Parathyroid crisis, also known as a parathyroid storm, is a rare and serious complication of primary hyperparathyroidism. Four cases are reported here in which patients presented to hospital with general complaints due to hypercalcemia secondary to hyperparathyroidism. Blood test results upon admission showed high levels of serum calcium and parathyroid hormone, and medical treatment initiated to lower the calcium level was ineffective. After relevant investigations, each patient underwent surgical exploration of the parathyroid glands, followed by excision of a pathological parathyroid tumor. There was a prompt decrease in parathyroid hormone level immediately after surgery. Histology reports revealed that patients had parathyroid adenoma. All patients recovered after surgery, with serum calcium levels restored back to normal and with resolution of all symptoms of hypercalcemia. This report illustrates how often this disease is initially misdiagnosed, and how prompt appropriate surgical treatment provides the best outcome for the patient.
parathyroid crisis; hypercalcemia; hyperparathyroidism; hypercalcemic crisis; parathyroid adenoma
Hypercalcemia can be associated with vitamin D (1,25(OH)2D3) -mediated granulomatous disorders in addition to primary hyperparathyroidism (PHPT). Although most patients with granulomatous disease-related hypercalcemia are asymptomatic, symptoms and signs of chronic hypercalcemia can occur. There are many reports about co-presentation of a parathyroid adenoma and a granulomatous disorder in the literature. However, granulomatous inflammation within a parathyroid adenoma is very rare.
A 50-year-old Caucasian woman presented with generalized bone pain and muscular weakness. Biochemical findings suggested PHPT. She underwent excision of an enlarged right inferior parathyroid gland. Histopathological analysis revealed features of a parathyroid adenoma with foci of epithelioid non-caseating granulomas. The etiology of the granulomatous infiltration could not be determined. She is still normocalcemic at the ninth month after surgery and is being followed for the possible manifestation of an occult disease.
Granulomatous infiltration of a parathyroid adenoma is a rare condition. Pathological examination of the excised adenoma is the only way to diagnose the underlying occult granulomatous disorder. Clinicians should also consider persistent hypercalcemia to be a possible indicator of concomitant parathyroid adenoma.
To describe a patient presenting with the rare constellation of synchronous parathyroid carcinoma, parathyroid adenoma, and papillary thyroid carcinoma.
Review of clinical presentation, diagnostic workup, surgical management, and pathological findings, and review of pertinent literature.
The patient was a 59-year old man who presented with severe clinical manifestations of longstanding primary hyperparathyroidism (HPT), serum calcium of 14.4 mg/dL, and parathyroid hormone level of 2,023 pg/ml. The patient was found to have a 3.4 cm parathyroid carcinoma on the left side and a 3.2 cm papillary thyroid carcinoma on the right side. In addition, a 917 mg parathyroid adenoma was found on the right side.
Synchronous parathyroid and thyroid carcinomas are extremely rare. Our patient is the first documented case with a parathyroid adenoma in addition to synchronous parathyroid and thyroid carcinomas. The presence of concurrent parathyroid carcinoma and adenoma can cause diagnostic confusion and should be considered in patients presenting with severe HPT. Any concomitant thyroid nodules need to be investigated to rule out thyroid carcinoma.
Parathyroid adenoma is the most common cause of primary hyperparathyroidism. Preoperative serum calcium and intact-parathyroid hormone levels are the most useful diagnostic parameters that allow differentiating primary hyperparathyroidism from non-parathyroid-dependent hypercalcemia. Parathyroidectomy is the definitive treatment for primary hyperparathyroidism. Approximately 5% of patients who underwent parathyroidectomy present with persistent or recurrent hyperparathyroidism due to ectopic localization of the adenoma. Functioning oxyphil parathyroid adenoma is an uncommon histological form, seldom causing primary hyperparathyroidism. Parathyroid adenoma with hypercalcemia exhibiting normal parathyroid hormone level is rare. An incidence of 5% to 33% has been documented in the literature; no etiologic explanation has been given. In 1987, parathyroid-hormone-related peptide was isolated as a causative factor of humeral hypercalcemia of malignancy. The presence of parathyroid-hormone-related peptide in parathyroid tissue under normal and pathological conditions has been described in the literature; however, its role in causing hyperparathyroidism has not yet been defined.
We present a case of persistent hypercalcemia with a normal level of intact-parathyroid hormone due to a substernal parathyroid adenoma, treated with radioguided parathyroidectomy. The final histological diagnosis was oxyphil adenoma, positive for parathyroid-hormone-related peptide antigens.
In clinical practice, this atypical biochemical presentation of primary hyperparathyroidism should be considered in the differential diagnosis of hypercalcemia. The parathyroid-hormone-related peptide should be considered not only in the presence of malignancy.
Primary hyperparathyroidism (PHPT) is not an uncommon endocrine disorder. However, acute primary hyperparathyroidism, or parathyroid crisis (PC), is a rare clinical entity characterized by life-threatening hypercalcemia of a sudden onset in patients with PHPT. We describe a patient with PC who presented with acute worsening of depressive symptoms, nausea and vomiting, and required emergency surgery. Serum calcium, alkaline phosphatase, and parathyroid hormone were elevated and serum phosphorus was low. An emergency hemithyroidectomy was performed because of none medical control of hypercalcemia. A giant intrathyroidal parathyroid adenoma was diagnosed. PHTP can be a life-threatening situation for patients, requiring immediate surgical treatment. A giant intrathyroidal parathyroid adenoma is an uncommon cause of PC.
Acute primary hyperparathyroidism; hemithyroidectomy; intrathyroidal parathyroid adenoma; parathyroid crisis; parathyroid glands
Parathyroid carcinoma is a very rare cause of hyperparathyroidism. The diagnosis is usually established on histopathological grounds of capsular and vascular invasion, but a potential clue to the diagnosis is also offered by the severity of clinical profile, abrupt onset of symptoms, and a high degree of hypercalcemia and raised serum parathyroid hormone (PTH). We report a case of an elderly female with a prolonged history of generalized weakness and bone pain along with bilateral renal calculi, classical bony lesions, and a high serum calcium and PTH level who underwent a right inferior parathyroidectomy considering a parathyroid adenoma as our diagnosis. However, the biopsy report was consistent with a parathyroid carcinoma, and so, she was further subjected to an ipsilateral hemithyroidectomy as a completion procedure. So, we would like to emphasize that its preferable to have a high index of suspicion for parathyroid carcinoma when these clues are present, than to miss the opportunity for surgical cure in the first go by failing to consider it in the differential diagnosis.
Carcinoma; parathyroid; rare endocrine malignancy
Hyperparathyroid crisis is a rare manifestation of parathyroid disease. We present the case of a 53-year-old gentleman with a review of the current literature. He presented in acute renal failure with epigastric pain and vomiting. His serum-corrected calcium (CCa2+) was raised at 5.2 mmol/L, in addition to a massively raised parathyroid hormone (PTH) level (3957 ng/L). Ultrasound studies of the neck revealed a 2 cm well-defined mass inferoposterior to right thyroid lobe. CT scans of the neck showed a normal mediastinum and confirmed no associated lymphadenopathy. Having undergone medical resuscitation for 9 days, a neck exploration revealed a cystic mass, which was excised. Histological investigations revealed a 9.25 g, cystic parathyroid adenoma with no features of malignancy. His PTH and CCa2+ returned to normal postoperatively. This suspicious presentation of benign disease, including a marked elevation in PTH, highlights the challenges facing the endocrine surgeon in dealing with parathyroid disease.
Parathyroid adenoma associated with a thymoma is a rare clinical entity and few cases have been reported in the literature. This association can be explained by the common embryologic origin of the parathyroid glands and the thymus. These patients may present only with clinical signs and symptoms of primary hyperparathyroidism making it difficult to suspect the coexistence of thymoma.
PRESENTATION OF CASE
A 68-year-old female with medical history of primary hyperparathyroidism underwent surgical excision of a single parathyroid adenoma. Intra-operatively a small mass was detected in the proximity of the parathyroid adenoma and was also removed. Pathologic examination revealed parathyroid adenoma as suspected pre-operatively whereas the small mass excised was identified as a type A thymoma. The postoperative course of the patient was favourable, with normal serum calcium levels and in six months follow-up the patient is symptom-free and with no recurrence.
Objective of this study is to report the rare case of a female adult with a parathyroid adenoma and a coexisting thymoma in order to underline the importance of these two pathologies and the ideal treatment that should be followed according to the latest records.
Surgical removal of parathyroid adenoma is the treatment of choice for primary hyperparathyroidism, with complete recovery and no postoperative complications. Thymoma may be associated with primary hyperparathyoridism, with or without clinical signs and symptoms of myasthenia gravis and this is a rare clinical entity as very few cases have been described worldwide. To our knowledge this is the first case reported in Greece.
Thymoma; Parathyroid; Adenoma; Hyperparathyroidism; Hypercalcemia
To report the use of immunohistochemical staining for parafibromin, APC and galectin-3 to evaluate the malignant potential of the resected parathyroid specimen in a patient initially presenting with primary hyperparathyroidism secondary to four-gland hyperplasia who subsequently developed metastatic parathyroid carcinoma.
We describe a patient with primary hyperparathyroidism who underwent a 3 gland resection of hypercellular parathyroids with post-operative normalization of her serum calcium and parathyroid hormone levels. She returned four years later with recurrent hypercalcemia and underwent partial resection of her remaining hypercellular gland without improvement of her hypercalcemia. Selective venous sampling localized the source as draining into her azygous vein, and she was ultimately diagnosed with metastatic parathyroid carcinoma.
Immunohistochemical staining for parafibromin, APC and galectin-3 suggested the malignant potential of the atypical adenoma removed during the patient’s original operation, which is believed to be the source of her metastatic disease. Access to this information by the treating surgeon may have prompted a more extensive en bloc resection or more vigilant follow-up that could have altered the patient’s clinical course.
Immunohistochemical staining for parafibromin, APC and galectin-3 can be used to help distinguish the source of metastatic disease in parathyroid carcinoma. Selective venous sampling may help localize metastatic parathyroid carcinoma when it is otherwise not apparent.
parathyroid carcinoma; hyperparathyroidism; hypercalcemia; immunohistochemistry; parafibromin; APC; galectin-3
Hypercalcaemic hyperparathyroid crisis is a rare but life-threatening complication of primary hyperparathyroidism. Parathyroid carcinoma is a rare malignancy with an incidence of 0.5% to 4% of all reported cases of primary hyperparathyroidism.
We report the case of a 60-year-old Caucasian man with hypercalcaemic hyperparathyroid crisis associated with parathyroid carcinoma. He presented with a classic hypercalcaemic syndrome and his serum calcium and parathyroid hormone levels were at 4.65 mmol/L and 1743 ng/L, respectively. He initially presented with a two-week history of weakness and lethargy and a one-week history of vomiting, polyuria and polydipsia. An emergency left thyroid lobectomy and left lower parathyroidectomy were performed. There was a prompt decrease in his parathyroid hormone level immediately after surgery. Histology revealed that our patient had a 4-cm parathyroid carcinoma.
In patients with parathyroid carcinoma, the optimal surgical treatment is en bloc resection with ipsilateral thyroid lobectomy and removal of any enlarged or abnormal lymph nodes. Surgery is the only curative treatment. In our patient, prompt surgical intervention proved successful. At six months the patient is well with no evidence of disease recurrence. This case highlights the importance of considering a hyperparathyroid storm in the context of a parathyroid carcinoma. Parathyroid carcinoma is a rare entity and our knowledge is mainly derived from case reports and retrospective studies. This case report increases awareness of this serious and life-threatening complication. This report also illustrates how prompt and appropriate management provides the best outcome for the patient.
A 60-year-old woman presented with abdominal pain and weight loss and was found to have serum calcium of 15.0 mg/dl. Serum parathyroid hormone-related peptide (PTHrP) returned elevated. Imaging suggested bilateral mature cystic teratomas. Her hypercalcemia was treated initially with intravenous saline, as well as intramuscular and subcutaneous calcitonin. She underwent total abdominal hysterectomy with bilateral salpingo-oophorectomy, and final pathology revealed malignant Brenner tumor in association with a mature cystic teratoma. Her postoperative PTHrP returned less than assay, and her total and ionized calcium fell below normal, requiring supplemental calcium and vitamin D. At follow-up one month after discharge, her calcium had normalized. We present the first reported case of hypercalcemia occurring in association with a malignant Brenner tumor. Malignancy-associated hypercalcemia occurs via four principal mechanisms: (1) tumor production of PTHrP; (2) osteolytic bone involvement by primary tumor or metastasis; (3) ectopic activation of vitamin D to 1,25-(OH)2 vitamin D, and (4) ectopic production of parathyroid hormone. PTHrP-mediated hypercalcemia is the most common mechanism and was responsible in this case. In patients with paraneoplastic hypercalcemia who undergo surgical treatment, close monitoring and management of serum calcium is necessary both pre- and postoperatively.
Hypercalcemia; Parathyroid hormone-related peptide; Mature cystic teratoma; Malignant Brenner tumor
Aim. Despite an incidence of parathyroid “incidentalomas” of 0.2%–4.5%, only approximately 135 cases have been reported in the literature. We present eight patients in whom an incidental abnormal parathyroid gland was found during routine thyroid surgery. We have reviewed the literature and postulate whether these glands could represent further evidence of a preclinical stage of primary hyperparathyroidism.
Methods. A retrospective analysis of all 236 thyroid operations performed by a single surgeon was performed to identify patients in whom abnormal parathyroid tissue was removed at surgery.
Results. 8/236 patients (3.39%) had a single macroscopically abnormal parathyroid gland removed and sent for analysis. Seven patients were found to have histological evidence of a parathyroid adenoma or hyperplasia. None of the patients had abnormal serum calcium detected preoperatively. Postoperatively, four patients had normal calcium, three had temporary hypocalcaemia and one refused followup. No patients had recurrent laryngeal nerve impairment.
Conclusions. Despite the risk of removing a histologically normal gland, we believe that when parathyroid “incidentalomas” are found during surgery they should be excised and sent for histological analysis. We have found this to be a safe procedure with minimal morbidity to the patient.
As the natural history of primary hyperparathyroidism is better understood, these glands found in normocalcaemic patients may in fact represent the early or preclinical phase of the disease. By removing them at the original operation, the patient is saved redo neck surgery with its high complication rate as or when clinically apparent primary hyperparthryoidism develops in the future.
The case is described of a woman who died with a functioning parathyroid carcinoma 19 years after removal of two parathyroid tumours, considered at the time to be benign. Following operation hyperparathyroidism subsided, with a short period of hypocalcaemia, and severe osteitis fibrosa cystica healed. Five years before death progressive renal failure developed with normal and later raised serum calcium levels. At necropsy a parathyroid carcinoma infiltrated the thyroid at the site of the previous operation and there was a solitary hepatic metastasis. A remaining parathyroid gland was nodularly hyperplastic and the bones showed evidence of active osteitis fibrosa. The relationship between parathyroid hyperplasia and neoplasia is discussed. Acceptable cases of parathyroid carcinoma are rare, but carcinoma is an important cause of recurrent hyperparathyroidism even months or years after removal of an `adenoma'.
Primary hyper-parathyroidism is attributed by a solitary parathyroid adenoma in 80-85% cases and mostly are non-palpable and needs imaging for localization. Cystic degeneration of parathyroid adenoma is rarely seen in primary hyperparathyroidism. We are presenting a case of giant cystic functioning parathyroid adenoma with associated localized co-morbids.
Cystic parathyroid adenoma; internal jugular vein thrombosis; primary hyperparathyroidism; Tc-99m methoxy isobutyl isonitrile
In the treatment of secondary hyperparathyroidism of chronic kidney disease, calcimimetics - allosteric modulators of the calcium-sensing receptor - inhibit glandular hyperplasia and significantly reduce circulating parathyroid hormone levels. They have a major impact on the management of secondary hyperparathyroidism.
We present the clinical case of a 41-year-old Caucasian man undergoing chronic hemodialysis, who had a parathyroidectomy to treat severe secondary hyperparathyroidism resistant to cinacalcet treatment. Preoperatively, 24 months after high-dose cinacalcet hydrochloride, we observed a persistently elevated intact parathyroid hormone serum level, and detected clear parathyroid gland hyperplasia regression on ultrasound. We performed a three-gland parathyroidectomy, which was assumed to be total, associated with a hemithyroidectomy. Our patient then entered a hypoparathyroid state. A histopathological examination showed that the removed parathyroid glands were of small size, with a total weight of 1g, associated with a multifocal small papillary thyroid cancer.
In the management of secondary hyperparathyroidism, cinacalcet hydrochloride effectively reduces total parathyroid gland hyperplasia. However, a persisting elevated intact parathyroid hormone serum level may be observed, demonstrating that reduced parathyroid hyperplastic tissue may still be associated with severe secondary hyperparathyroidism. Even if calcimimetics are very effective in secondary hyperparathyroidism treatment, further studies are necessary for a better understanding of their actions.
Parathyroid carcinoma is a rare cause of primary hyperparathyroidism and may be associated with significant disease related morbidity and mortality. Preoperative diagnosis remains a challenge, which may jeopardize appropriate and successful patient treatment.
We report a case of parathyroid carcinoma diagnosed in a 60-year-old woman that presented with a tender nodule located at the left lower thyroid pole and had been present for several years. Ultrasound examination revealed a 2.7 × 1.6 × 2.7 cm mass within the lower left lobe of the thyroid with cystic and solid areas. Lab measurement of the intact PTH level revealed it to be three times the upper limit of normal and the serum calcium level was within normal limits. A left thyroid lobectomy and isthmusectomy was carried out. Histopathological evaluation was diagnostic for a parathyroid carcinoma. At greater than two years of follow-up, the patient has had no evidence of disease recurrence and her serum PTH and calcium levels have remained within normal.
Parathyroid carcinoma is a rare endocrine tumor which must be considered in the differential diagnosis of a nodular thyroid mass. En bloc resection remains the treatment of choice for this malignancy. Disease prognosis is influenced by the extent of the initial resection, the presence of metastases, and adequate long-term follow-up.
A patient admitted for acute abdomen was incidentally found with elevated serum calcium level. In surgery, under conservative treatment of the hypercalcemia, a perforated duodenal ulcer was found and simple closure was performed. Postoperatively, calcium level continued to rise, parathyroid hormone was elevated and ultrasonographic examination showed a lesion in the right anterior neck, while serum gastrin level was normal, thus documenting the diagnosis of primary hyperparathyroidism. Conservative treatment had no effect on calcium level and the patient was subjected to emergency neck exploration, where a large parathyroid adenoma was removed. After surgery, calcium and PTH levels were normalized and the patient was discharged on the 5th postoperative day. Peptic ulcer and its complications are usual manifestations of primary hyperparathyroidism, with or without increased gastrin level. On the other hand, cases of a perforation of peptic ulcer as the first clinical manifestation of primary hyperparathyroidism are extremely rare.
Peptic ulcer; Primary hyperparathyroidism; Parathyroidectomy
The case history of a 40-year-old woman with a fatal haemorrhage from a duodenal ulcer, jejunitis, renal lithiasis, hypertrophy of the adrenal glands, multiple adenomas of the pancreas, and three adenomas of both the parathyroid and pituitary glands is reported. Though there was histological evidence, in the bones, of hyperparathyroidism, the serum levels of calcium, phosphorus, and phosphatase and the urinary excretion of calcium were normal.
The significance of low calcium and high phosphorus excretion in the urine as an indication of parathyroid disorder is discussed.
“Multiple endocrine adenoma syndrome” is suggested as the appropriate designation for the clinical disorders in which adenomas of one or more endocrine glands are associated with disorders of the alimentary tract when patients present with recurrent peptic ulceration, pancreatic dysfunction, or watery diarrhoea.
Pseudohypoparathyroidism is a rare disorder of calcium metabolism that involves target organ resistance to the action of the parathyroid hormone. As a result, calcium levels may become dangerously low, sometimes leading to seizures and other symptoms. We present a case of a 23-year-old Somalian female on antiepileptic therapy presenting with intractable epilepsy. She was subsequently found to have pseudohypoparathyroidism type Ia. She had multiple reasons accounting for loss of seizure control, including worsening hypocalcemia from resistance to the parathyroid hormone; vitamin D deficiency, which could have resulted from lack of exposure to direct sunlight and her antiepileptic medication; and extensive calcium deposition in the brain due to pseudohypoparathyroidism. The patient was stabilized with intravenous therapy and oral calcium, vitamin D, and calcitriol. Her antiepileptic therapy was changed to a medication that did not interfere with vitamin D metabolism or contribute to worsening hypocalcemia.
calcium; pseudohypoparathyroidism; epilepsy
No clinical or laboratory data allow a preoperative diagnosis of parathyroid carcinoma and only occasionally does a definitive finding differentiate an adenoma from a carcinoma. We present a case of primary hyperparathyroidism presenting with severe elevation of parathyroid hormone and serum calcium as well as complications. We go on to discuss the case in the light of a literature review. The severity of the elevation of the parathyroid hormone, other biochemical alterations, the presence of skeletal and renal complications and of a neck mass should alert the clinician to a possible parathyroid carcinoma. Radical surgery is the only effective therapy for parathyroid carcinoma, and should always be performed if a preoperative suspicion is entertained.
Hyperparathyroidism; Parathyroid carcinoma; Surgery; Complications; Treatment; Case report; Kuwait
We report a case of parathyroid hormone-related protein-mediated hypercalcemic crisis in a 36-year-old pregnant woman, who was admitted to women hospital with recurrent vomiting and epigastric pain. She was diagnosed with uterine fibroid since the first month of her pregnancy, but the pregnancy had been uneventful. Serum calcium was 4.8 mmol/l, while parathyroid hormone was low. Hypercalcemia was attributed to humoral hypercalcemia associated with uterine fibroid as other causes of hypercalcemia were excluded.
Humoral hypercalcemia; hypercalcemic crisis; uterine fibroid
Two cases of vitamin D deficient osteomalacia with secondary hyperparathyroidism are presented. In both cases treatment with vitamin D replacement therapy resulted in elevated calcium levels and a failure of parathormone levels to normalise, indicating autonomous parathyroid activity. Subsequent surgery in one case resulted in removal of a parathyroid adenoma. The importance of osteomalacia and its complications are discussed.
We present a case of a parathyroid adenoma on the ipsilateral side of thyroid hemiagenesis—which, to our knowledge, is the third reported case of this entity. A 41-year-old man with nephrolithiasis was found to have elevated calcium and intact parathyroid hormone levels. Both ultrasound and technetium sestamibi scintigraphy with single photon emission computed tomography confirmed left thyroid hemiagenesis and an adenoma in the left inferior thyroid bed. The patient underwent left neck exploration, which confirmed left thyroid hemiagenesis and a left inferior parathyroid adenoma. The left inferior parathyroid gland was resected. The patient was discharged home the same day of surgery and has remained normocalcemic for 14 months without evidence of hyperparathyroidism.
To present a case of hypercalcemia associated with thyrotoxicosis in a patient with vitamin D deficiency and review biochemical changes during the course of treatment.
We report a case, describe the changes in serum calcium, phosphorus, parathyroid hormone in Graves’ disease and concomitant Vitamin D deficiency. We compare our findings to those reported in literature.
Our patient had hypercalcemia secondary to thyrotoxicosis alone, which was confirmed by low parathyroid hormone level and resolution of hypercalcemia with treatment of thyrotoxicosis. The case was complicated by a concomitant vitamin D deficiency. Serum calcium elevation in patients with thyrotoxicosis occurs secondary to hyperthyroidism alone or due to concurrent hyperparathyroidism. Hypercalcemia from thyrotoxicosis is usually asymptomatic and is related to bone resorption. Vitamin D deficiency can be seen in patients with thyrotoxicosis because of accelerated metabolism, poor intestinal absorption and increased demand during bone restoration phase. Coexistence of hypercalcemia and Vitamin D deficiency in patients with thyrotoxicosis is rare, but possible, and 25-hydroxyvitamin D levels should be checked. The definite treatment for hypercalcemia in thyrotoxicosis is correction of thyroid function.
Hypercalcemia in thyrotoxicosis should be distinguished from concomitant hyperparathyroidism and confirmed by resolution of hypercalcemia with control of thyrotoxicosis. Patients with hypercalcemia and thyrotoxicosis may also have vitamin D deficiency and 25-OH Vitamin D levels should be checked.
hypercalcemia; thyrotoxicosis; vitamin D deficiency
A patient with many symptoms and signs of primary hyperparathyroidism had hypocalcaemia when first seen. Bone section histology showed osteomalacia and osteitis fibrosa, and the hyperparathyroidism at this stage was considered to be secondary to osteomalacia with postgastrectomy steatorrhoea. On treatment with vitamin D (with disappearance of her bone pains and weakness) she developed hypercalcaemia. She regained her health after removal of a 6-g. parathyroid adenoma. Normal histology was shown in another parathyroid gland.
We believe that the initial hypocalcaemia was due to vitamin-D deficiency, which produced ineffective hyperparathyroidism until it was corrected. A review of the few reports of patients with autonomous hyperparathyroidism with steatorrhoea and osteomalacia does not support the argument that these patients had “tertiary” disease. It suggests that most of them, like our patient, had primary hyperparathyroidism.