch and one to the pulmonary artery-size ranging from 1.2-5.4 cm. Preoperative localization techniques: venous sampling in four cases; technetium scanning in three cases. No preoperative localization techniques were used in the other three cases. There was no mortality nor other significant postoperative complications.(ABSTRACT TRUNCATED AT 250 WORDS)     

A hemodialysis patient with secondary hyperparathyroidism in whom primary parathyroid adenoma was resected 27 years previously

We report here a dialysis patient with secondary hyperparathyroidism who had a history of parathyroidectomy for primary hyperparathyroidism 27 years previously. The patient was a 48-year-old male. In 1974, he was diagnosed as having primary hyperparathyroidism and an adenoma was completely resected in the Department of Urology, Osaka University Hospital. In 1997, he started hemodialysis for chronic renal failure by diabetic nephropathy. Since his intact-PTH was high, we started intravenous vitamin-D pulse therapy, but intact-PTH did not decrease. We could not detect any parathyroid glands by ultrasonography and 201TlCl-99mTcO4-scintigraphy around the thyroid gland. Finally, chest-CT and 99mTc-MIBI scintigraphy revealed a ectopic parathyroid gland in the mediastine, and the ectopic parathyroid gland was successfully resected in July, 2001. In order to distinguish whether the resected ectopic parathyroid gland was due to primary adenoma or secondary hyperplasia, we used an immunohistochemical technique to examine the expression of PRAD1/cyclin D1, Ki67, and p27 and sequence analysis of the MEN1 gene. As a result, the labeling index (LI) of PRAD1/cyclin D1 was 4, LI of Ki67 was 36, and LI of p27 was 257. Moreover, germline-mutation and somatic-mutation of MEN1 gene was not detected. These findings suggested that the resected parathyroid gland was a nodular hyperplasia of secondary hyperparathyroidism. In conclusion, immunohistochemical findings of parathyroid tissue and sequence analysis of MEN1 gene could be useful for the differential diagnosis of primary adenoma and secondary hyperplasia.     

Long-term outcome of unilateral parathyroid exploration for primary hyperparathyroidism due to presumed solitary adenoma

Abstract Unilateral cervical exploration for primary hyperparathyroidism (PHPT) remains controversial. Critics of unilateral exploration have argued that this surgical strategy results in an unacceptably large number of patients with persistent or recurrent hypercalcemia following the initial operation due to undetected additional parathyroid pathology on the contralateral, unexplored side of the neck. We have reviewed retrospectively a large cohort of patients who were submitted to scan-directed unilateral cervical exploration for PHPT following dual-isotope subtraction scintigraphy and according to a standard protocol. Between January 1, 1985 and June 30, 1999 a series of 184 patients underwent scan-directed unilateral neck exploration for PHPT. Following the initial operation three individuals (1.6%) demonstrated persistent hypercalcemia. At a mean follow-up of 59 months (range 6–168 months) none of the patients had developed recurrent hypercalcemia. Scan-directed unilateral cervical exploration represents a valid surgical strategy for a significant proportion of patients with PHPT; and in this group of patients it does not lead to an increased incidence of persistent or recurrent hypercalcemia. Electronic Publication     

A parathyroid cyst and an adenoma associated with primary hyperparathyroidism: report of a case

A case of a parathyroid cyst associated with an adenoma in a different gland is reported. A 55-year-old female was explored with preoperative diagnosis of primary hyperparathyroidism after endoscopic removal of a right ureteral stone. The operation revealed a cyst, 20 x 10 mm in size at the left lower gland, and a solid tumor, 32 x 12 x 7 mm in size and 1,300 mg in weight in the left upper gland. Histological examination disclosed a parathyroid cyst and an oxyphilic and chief cell adenoma, respectively. Postoperative course was uneventful including normalized serum Ca level. To our knowledge, 59 cases of parathyroid cysts have been reported in the Japanese literature, of which 31 were in the hyperparathyroid status. The most common causes were cystic degeneration of the adenomas. A parathyroid cyst with a concomitant adenoma in a different gland like our case is very rare. This is the first reported case in Japan and the sixth reported case in the world literature.     

Hypercalcaemia due to parathyroid adenoma and hypertrophic cardiomyopathy. A case report

A patient with hypercalcaemia due to a parathyroid adenoma, and hypertrophic cardiomyopathy (HCM) is described. Although both conditions are common, it is suggested that the association between HCM and parathyroid hypercalcaemia may be more frequent than is accounted for by coincidence.     

Water clear cell ectopic non-iatrogenic giant parathyroid adenoma in sternohyoid muscle with thyroid nodule and asymptomatic hypercalcemia due to primary hyperparathyroidism: Case report and literature review

BackgroundEctopic parathyroid gland is not uncommon, and is associated with primary hyperparathyroidism. Giant parathyroid adenoma (PA) and concurrent presence of enlarged thyroid nodule increases the probability of ectopic location. The combination of a giant PA that is ectopic (within the strap muscle) in the neck is very rare, especially in cases with no previous surgery. The rare histopathological findings of the current case, water clear cell parathyroid adenoma (WCCPA), could explain the patient's presentation, since it has low endocrine function.Case presentationA 56-year-old Qatari female on routine visit to primary health care physician for hypertension, was incidentally discovered to be hypercalcemic and was referred to the emergency department of our institution. Neck ultrasound showed a thyroid nodule on the left side, but ^99mTc-sestamibi scintigraphy identified a left PA. FNAC of the thyroid nodule showed that it was a colloid nodule. She underwent left hemithyroidectomy and excision of left PA. Intraoperatively, the PA was giant and in the sternohyoid muscle. Intraoperative monitoring of intact PTH (IOiPTH) confirmed successful excision.DiscussionEctopic giant parathyroid adenoma is rare especially with the intramuscular location in sternohyoid muscle in the neck without previous neck surgeries. The presence of thyroid nodule could be a precipitating factor for migration of the PA. Preoperative assessment with the radiological image is crucial for diagnosis but sometimes fail to localized the PA.ConclusionGiant asymptomatic PA with long standing low function before hyperfunctioning should raise the suspicion of WCCPA. If diagnosis is confirmed, metastasis from a clear cell renal cell carcinoma should be ruled out.     

Severe hypercalcemia due to acute primary hyperparathyroidism: role if parathyroidectomy

Grave hypercalcemia may complicate primary hyperparathyroidism. This clinical condition is potentially life-threatening, if it is untreated. The emergency therapy consist in rehydratation, stimulation of diuresis and somministration of biphosphonates. A urgent surgery is required if conservative therapy is not successful. The Authors herein present a consecutive series of 6 cases with acute hypercalcemic crisis due primary hyperparathyroidism, successful treated with urgent parathyroidectomy after conservative treatment.     

A case of primary lung cancer complicated with post-operative intractable pulmonary fistula

The case was a 76-year-old male. After thoracoscopic left upper lobectomy against primary lung cancer with poor risks such as complication of systemic lupus erythematosus (SLE) and pulmonary emphysema, oral steroid treatment, heavy smoker, decreased renal function, the patient was complicated with intractable pulmonary fistula and MRSA pyothorax. Intracavitary administration of albumin preparation and fibrin glue (Beriplast P) was effective against pulmonary fistula though it was not helpful in the pleurodesis which was conducted 7 times. Teicoplanin (Targosid) was effective against MRSA pyothorax and maintained the renal function.     

Intrathyroid cystic parathyroid adenoma: a rare case of hyperparathyroidism

A case is presented of a 52-year-old man with a swelling in the neck and dysphagia resulting from an intrathyroid cystic parathyroid adenoma. Hypercalcaemia and unusually high parathyroid hormone levels were detected preoperatively. The adenoma was removed by partial hemithyroidectomy. The literature is reviewed.     

A case of lung cancer in which FDG-PET was useful for preoperative diagnosis

The patient was 48-year-old woman. The patient was referred to our hospital due to detection of an abnormal shadow on a chest X-ray image during a health checkup. The results of a CT scan morphologically suggested a benign tumor, but a diagnosis could not be made even after bronchoscopic cytology. FDG-PET revealed accumulation at the tumor site suggestive of malignancy, and an operation was performed. Prompt pathological examination showed that the tumor was adenocarcinoma, and right lower lobectomy and lymphnode dissection were performed. FDG-PET is thought to be useful for diagnosis in cases of lung tumor in which a preoperative diagnosis can not be established.     

Recurrence of primary hyperparathyroidism following spontaneous remission with intracapsular hemorrhage of a parathyroid adenoma

A few patients with primary hyperparathyroidism (PHPT) have a chance of spontaneous remission by either infarction of or hemorrhage into or around the parathyroid adenoma. In most cases, biochemical derangements associated with PHPT are permanently improved after spontaneous remission. Here we report a case with a recurrence of PHPT 4 months after spontaneous remission with acute intracapsular hemorrhage of parathyroid adenoma. In the literature, only two cases have been reported to have experienced a recurrence of clinical features of PHPT after infarction but not hemorrhage of parathyroid adenomas. Thus, the spontaneous remission of biological derangements in PHPT upon hemorrhage or infarction of parathyroid adenoma could be temporary. One should carefully observe such patients thereafter.     

Unilateral parathyroidectomy in hyperparathyroidism due to single adenoma.

As a general principle in the treatment of primary hyperparathyroidism due to single adenoma, unilateral parathyroidectomy was applied to 50 patients and compared with another group of 50 conventionally explored patients. Twenty-five patients were explored only on the "adenoma" side. The other 25 patients were explored on both sides, avoiding biopsies at the first. In the conventionally explored patients, the adenoma was removed and one to three normal glands were biopsied. Oil-red-O technique was used in the intraoperative microscopical examination. The patients in whom the operation could be limited to the "adenoma" side had a statistically more favorable situation concerning early postoperative hypocalcemia, length of operation time, and need for calcium and vitamin D substitution. The principle of unilateral parathyroidectomy in conjunction with intraoperative oil-red-O staining technique is advocated in hyperparathyroidism due to single adenoma because it offers more reliable peroperative distinction between uni- and multi-glandular involvement, reduced operation time, decreased risk for complication, reduced early hypocalcemia, and more favorable technical conditions for reoperation.     

Surgical strategy in hyperparathyroidism due to solitary adenoma.

Based on the postulate that parathyroid adenoma is practically always a solitary lesion, unilateral parathyroidectomy including the homolateral normal parathyroid was applied as a principle in the treatment of this form of primary hyperparathyroidism. The exploration was confined to the adenoma side if this was the first to be explored. Intraoperative oil-red-O staining of frozen sections was used to exclude the possibility of a multiglandular involvement. This principle was applied in a consecutive series of 102 patients operated for hyperparathyroidism from 1977 to 1981 and diagnosed as parathyroid adenoma. In 43 patients where the abnormal gland was found on the side explored first, unilateral parathyroidectomy was performed on that side, avoiding exploration of the contralateral side. In 45 patients where normal glands were found on the side first explored, unilateral parathyroidectomy was performed on the contralateral side. In 14 patients other types of operations were performed as the above-mentioned principle could not be achieved. At follow-up 1 to 5 years after surgery, no cases of hypocalcemia were recorded. The results of the different operations were compared as to early and late hypocalcemia. Early hypercalcemia was more pronounced after a bilateral exploration. Two of the patients who had an atypical operation had a permanent need for vitamin D in order to maintain an adequate serum calcium level. Surgical principles for various possible exploratory findings are outlined. These are based upon the idea of performing a unilateral parathyroidectomy whenever intraoperative oil-red-O staining excludes multiglandular involvement as a cause for the hyperparathyroidism.     

Mortality in patients surgically treated for primary hyperparathyroidism due to solitary adenoma

In a multicentre study including 5 surgical departments in Europe and USA the results of surgical treatment in primary hyperparathyroidism (HPT) due to single adenoma were analysed. At long term follow-up 60 out of 282 patients operated on for solitary parathyroid adenoma were found to be dead. The average age at operation was 69.2 years and the time of survival after surgery 3.9 years. The cumulative relative survival in patients aged 60-64 years was reduced by 18% (P less than 0.01) as compared to a control group corrected for nationality, age and gender. In patients above the age of 65 no such reduction could be demonstrated. The operative mortality was 1% and attributed to cardiac failures. Late mortality was caused by cardiovascular conditions in 52% and by malignant disease in 15%. None of these figures were statistically different from the age, gender and nation corrected control group. Among the miscellaneous causes of death were two patients who committed suicide during the first postoperative year. The results indicate that surgery for parathyroid adenoma can be performed with low morbidity and mortality.     

A case of sellar germinoma which was misdiagnosed as pituitary adenoma]

The authors reported a rare case of sellar germinoma which was misdiagnosed as nonfunctioning pituitary adenoma. A 32-year-old woman was admitted to our hospital because of amenorrhea and disturbance of left visual acuity. She had become amenorrhagic after her second delivery two years before. Neurological examination revealed she was normal except for diminished left visual acuity (Rt. = 1.2, Lt. = 0.5). The general condition was good. Urine volume and its specific gravity were within normal range. Endocrinological examination showed hyperprolactinemia (PRL 72 ng/ml) accompanied with impairment of GH, TSH, LH and FSH's reserve. Basal levels and reserve of the blood cortisol were normal. AFP and hCG were within normal range. CT scan revealed a homogenously enhanced intrasellar tumor which had a suprasellar portion (vertical length: 15 mm). T1 weighted MRI revealed low intensity tumor, and T2 weighted image revealed high intensity tumor. Sagittal MR image with gadolinium enhancement showed the pituitary gland anterior to the tumor. Transsphenoidal removal was performed. The histological diagnosis was pure germinoma. After the operation, the intracranial and spinal disseminations were disclosed. Complete neuraxis irradiation resulted in the complete remission of the tumor. Sellar germinoma without diabetes insipidus is considered to be very difficult to diagnose preoperatively. However, the authors proposed that anterior shift of the pituitary gland in sagittal MR image may be a clue to the diagnosis of sellar germinoma.     

Recurrent secondary hyperparathyroidism due to implanted parathyroid tissue: a case report

Hyperparathyroidism is a common complication of chronic renal failure. Although many patients can be managed by conservative measures, surgery is sometimes necessary. One of two operations can be performed: subtotal parathyroidectomy or total parathyroidectomy with reimplantation of parathyroid tissue into muscle. A case is presented of a patient who underwent the first and then the second of these procedures for recurrent hyperparathyroidism. A further recurrence was found to be caused by the implanted parathyroid tissue in a forearm muscle, requiring a third procedure for control of the disorder.