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Purpose: To evaluate whether patients presenting with laboratory results consistent with primary hyperparathyroidism (pHPT) are managed in accordance with guidelines.Methods: The laboratory database at a hospital in Sweden, serving 127,000 inhabitants, was searched for patients with biochemically determined pHPT. During 2014, a total of 365 patients with biochemical laboratory tests consistent with pHPT were identified. Patients with possible differential diagnoses or other reasons for not being investigated according to international guidelines were excluded after scrutinizing records, after new blood tests, and clinical assessments by endocrine surgeons.Results: Altogether, 92 patients had been referred to specialists and 82 had not. The latter group had lower serum calcium (median 2.54 mmol/L) and PTH (5.7 pmol/L). Out of these 82 cases, 9 patients were diagnosed with pHPT or had some sort of long-term follow-up planned as outpatients.Conclusion: Primary hyperparathyroidism is overlooked and underdiagnosed in a number of patients in the clinical setting. It is important to provide local guidelines for the management of patients presenting with mild pHPT to ensure that these patients receive proper evaluation and follow-up according to current research. [ABSTRACT FROM AUTHOR]

Introduction. Brown tumors of hyperparathyroidism are locally destructive bone lesions. They are the late clinical consequence of the disease. They can occur in primary, secondary, and rarely tertiary forms. They affect usually long bones and less frequently those of the maxilla. Case Report. Our 45-year-old female patient presented with a mandibular tumor next to the first right lower molar. At first, we have chosen tooth extraction and tumor excision. When the histological report showed the giant cell tumor we suspected a metabolic bone disorder. Biochemical tests screened hyperparathyroidism and severe vitamin D deficiency, and parathyroid scintiscan revealed parathyroid adenoma. Discussion. The association of hyperparathyroidism and vitamin D deficiency leads to diagnostic uncertainty. First, secondary hyperparathyroidism can be due vitamin D deficiency. Second, data available show that vitamin D deficiency is more prevalent in patients with primary hyperparathyroidism than in general population. Hyperparathyroidism management is based on correct and precise diagnosis. Furthermore, the resolution of brown tumors depends on the cure of hyperparathyroidism. In fact, bone lesions should regress after biological tests’ normalization. Conclusion. Clinicians should be aware of such rare and complicated presentation. They must consider the diagnosis of the brown tumor to avoid extensive surgical excision and teeth extractions. [ABSTRACT FROM AUTHOR]

Context and objective Detailed information on the epidemiology of parathyroid disorders in India is lacking. Most of the available data pertain to primary hyperparathyroidism ( PHPT) rather than the overall burden of parathyroid disorders. We studied the incidence of parathyroid disorders in a cohort of service personnel followed for a long duration. Design, setting and patients: The data for this retrospective, descriptive epidemiological study were derived from the electronic medical records ( EMR) of health care personnel enrolled between 1990 and 2015. Subjects were recruited between the ages of 17 and 20 years in good health, and the data pertaining to parathyroid disorders were derived from the EMR. Main outcomes measures We calculated the incidence rates per person-years of parathyroid disorders using appropriate statistical methods. Results Our analysis includes 51 217 participants (median age 33, range 17-54 years) with a mean follow-up of 12.5 years. Yearly evaluation of the data gave cumulative follow-up duration of 613 925 person-years. PHPT was diagnosed in 37 patients and hypoparathyroidism in 16 patients, giving incidence rates of 6 and 2.6 per 100 000 person-years, respectively. Only one patient was diagnosed with pseudohypoparathyroidism (0.16 per 100 000 person-years). Of the 37 patients with PHPT, 16 (43%) developed postsurgical hypoparathyroidism. Conclusions Our cohort had a low incidence of PHPT when compared to Western populations. Long-term epidemiological studies are essential to identify the demographic trends of metabolic bone disorders in India. [ABSTRACT FROM AUTHOR]

Objective. The brown tumour of hyperparathyroidism is a result of a metabolic disorder caused by primary hyperparathyroidism. Report. We described a case of a 37-year-old female patient presenting bimaxillary intraoral lesions and swelling in the neck. Incisional biopsy of the oral lesion was performed and histopathological examination revealed a central giant cell lesion composed by intense haemorrhagic exudate, abundant presence of giant cells, and areas with hemosiderin pigment. The patient also presented high levels of serum calcium and parathyroid hormone, hyperfunctioning parathyroid tissue, bilateral parenchymal nephropathy, and densitometry lower than expected, showing an advanced stage of osteitis fibrosa cystica. Synchronous parathyroid adenoma and papillary thyroid carcinoma were confirmed by imaging exams and histopathologically. Conclusion. The composition of all the clinical, pathological, and imaging findings led to the final diagnosis of brown tumour of hyperparathyroidism. The occurrence of parathyroid adenoma, papillary thyroid carcinoma, and brown tumours of hyperparathyroidism in their late stage (osteitis fibrosa cystica) associated with oral brown tumours involving the mandible and maxilla is extremely rare. [ABSTRACT FROM AUTHOR]

Osteitis fibrosa cystica ( OFC) is the most frequent type of osseous change in renal osteodystrophy affecting the majority of dialysis patients. Brown tumors are a severe form of OFC. The involvement of the craniofacial skeleton causing facial disfigurement in patients on dialysis appears to be limited to case reports. After searching PubMed, we performed a systematic review of 127 cases with a severe form of OFC resulting in a facial disfigurement to understand possible determinants for this condition. We found that since the first published case in 1974, and after a peak in 1996, there appears to be an increase in published reported cases. Only 27.6% of these cases were published in nephrology journals. The most common region for reported cases was North America. Mean age of these patients was 31.2 years with a mean dialysis duration of 7 years. Almost 67% were women, and almost all were on hemodialysis. The disease tended to most commonly localize to the maxilla (73.2%) and mandible (57.5%). As part of the treatment, 59% of patients had a parathyroidectomy. More than one-third (35.4%) had symptomatic improvement at follow-up. Mean follow-up was 1.6 years. Clinicians should be aware of this clinical presentation of a severe form of OFC and/or brown tumors. Timely diagnosis and intervention may help to prevent or decrease destructive bone changes and reduce negative psychological consequences of facial disfigurement. [ABSTRACT FROM AUTHOR]

Background: A recent study of focused minimally invasive parathyroidectomy (FPTX) in sporadic primary hyperparathyroidism (pHPT) using intraoperative parathyroid hormone (ioPTH) measurements shows that inadequate ioPTH drop and multiglandular disease are more commonly found when a first gland <200 mg is resected. Our aim was to study if a resected gland that weighed <200 mg was associated with an increased persistence rate after FPTX. Methods: This is a cohort study of FPTX for pHPT performed in the period 1998-2013. FPTX was performed in patients with pHPT where Sestamibi and Ultrasound imaging localized single-gland disease, only one gland was excised and the weight recorded. IoPTH was not used routinely. Two groups were composed according to the weight of the resected gland: Group A <200 mg and Group B ≥200 mg. Persistent or recurrent disease was defined if it occurred within, or after 6 months. The primary outcome measure was the rate of persisting pHPT. Results: A total of 3,511 parathyroidectomies were performed, and a total 1,745 FPTX (1,347 female) met inclusion criteria. There were 245 and 1,500 patients in groups A and B, respectively. The rate of persistent pHPT was higher in Group A, 6.1 versus 2.0 % ( p < 0.001). Findings at re-operative surgery showed that the ipsilateral gland was diseased in 47 % (7/15) of persistent cases in group A. Conclusion: The risk of persistent disease after MIP was higher if the resected gland weighed ≤200 mg, and this corroborates the findings of a recent study. A heightened awareness of the possibility of multigland disease should be raised, and ioPTH monitoring, identification of the ipsilateral gland or bilateral exploration may be advisable in such cases. [ABSTRACT FROM AUTHOR]

Uremic osteodystrophy is an expected complication in subjects with chronic renal insufficiency. It develops gradually and progressively already during the conservative treatment and then during the dialysis treatment. It can present a wide histopathological spectrum including typical alterations (from osteitis fibrosa to osteomalacia and/or mixed lesions) or, more rarely, isolated bone lesions indicative of a brown tumor of the bone. These conditions must be clearly identified in the pretransplant phase, especially if a bone lesion indicative of a pathological condition possibly evolving into a neoplasm is detected fortuitously. We report the case of a 19-yr-old boy with renal insufficiency and candidate for a pre-emptive renal transplantation from a living donor, in whom the diagnosis of ABC of the pubic symphysis - asymptomatic and fortuitously detected while performing instrumental investigations - was suspected through the imaging studies (CT scan, MRI) and was confirmed by the histological examination. This made it possible to perform the renal transplant. The immunosuppressive treatment, which was subsequently administered, was based on steroids, calcineurin inhibitors (tacrolimus), and mycophenolate and did not determine any modification in the radiological aspect of the bone lesion, even after more than one yr from the transplant. [ABSTRACT FROM AUTHOR]

Brown tumors are osteolytic bone lesions that occur as a result of persistent hyperparathyroidism. They usually appear late in the natural history of the disease and are currently very rare due to an earlier diagnosis of primary hyperparathyroidism. We present the case of a 53-year-old female with a 2-month history of bitemporal hemianopsia and diplopia. A computed tomography showed an osteolytic bone lesion that involved the sphenoid corpus and clivus. A biopsy was made and the histopathology result was consistent with a brown tumor. The aforementioned location is very infrequent to such tumors, and therefore represented a diagnostic challenge. However, in this case, its association to primary hyperparathyroidism was the clue for the diagnosis. The association of a brown tumor secondary to a parathyroid carcinoma has been seldom reported. This case represents, to our knowledge, one of the few brown tumors described in such location and the first secondary to a parathyroid carcinoma. [ABSTRACT FROM AUTHOR]

Primary hyperparathyroidism is an endocrinopathy which is characterized with the hypersecretion of parathormone. During the progress of the disease bone loss takes place due to resorption on the subperiosteal and endosteal surfaces. Brown tumor is a localized form of osteitis fibrosa cystica, being part of the hyperparathyroid bone disease. It is rarely the first symptom of hyperparathyroidism. Nowadays, the diagnosis is made at an asymptomatic or minimally symptomatic stage. We present a male patient presented with a massive painless swelling in the left maxilla as the firstmanifestation of primary hyperparathyroidism due to a parathyroid adenoma. Parathyroidectomy was performed, and there was a regression of the bone lesion, without the need of performing other local surgical procedures. [ABSTRACT FROM AUTHOR]

Clin. Otolaryngol. 2010, , 468-473 Calcium levels are often measured to diagnose postoperative hypocalcaemia following thyroidectomy. The aims of this study were to (i) prospectively determine the incidence of symptomatic and biochemical hypocalcaemia following thyroidectomy, (ii) to identify if any associations exist between hypocalcaemia, type of surgery, histological diagnosis, specimen size/weight and the presence of histological parathyroid tissue and (iii) to evaluate the necessity of routine measurement of calcium levels following all thyroidectomies. Prospective clinical study. University teaching hospital. Eighty-six patients presenting consecutively for thyroid surgery. Type of surgery, indications, perioperative calcium levels, symptoms of hypocalcaemia and histology were documented. Fifty-four patients underwent thyroid lobectomy and isthmusectomy, 19 underwent total and 13 completion thyroidectomy. Significantly, no patient undergoing thyroid lobectomy developed hypocalcaemia versus 26% of total thyroidectomies ( P = 0.001) and 23% of completion thyroidectomies ( P = 0.006). All eight patients with hypocalcaemia required treatment. Seven were initially identified clinically. Logistic regression analysis revealed that operation type was the only independent risk factor for developing hypocalcaemia ( P = 0.021). No patient developed hypocalcaemia following thyroid lobectomy and isthmusectomy. Considering the majority (63%) of thyroid surgeries were lobectomies, most patients tested appear low risk for hypocalcaemia. Definitive prediction of hypocalcaemia postoperatively remains a challenge. However, our results suggest that analysing calcium levels routinely following thyroid lobectomy is unwarranted. [ABSTRACT FROM AUTHOR]

Objective To describe our 3-year experience in the long-term efficacy and safety of percutaneous ethanol injection therapy (PEIT), as an alternative to surgery for the management of patients with primary hyperparathyroidism (p-HPT). Design Prospective study with a mean follow-up of 19·6 ± 10·6 months. Patients Our study population included 19 consecutive high risk patients with p-HPT, who met the criteria for surgery. Measurements Under ultrasonic guidance, ethanol (95%) was injected into parathyroid glands with a volume of ≥ 0·15 cm3. With the aim of normalizing intact parathormone (iPTH) values, repeated ethanol injections were carried out, in an interval of 2 weeks, until normalization of iPTH was reached or until no residual blood supply was detected by ultrasound in the gland. Biochemical parameters were monitored throughout the study. Results At 6-month follow-up, normalization of iPTH levels (10–65 ng/l) was achieved in 11 (58%) patients (responders). Of the eight remaining patients (nonresponders), six patients had reduced (but not normalized) iPTH levels and two patients required parathyroid surgery. Seventeen (11 responders and 6 nonresponders) of the 19 patients (89·5%) became normocalcaemic (serum Ca ≤ 2·57 mmol/l) and remained so for a mean follow-up of 21 months (8–39 months). The odds ratio ( P < 0·05) of response vs. no response to PEIT was 16·7-fold for pretreatment iPTH < 200 vs. > 200 ng/l. The only complication was a transient dysphonia noticed in three patients. Conclusions PEIT is a safe and effective nonsurgical treatment for patients with p-HPT, who are unsuitable for surgical intervention. [ABSTRACT FROM AUTHOR]

Chronic renal disease is characterised by progressive and irreversible loss of renal function. A number of systemic and oral complications are associated with chronic renal disease. We report a rare case of jaw enlargement caused by the simultaneous presence of osteitis fibrosis and brown tumour secondary to hyperparathyroidism. Both conditions are related to increased levels of parathyroid hormone and dental surgeons should be aware of this important complication associated with chronic renal disease. [ABSTRACT FROM AUTHOR]

Renal osteodystrophy (ROD) develops as the early stages of chronic renal failure (CRF) and covers a spectrum of bone changes observed in the uraemic patient, which extend from high remodelling bone disease (frequently known as osteitis fibrosa) to low turnover, or adynamic disease. Between these two extremes there are also cases of bone mineralization compromised in variable degrees, as is the case of ‘mixed bone disease’ and osteomalacia. The dynamic process of bone remodelling is compromised in CRF, and a positive or negative bone balance can be observed in uraemic patients. In addition to the classic modulators of bone remodelling, like parathyroid hormone, calcitriol and calcitonin, other factors were recently identified as significant modulators of osteoblast and osteoclast activation in uraemic patients. In fact, different cytokines and growth factors, acting at an autocrine or paracrine level, seem to play a relevant role in the bone and mineral changes observed in uraemia. Recently, observations have been made of the development of more sensitive and specific techniques to assay different biochemical markers of bone turnover and mineral metabolism. Analogously, new contributions of conventional bone histology, bone immunocytochemistry and molecular biology, which enabled the understanding of some etiopathogenic mechanisms of ROD, were observed. [ABSTRACT FROM AUTHOR]

The parathyroid gland (PTG) is a unique endocrine organ in which the quiescent glandular cells begin to proliferate in rsponse to the demand for maintaining calcium (Ca) homeostasis in the progressive course of renal failure, leading to secondary hypereparathyroidism (SHPT). SHPT is characterized with continuous over-secretion of parathyroid hormone (PTH) and high turn-over bone disease, osteitis fibrosa, and the major factors include a deficiency of active vitamin D, hypocalcemia, and phosphate retention. With long-term end-stage renal failure, SHPT becomes resistant to conventional medical treatment such as phosphate binders and active vitamin D supplementation, and the growth of the PTG accelerates with the pattern of hyperplasia changing from diffuse to nodular type. In this process, the sigmoid curve between extracellular Ca concentration (exCa) and the plasma level of PTH shifts to the upper-rightward, indicating both an absolute increase in PTH secretion and the resistance of PT cells to exCa. Many experimental and human studies have revealed down-regulation of vitamin D receptor (VDR), calcium-sensing receptor (CaSR), and retinoid X receptor (RXR) in PT cells. The sustained proliferation of PT cells after obtaining autonomicity is another characteristic feature of SHPT. In this context, it has been demonstrated that the cell cycle is markedly progressed, where the expression of cyclin-dependent kinase inhibitor (CDKI), p21 and p27, is depressed in a VDR-dependent manner. These pathological features are most evident in nodular hyperplasia, in which monoclonal proliferation is obvious, indicating the phenotypic changes have occured in PT cells. It has been observed by Fukagawa and colleagues that pharmacologically high dose of active vitamin D administered orally can cause small-size PTG hyperplasia to regress in patients with advanced SHPT. Successful renal transplantation may also restore VDR and CaSR expressions in the diffuse type, in association with increasing TUNEL-positive cells. Thus, it is important to vigorously treat SHPT when the PT cell proliferation is in the reversible stage of diffuse hyperplasia. [ABSTRACT FROM AUTHOR]

Osteitis Fibrosa Cystica (OFC) is defined as the classic skeletal manifestation of advanced primary hyperparathyroidism. With the increased detection by means of routine calcium screening, the clinical profile of primary hyperparathyroidism in Western countries has shifted from symptomatic disease to one with subtle or no specific symptoms ("asymptomatic" primary hyperparathyroidism). The authors describe a classical feature of advanced primary hyperparathyroidism due to a parathyroid adenoma and its successful treatment. [ABSTRACT FROM AUTHOR]

Currently available cinacalcet hydrochloride has a greater advantage over conventional vitamin D therapy, although the ability of cinacalcet to suppress parathyroid hormone (PTH) varies among individuals. This article deals with the clinical issues of cinacalcet. Studies examining the impact of cinacalcet on parathyroid gland cells showed that cinacalcet may cause changes in PTH secretion, reduce the parathyroid gland size, and alter parathyroid gland function, suggesting the possibility that use of cinacalcet can eventually be stopped. Combined cinacalcet and vitamin D can reportedly increase vitamin D receptor expression. In a double-blind, placebo-controlled randomized study involving 1184 previously untreated secondary hyperparathyroidism (SHPT) patients, cinacalcet significantly reduced the likelihood of hospitalization for parathyroidectomy, fractures, and cardiovascular disease. Fibrous osteitis was suppressed in the rat model of renal failure treated with cinacalcet. Considering that the drug price of cinacalcet is lower in Japan than in the USA and Europe, it is highly likely that cinacalcet will be used to suppress PTH in Japan. [ABSTRACT FROM AUTHOR]

We aimed to assess the impact of oral calcium supplementation (OCS) on the prevalence of nephrolithiasis among a cohort of patients undergoing surgery for primary hyperparathyroidism (PHPT) . There were 339 patients undergoing surgery for PHPT with detailed past medical history data that were analyzed. 73 patients (22%) had a history of nephrolithiasis prior to parathyroid surgery. Nephrolithiasis was more common among men than women (40% vs 15%, P < 0.001), despite the predominance of women (73% of patients) with hyperparathyroidism. 83 patients (25%) used OCS. OCS was associated with a lower prevalence of nephrolithiasis (9.6% vs 25.4% without OCS, P = 0.002). This protective effect included both men and women (rates of nephrolithiasis with and without supplements: men 19% vs 46%, P = 0.027; women 7% vs 17%, P = 0.04). The mechanism for the apparent protective effect of OCS on rates of nephrolithiasis is unclear, and further research is required to elucidate the variable penetrance of nephrolithiasis among PHPT patients. [ABSTRACT FROM AUTHOR]

We report on a 62-year-old female initially suffering from extreme pain in both her lower extremities. Plain radiographs revealed multiple osteolytic lesions. Laboratory analyses indicated high levels of serum calcium and parathyroid hormone related protein (PTHrP) and detected HTLV-1 antibody. Histological examination of biopsied specimens which were obtained from osteolytic lesions indicated osteitis fibrosa cystica (OFC) without tumour cells. After the initial biopsy, a subcutaneous mass developed in the left thigh. Microsections of the biopsied mass disclosed adult T-cell leukaemia (ATL) neoplastic cells. Immunohistochemical findings revealed that PTHrP secreted from ATL neoplastic cells induced the OFC. [ABSTRACT FROM AUTHOR]

The article describes the case of a 41-year-old Sri Lankan woman who was presented with primary hyperparathyroidism and osteitis fibrosa cystica.

Presents the case of a woman that was found to have osteitis fibrosa cystica without tumor cells. The prevalence of HTLV-1 infection in Japan; Disorders it can cause.