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Recalcitrant Hypocalcemia after Thyroidectomy in Patients Post Sleeve Gastrectomy--Challenges in Management.
Vemuri, SR, Koganti, SB, Mukerji, A, Razi, S, Shah, A, Gilchrist, BF
The American surgeon. 2015;(12):E426-7
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2.
An unexpected cause of severe and refractory PTH-independent hypercalcemia: case report and literature review.
Rados, DV, Furlanetto, TW
Archives of endocrinology and metabolism. 2015;(3):277-80
Abstract
Hypercalcemia is a common condition in the internal medicine practice. Sometimes its cause is not readily apparent, so extensive investigation is appropriate. Here we report an unexpected cause for hypercalcemia in an elderly woman. The case of an 82-years old woman with PTH-independent hypercalcemia, lymphocytosis, normal serum 1,25 (OH)vitamin D levels, and low serum PTHrp levels, is described. Medical history and complementary investigation were unremarkable, except for increased metabolic activity in the glutei regions, as measured by whole body 18F-FDG PET-CT. Reviewing her medical history, her sister recalled that she had been submitted to intramuscular methylmethacrylate injections, for cosmetic purposes, five years before presentation, which was confirmed by muscle biopsy. Low calcium intake, parenteral bisphosphonates, calcitonin, and glucocorticoids were used to control serum calcium levels. Methylmethacrylate injections, used cosmetically, are a new cause for hypercalcemia, even after many years. Hypercalcemia was probably due to calcitriol overproduction in foreign body granulomas. Persistent reactive lymphocytosis could be a clue for this inflammatory cause of hypercalcemia.
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3.
Use of recombinant human parathyroid hormone in hypocalcemic cardiomyopathy.
Ballane, GT, Sfeir, JG, Dakik, HA, Brown, EM, El-Hajj Fuleihan, G
European journal of endocrinology. 2012;(6):1113-20
Abstract
Hypocalcemia secondary to hypoparathyroidism is a rare cause of congestive heart failure. However, its early recognition and treatment lead to significant improvement in cardiac function. We report a middle-aged woman presenting with symptoms of heart failure with a serum calcium level of 3.7 mg/dl and a serum inorganic phosphate level of 17.6 mg/dl 22 years after subtotal thyroidectomy. Besides calcium and calcitriol supplementation, she was the first patient with severe hypocalcemic cardiomyopathy to be given off-label recombinant human parathyroid hormone (PTH) because of an elevated serum calcium-phosphate product. We discuss the management and outcome of the patient and then present a brief review of similar previously reported cases. We also describe the pivotal role of calcium ion and the potential role of PTH in maintaining myocardial contractility, effective natriuresis, and possible pathogenic mechanisms contributing to heart failure secondary to hypocalcemia and hypoparathyroidism.
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4.
Normocalcemic primary hyperparathyroidism-characteristics and clinical significance of an emerging entity.
Shlapack, MA, Rizvi, AA, Lopez, FA
The American journal of the medical sciences. 2012;(2):163-166
Abstract
The epidemiology and presentation of primary hyperparathyroidism (PHPT) is changing. Full-blown disease in its tertiary stage is rarely seen. Instead, asymptomatic hypercalcemia draws more attention because of the widespread use of screening laboratory tests. In recent years, clinicians have started ordering intact parathyroid hormone (iPTH) assays for a variety of reasons (eg, during the evaluation of fractures, nephrolithiasis, osteomalacia and low bone mass), even in individuals who have no evidence of hypercalcemia. The increased levels of iPTH without an underlying etiology may represent an early phase, or a distinct subtype, of PHPT, termed as normocalcemic primary hyperparathyroidism (NPH). The prevalence and clinical significance of NPH are unknown and are a matter of great debate. No guidelines regarding management of this entity exist. The authors describe a patient who had increased levels of iPTH, osteopenia and normal calcium and vitamin D. There was no discernible secondary cause for hyperparathyroidism (renal insufficiency, osteomalacia, vitamin D deficiency, lithium use, etc), and no intervention was offered aside from periodic monitoring of laboratory values and bone density. Analysis of data in patients with NPH should include elucidation of its natural history, temporal patterns in calcium level and prevalence of low bone mass, nephrolithiaisis and fragility fractures and could assist in devising evidence-based guidelines for management of the condition. Further characterization of NPH will be of benefit in defining its clinical impact and associated complications, to clarify the rationale behind the use of the iPTH assay as a screening test, and to assist in management of this entity.
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5.
Diagnostic and therapeutic review of cystic parathyroid lesions.
Pontikides, N, Karras, S, Kaprara, A, Cheva, A, Doumas, A, Botsios, D, Moschidis, A, Efthimiou, E, Wass, J, Krassas, G
Hormones (Athens, Greece). 2012;(4):410-8
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Abstract
BACKGROUND Parathyroid cysts (PC) are uncommon entities in routine clinical practice. The vast majority are nonfunctioning and are commonly present as asymptomatic nodular cervical lesions. PC should be considered in the differential diagnosis of an asymptomatic neck mass. Large PC can manifest with compressive symptoms of the surrounding tissues. OBJECTIVE The aim of this study is to describe nine new cases of PC and review the current literature regarding the clinical presentation, the aetiopathology, the diagnostic procedures, as well as the therapeutic approaches for this relatively rare clinical entity. METHODS-PATIENTS We present nine new patients (7 females and 2 males) diagnosed with PC, which in three were ectopic. The diagnosis of PC was based on the elevated levels of PTH in the cysts fluid. Six of the patients had nonfunctioning parathyroid lesions, while the other three had functioning ones. Patients with functioning PC had elevated serum calcium and PTH levels. Five out of nine of the cases had no symptoms, while two patients had compressive symptoms and the other two had signs and symptoms of hypercalcaemia. Needle aspiration (NA) was performed in five out of six patients with nonfunctioning PC. Surgery was the treatment in all three patients with functioning PC. RESULTS Remission after NA was achieved in four out of five patients with non-functioning PC (follow-up time: 17.7±2.3 months). In two of them, two and three aspirations were needed. One patient with nonfunctioning PC submitted to surgery with no previous NA. Patients with functioning PC maintained remission after surgery (mean follow-up time: 22.1±2.9 months). In one of them, a second surgery was performed due to the co-existence of an ectopic parathyroid adenoma. CONCLUSIONS The diagnosis of a PC can be established by finding high levels of PTH in the fluid collected by the aspiration of the cyst. PTH and Ca levels in the serum can differentiate functioning from nonfunctioning PC. The treatment of choice in nonfunctioning cysts is aspiration. Surgical removal of the cyst is indicated in hyperfunctioning cysts in cases of relapse after NA in nonfunctioning cysts and when compressive symptoms are present. Based on our series, which appears to be one of the largest reported, we propose a diagnostic algorithm to guide the diagnostic and therapeutic approach to PC.
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6.
[Anabolic therapy of induced osteoporosis in beta-thalassaemia major: case report and literature review].
Trotta, A, Corrado, A, Cantatore, FP
Reumatismo. 2010;(2):119-26
Abstract
Transfusion program and chelating therapy treatment has extended the life expectancy of thalassaemic patient; osteoporosis is considered an important cause of morbidity in adult patients who display increased fracture risk. This is a case report is about a thalassaemic young female with multiple spine fractures (D11, D12 e L2) and lumbar spine DEXA - T score = -3,1 and femoral = -3,4. This was in spite of therapy with alendronate 70 mg/week from January 2006 to September 2007. The patient was subsequentently treated for 18 months with 1-34 recombinant human parathyroid hormone and colecalciferol (100.000 U/monthly). After 4 months of therapy, the patient showed a decrease in spinal pain (Roland and Morris Disability Questionnaire) and an improvement of quality of life (Qualeffo) with normalization of osteocalcin and 25-OHcolecalciferol haematic levels after 6 months. Lumbar spine and femoral DEXA - Tscore, at 18 months, rose respectively to -2,5 and -2,4. Thalassaemia-induced osteoporosis is multifactorial and its management is very difficult. Bone marrow expansion, endocrine dysfunction, iron overload and genetic factors all seem to play important roles in the development of low bone mass in these patients. Bisphosfonates have been used in the management of thalassemia induced osteoporosis but there is no data about fracture risk. Anabolic therapy for thalassemic patients requests additional study on a large scale.
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7.
[A case of bladder tumor producing granulocyte-colony stimulation factor and parathyroid hormone-related protein].
Tsuchiya, F, Ikeda, I, Kanda, F, Fukuoka, H
Hinyokika kiyo. Acta urologica Japonica. 2001;(12):873-6
Abstract
A 68-year-old woman presented with urinary pain and frequency. Cystoscopy, intravenous pyelography and magnetic resonance imaging showed a huge bladder mass and hydronephrosis of the left kidney. Transurethral resection of bladder tumor (TUR-Bt) was done. Histopathological findings of TUR-biopsy was high grade transitional cell carcinoma. Post operatively, the laboratory examination showed marked leukocytosis with a maximum of 99,600/mm3 in the peripheral blood and a high level of granulocyte colony stimulating factor (G-CSF), 70 pg/ml in the serum (normal: less than 9.8 pg/ml). Serum calcium level increased gradually and parathyroid hormone-related protein (PTH-rP) revealed high, 8.4 pMol/l (normal: less than 0.6 pMol/l). The tumor cells were positive for G-CSF and PTH-rP immunohistochemical staining. She died of the disease 46 days after the operation. This is the third case of G-CSF and PTH-rP producing bladder tumor in the literature.
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8.
Chronic hypocalcaemia due to selective skeletal resistance to parathyroid hormone.
Tucci, JR
Clinical endocrinology. 2001;(6):815-8
Abstract
A 71-year-old man was referred for evaluation of asymptomatic hypocalcaemia dating back at least 20 years. There were no somatic abnormalities and Chvostek and Trousseau signs were negative. Serum total calcium varied from 1.88 to 2.03 mmol/l, albumin 37-44 g/l, phosphate 0.54-1.12 mmol/l and ionized calcium 1-1.13 mmol/l. Serum intact PTH levels were 69 and 55 ng/l (10-65), 25-OHD was 40 nmol/l (2.25-107.5) and 1,25-(OH)2D was 54.6 nmol/l (39-156). Serum and urine magnesium and creatinine clearance were normal. Twenty-four-hour urine calcium was 2.15 mmol and calcium/creatinine ratio 0.07. TM phosphate (maximal rate of tubular reabsorption of phosphate in mmol/l glomerular filtrate (GF)) was 0.84 mmol/l GF (0.80-1.34). Bone formation and resorption markers were normal. Bone mineral densities measured by dual-energy X-ray absorptiometry (DEXA) were within normal limits at the hip, forearm and lumbar spine. Infusion of 200 units of synthetic 1-34 PTH was associated with a rise in urinary cyclic AMP from 43 mmol/l GF to 344 mmol/l GF and TM phosphate fell from 0.93 to 0.76 mmol/l GF; 1-34 PTH infusions of 300 units twice daily for 5 days were associated with an increase in serum 1,25-(OH)2D from 80.6 to 114.4 pmol/l but no increase in serum calcium. This is a most unusual case of chronic hypocalcaemia similar to that reported by Frame et al. resulting from isolated skeletal resistance to PTH that is not related to renal insufficiency, osteomalacia or a magnesium-deficient state. These two cases appear to represent a new variant of pseudohypoparathyroidism ?type III.
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9.
Primary hyperparathyroidism in a twin pregnancy and review of fetal/maternal calcium homeostasis.
Haenel, LC, Mayfield, RK
The American journal of the medical sciences. 2000;(3):191-4
Abstract
BACKGROUND Hyperparathyroidism occurs rarely in pregnancy; this is the first reported case in a twin gestation. Management of this unusual case is described and an overview of fetal/maternal calcium homeostasis is discussed. METHODS The patient presented at 33 weeks' gestation with hypertension and premature labor. Serum calcium and phosphorus were 14.6 and 1.7 mg/dL, respectively. An intact parathyroid hormone (PTH) level was 243 pg/mL (normal, 10-65). RESULTS The patient was treated with parenteral saline hydration and oral phosphate supplementation that was continued through week 37. Although the calcium remained elevated between 12.6 and 13.3 mg/dL, medical therapy was continued because of the risks of surgery in the third trimester. Alternative medical treatments (bisphosphonates, calcitonin) were considered ill advised in pregnancy. The patient remained asymptomatic without further labor, and at week 37, fraternal twins were delivered by cesarean section. The infants were monitored closely and experienced no hypocalcemic symptoms after delivery. Postpartum, the mother's parathyroid scan and ultrasound were negative. She underwent neck exploration and a single 700-mg adenoma was removed. Transient asymptomatic hypocalcemia (7.5 mg/dL) occurred postoperatively, and she was placed on oral calcium (1500 mg/day) and calcitriol (0.25 mg/day). These were stopped at 8 weeks, when both PTH and parathyroid hormone-related peptide levels were normal. CONCLUSION Mother and infants continue to do well after 18 months. This case provides an interesting setting to consider the interrelationships between elevated maternal PTH and the fetal/placental factors that regulate calcium metabolism in pregnancy.