Expect the unexpected:Brown tumor of the mandible as the first manifestation of primary hyperparathyroidism

Hyperparathyroidism(HPT)is a condition in which one or more parathyroid glands produce increased levels of parathyroid hormone(PTH),causing disturbances in calcium homeostasis.Most commonly HPT presents with asymptomatic hypercalcemia but the clinical spectrum may include disturbances reflecting the combined effects of increased PTH secretion and hypercalcemia.Brown tumors are rare,benign,tumor-like bone lesions,occurring in 1.5%to 4.5%of patients with HPT,as a complication of an uncontrolled disease pathway,and are nowadays rarely seen in clinical practice.The tumor can appear either as a solitary or multifocal lesion and usually presents as an asymptomatic swelling or a painful exophytic mass.Furthermore,it can cause a pathological fracture or skeletal pain and be radiologically described as a lytic bone lesion.The diagnosis of a brown tumor in HPT is typically confirmed by assessing the levels of serum calcium,phosphorus,and PTH.Although when present,brown tumor is quite pathognomonic for HPT,the histologic finding often suggests a giant cell tumor,while clinical presentation might suggest other more frequent pathologies such as metastatic tumors.Treatment of brown tumors frequently focuses on managing the underlying HPT,which can often lead to regression and resolution of the lesion,without the need for surgical intervention.However,in refractory cases or when dealing with large symptomatic lesions,surgical treatment may be necessary.

Update on the current management of persistent and recurrent primary hyperparathyroidism after parathyroidectomy

Primary hyperparathyroidism(pHPT)is the third most common endocrine disease.The surgical procedure aims for permanent cure,but recurrence has been reported in 4%-10%of pHPT patients.Preoperative localization imaging is highly valuable.It includes ultrasound,computed tomography(CT),single-photonemission CT,sestamibi scintigraphy and magnetic resonance imaging.The operation has been defined as successful when postoperative continuous eucalcemia exists for more than the first six months.Ongoing hypercalcemia during this period is defined as persistence,and recurrence is defined as hypercalcemia after six months of normocalcemia.Vitamin D is a crucial factor for a good outcome.Intraoperative parathyroid hormone(PTH)monitoring can safely predict the outcomes and should be suggested.PTH≤40 pg/mL or the traditional decrease≥50%from baseline minimizes the likelihood of persistence.Risk factors for persistence are hyperplasia and normal parathyroid tissue on histopathology.Risk factors for recurrence are cardiac history,obesity,endoscopic approach and low-volume center(at least 31 cases/year).Cases with double adenomas or four-gland hyperplasia have a greater likelihood of persistence/recurrence.A 6-mo calcium>9.7 mg/dL and eucalcemic parathyroid hormone elevation at 6 mo may be associated with recurrence necessitating long-term follow-up.18F-fluorocholine positron emission tomography and 4-dimensional CT in persistent and recurrent cases can be valuable before reoperation.With these novel advances in preoperative imaging and localization as well as intraoperative PTH measurement,the recurrence rate has dropped to 2.5%-5%.Sixmonth serum calcium≥9.8 mg/dL and parathyroid hormone≥80 pg/mL indicate a risk of recurrence.Negative sestamibi scintigraphy,diabetes and elevated osteocalcin levels are predictors of multiglandular disease,which brings an increased risk of persistence and recurrence.Bilateral neck exploration was considered the gold-standard diagnostic method.Minimally invasive parathyroidectomy and neck exploration are both effective surgical techniques.Multidisciplinary diagnostic and surgical management is required to prevent persistence and recurrence.Long-term follow-up,even up to 10 years,is necessary.

Primary Hyperparathyroidism Revealed by Impaired Renal Function with Persistent Elevation of Parathormon (PTH) and Normal Calcemia after Surgical Removal of the Parathyroid Adenoma

Introduction: The curative management of primary hyperparathyroidism is based on surgery. We report the case of a patient presenting with elevated parathormone despite surgical removal of the parathyroid adenoma. Observation: A 48-year-old patient presented for more than 6 months with significant weight loss, polyuria with polydipsia and impaired renal function. The assessment carried out revealed hypercalcaemia at 155 mg/L with hypophastetaemia at 24 mg/L and an elevation of the parathyroid hormone at 218.9 pg/ml. Cervical ultrasound showed a mass at the expense of the lower left parathyroid gland. These results made it possible to conclude primary hyperparathyroidism by parathyroid adenoma which was supported surgically. The postoperative biological assessment revealed a normocalcemia at 85 mg/L associated with an increase in parathormone (PTH) at 271.9 pg/ml. In front of the normalization of calcemia in the subsequent controls and the amendment of all the signs, monitoring was carried out. 9 months after surgery, spontaneous normalization of PTH was observed at 38.4 pg/ml with normal serum calcium at 90 mg/l. Discussion: Primary hyperparathyroidism can be revealed by other manifestations. Post-operative follow-up is generally simple with normalization of PTH and serum calcium. However, there may persist an elevation of PTH with normal self-limiting calcemia.

¹¹C-Choline PET/CT in the Management of Primary Hyperparathyroidism

Primary hyperparathyroidism (PHPT) is a relative common medical problem caused by the inappropriate secretion of parathyroid hormone (PTH) by one or more parathyroid glands. The diagnosis is established by serum calcium and PTH levels and once the diagnosis is established imaging studies help localize the hyperfunctioning adenoma in preparation for curative surgery. Until now, the imaging studies most commonly utilized in PHPT are ultrasonography and 99mTc-Sesta-methoxyisobutylisonitrile (MIBI) parathyroid scintigraphy. However, these studies often fail to localize the adenoma and inappropriately delay patient referral to a potentially curative surgery. We present the case of a 64-year-old female with symptomatic PHPT who had 3 negative 99mTc-Sestamibi Scans over a period of 5 years who eventually had a PET/CT with 11C-Choline that identified a right lower parathyroid adenoma. She underwent a right lower parathyroidectomy and had a successful outcome. We present a review the current imaging techniques used in the management of PHPT including 99mTc-Sesta-MIBI scintigraphy and its limitations and novel use of PET/CT with 11C-Choline and 18F-Choline in this disease and emphasize the fact that, according to current guidelines, failure to localize the adenoma should not delay referral for curative surgery.

Surgical Treatment of Hyperparathyroidism Secondary to Chronic Renal Failure: Our Experience with the 7/8 Subtotal Parathyroidectomy Technique

Introduction: Surgical management takes place, after a certain period of evolution, on particularly severe forms of secondary hyperparathyroidism, resistant to medical treatment. Subtotal parathyroidectomy is an effective technique in the treatment of these disorders. Method: Our study is retrospective of 33 cases of hyperparathyroidism secondary to chronic renal failure in dialysis patients operated by the 7/8 technique. Identified over a period of 10 years (January 2010 to December 2019), in the ENT department of the Fann University Hospital. Results: Out of 33 cases of secondary hyperparathyroidism, the average age of our patients was 51.24 years with a sex ratio of 0.43. Causal nephropathy was dominated by nephro-angiosclerosis, which was found in 27.27% of cases. Bone pain found in 23 patients or 69.69% was the predominant clinical sign. The average calcemia was 92.7 mg/l. Parathormone was dosed in all our patients and the average was 1611.05 ng/l. The consequences were clinically marked by recurrent paresis in one patient. No case of hematoma or postoperative infection was found. On the biological level 10 patients or 30.30% had a transient hypocalcaemia. The results were marked by a drop in PTH in 23 patients or 78.78% of cases. Conclusion: Subtotal parathyroidectomy remains an effective and beneficial therapeutic method for kidney failure patients with secondary hyperparathyroidism.

Acute necrotizing pancreatitis as fi rst manifestation of primary hyperparathyroidism

We report the case of a female patient with severe acute necrotizing pancreatitis associated with hypercalcemia as first manifestation of primary hyperparathyroidism caused by a benign parathyroid adenoma.Initially the acute pancreatitis was treated conservatively.The patient subsequently underwent surgical resection of the parathyroid adenoma and surgical clearance of a large infected pancreatic pseudocyst.Although the association of parathyroid adenoma-induced hypercalcemia and acute pancreatitis is a known medical entity,it is very uncommon.The pathophysiology of hypercalcemia-induced acute pancreatitis is therefore not well known,although some mechanisms have been proposed.It is important to treat the provoking factor.Therefore,the cause of hypercalcemia should be identif ied early.Surgical resection of the parathyroid adenoma is the ultimate therapy.

Primary Hyperparathyroidism:A Tale of Two Cities Revisited-New York and Shanghai

In the 1970s, with the advent of biochemical multichannel screening in the United States and other western countries, the clinical presentation of primary hyperparathyroidism （PHPT） changed from a symptomatic to an asymptomatic disorder. However, in Asian countries, like China, PHPT did not show this evolution, but rather continued to be a symptomatic disease with target organ involvement. In this paper, we revisit the clinical features of PHPT in New York and Shanghai, representative United States and Chinese cites, over the past decade. The questions we address are whether the disease evolved in China to a more asymptomatic one and, whether in the United States further changes are evident. The results indicate that while PHPT con- tinues to present primarily as an asymptomatic disease in the United States, a new phenotype characterized by normal serum calcium and high parathyroid hormone levels, normocalcemic PHPT, has emerged. Data from Shanghai demonstrates a trend for PHPT to present more commonly as an asymptomatic disorder in China. However, most patients with PHPT in China still manifest classical symptoms, i.e. nephrolithiasis and fractures. A comparison of the two cohorts shows that Chinese patients with PHPT are younger, with higher serum calcium and PTH levels, and lower 25-hydroxyvitamin D levels than patients in New York. Normocalcemic PHPT has not yet been recognized in Shanghai. In summary, although the phenotypes of PHPT in both cities are evolving towards less evident disease, sharp clinical and biochemical differences are still apparent in PHPT as expressed in China and the United States.

Paricalcitol in hemodialysis patients with secondary hyperparathyroidism and its potential benefits

BACKGROUND Secondary hyperparathyroidism(SHPT)is a common complication in patients with end-stage renal disease and it is also common in hemodialysis patients.SHPT can increase bone fragility and calcification of blood vessels and soft tissues,which greatly increases the risk of death.AIM To discuss the outcome,safety and other potential benefits of paricalcitol injection in hemodialysis patients with SHPT.METHODS We recruited 40 patients who received hemodialysis at our hospital for chronic renal failure with SHPT between March and December 2019.They received paricalcitol injection for 24 wk(starting dose,0.06–0.08μg/kg),three times per week.They were followed up at the baseline(week 0),week 4,week 12 and week 24.The primary outcome indicator was the percentage of patients with a>30%decrease in intact parathyroid hormone(iPTH)levels at week 24 compared with the baseline.The secondary outcome indicators included percentage decrease in iPTH levels at week 24,standard-reaching rate of iPTH(percentage of patients with iPTH down to 130–585 pg/mL),changes in serum levels of calcium(Ca),phosphate(P),Ca×P product,alkaline phosphatase(ALP),creatinine(Cre),hemoglobin(Hb),and C-reactive protein(CRP),and incidence of adverse events(AEs).RESULTS After 24 wk of treatment,iPTH levels decreased significantly(598.88±381.29 pg/mL vs 888.84±376.88 pg/mL,P<0.05).More than 30%decrease of iPTH was found in 21 of 36(58.33%)patients.The average decrease in iPTH levels was 32.16±4.33%;the standard-reaching rate of iPTH levels was 66.67%(24/36);and ALP levels decreased significantly compared with the baseline(113.72±41.73 IU/L vs 133.45±56.86 IU/L)(t=2.798,P<0.05).There were no significant differences in the serum levels of calcium,Hb,Cre and CRP compared with the baseline(P>0.05).After 24 wk of treatment,serum P levels decreased compared with the baseline(1.91±0.40 mmol/L vs 2.16±0.66 mmol/L)(t=2.830,P<0.05).Ca×P product decreased significantly compared with the baseline(56.38±13.22 mg2/dL2 vs 63.97±20.30 mg2/dL2)(t=2.717,P<0.05).No serious adverse events occurred.CONCLUSION Paricalcitol was a safe and effective treatment for hemodialysis patients with SHPT.It decreased serum levels of iPTH,ALP and P and maintained stability of serum Ca levels.

Postsurgical Evaluation of Secondary Nephrogenic Hyperparathyroidism

Parathyroidectomy is useful for the treatment of secondary hyperparathyroidism(SHPT)caused by chronic renal failure.The following three types of parathyroidectomy can be performed:subtotal parathyroidectomy,total parathyroidectomy and total parathyroidectomy plus autologous transplantation(tPTX+AT).Each of the three types of surgery has advantages and disadvantages.The present study retrospectively analyzed the efficacy of tPTX+AT for the treatment of SHPT over 1 year.Thirty-seven patients who were diagnosed with secondary nephrogenic hyperparathyroidism and treated with tPTX+AT were selected between September 2014 and October 2016 and followed up for 1 year.Their average age was 66.5±46.0 years,and the average time of dialysis was 48.1±8.2 months.The patients1 conditions,including the levels of intact parathyroid hormone(iPTH)and bone metabolism,were compared preoperatively and 1 and 7 days and 1,3,6 and 12 months after surgery.In addition,the postoperative complications,pathological data,SHPT recurrence and prognosis were examined.The results showed that the postoperative level of ostalgia and cutaneous pruritus significantly decreased in the patients.An inspection of the parathyroid tissues during the operation confimied the presence of parathyroid gland hyperplasia with no carcinoma detected?Three patients with hoarseness recovered within 1 month,and 1 patient with unilateral recurrent laryngeal nerve injury improved after 6 months of voice training.Conipared to the preoperative condition,the postoperative serum iPTH,serum calcium and serum phosphate levels were significantly decreased(P<0.001),and these differences remained significant 12 months after surgery.Compared to the preoperative condition,the alkaline phosphatase(ALP)concentration was decreased on postoperative day 1(P<0.05),but no differences were observed on day 7 or at 1 month(P>0.05).The ALP levels continuously decreased at 3,6 and 12 months(P<0.01).In conclusion,tPTX+AT significantly improves the quality of life and serum biomarker levels of these patients.The convenient surgical removal of the hyperplastic parathyroid gland for postoperative recurrence supports tPTX+AT as the recommended treatment for relevant patients.

Acute pancreatitis connected with hypercalcemia crisis in hyperparathyroidism: A case report

BACKGROUND The association between primary hyperparathyroidism (PHPT) and acute pancreatitis is rarely reported.Here we describe the process of acute pancreatitismediated PHPT induced by hypercalcemia in a male patient.Hypercalcemia induced by undiagnosed PHPT may be the causative factor in recurrent acute pancreatitis.CASE SUMMARY We report a case of hypercalcemia-induced acute pancreatitis caused by a functioning parathyroid adenoma in a 57-year-old man.The patient initially experienced a series of continuous gastrointestinal symptoms including abdominal distension,abdominal pain,nausea,vomiting,electrolyte disturbance,renal dysfunction,and acute pancreatitis.Due to prolonged hypercalcemia,the patient subsequently underwent surgical resection of the parathyroid adenoma.Two weeks after surgery,his serum calcium,amylase,and lipase concentrations were normal.The patient had a good recovery after a series of other relevant therapies.CONCLUSION Acute pancreatitis as the first presentation is a rare clinical symptom caused by PHPT-induced hypercalcemia.

Effect of calcium supplementation on severe hypocalcemia in patients with secondary hyperparathyroidism after total parathyroidectomy

BACKGROUND Severe hypocalcemia(SH)is a dreaded complication of total parathyroidectomy(TPTX)without auto-transplantation.AIM To compare conventional and preventive calcium supplementation(CS)regimens in terms of SH occurrence after TPTX.METHODS This retrospective study included patients who underwent TPTX between January 2015 and May 2018 at the China-Japan Friendship Hospital.From January 2015 to May 2016,conventional CS was performed in patients who underwent TPTX,with calcium amounts adjusted according to postoperative serum calcium levels.From October 2016 to May 2018,preventive CS was performed according to preoperative alkaline phosphatase(ALP)levels.The patients were defined as lowrisk(ALP<500 U/L)and high-risk(ALP>500 U/L)for SH.All preoperative blood samples were collected in the fasting state on the day before surgery.Postoperative blood samples were obtained at 6-7 AM from the first postoperative day.RESULTS A total of 271 patients were included.These patients were 47.7±11.1 years old,and 57.6%were male.Their mean body mass index(BMI)was 22.9±3.8 kg/m^(2).There were no significant differences in sex,age,BMI,preoperative ALP,serum calcium,serum phosphorus,calcium-phosphorus ratio,and intact parathyroid hormone(iPTH)between the two CS groups.Compared with conventional CS,preventive CS led to lower occurrence rates of hypocalcemia within 48 h(46.0%vs 74.5%,P<0.001)and SH(31.7%vs 64.1%,P<0.001).Multivariable analysis showed that preoperative iPTH levelsodds ratio(OR)=1.001,95%confidence interval(CI:1.000-1.001,P=0.009),preoperative ALP amounts(OR=1.002,95%CI:1.001-1.003,P=0.002),preoperative serum phosphorus levels(OR=8.729,95%CI:1.518-50.216,P=0.015)and preventive CS(OR=0.132,95%CI:0.067-0.261,P<0.001)were independently associated with SH.In patients with preoperative ALP≥500 U/L,only preventive CS(OR=0.147,95%CI:0.038-0.562.P=0.005)was independently associated with SH.CONCLUSION This study suggests that preventive CS could reduce the occurrence of SH,indicating its critical value for hypocalcemia after TPTX.

Acute pancreatitis with hypercalcemia caused by primary hyperparathyroidism associated with paraneoplastic syndrome:A case report and review of literature

BACKGROUND Although acute pancreatitis associated with hyperparathyroidism has occasionally been reported,acute pancreatitis with metabolic encephalopathy caused by hyperparathyroidism combined with paraneoplastic syndrome is an extremely rare entity and poorly described in the literature.CASE SUMMARY We present a case of a 56-year-old female with upper abdominal discomfort and intermittent nausea and vomiting for 1 wk,without apparent abdominal pain or bloating,no jaundice and decreased blood pressure at the outset.The patient was ultimately diagnosed with moderately severe acute pancreatitis(according to the revised Atlanta classification of acute pancreatitis)combined with metabolic encephalopathy secondary to hypercalcemia caused by primary hyperparathyroidism associated with paraneoplastic syndrome.After active treatment of acute pancreatitis,massive fluid resuscitation,resection of parathyroid and uterine malignant tumors,neoadjuvant chemotherapy and other treatments,her serum calcium eventually returned to the normal level.The patient was successfully discharged from hospital.CONCLUSION This is the first case of acute pancreatitis caused by primary hyperparathyroidism associated with paraneoplastic syndrome.

Parathyroid ultrasonography and bone metabolic profile of patients on dialysis with hyperparathyroidism

AIM To evaluate the parathyroid ultrasonography and define parameters that can predict poor response to treatment in patients with secondary hyperparathyroidism due to renal failure.METHODS This cohort study evaluated 85 patients with chronic kidney disease stage V with parathyroid hormone levels above 800 pg/mL. All patients underwent ultrasonography of the parathyroids and the following parameters were analyzed: Demographic characteristics(etiology of chronic kidney disease, gender, age, dialysis vintage, vascular access, use of vitamin D), laboratory(calcium, phosphorus, parathyroid hormone, alkaline phosphatase, bone alkaline phosphatase), and the occurrence of bone changes, cardiovascular events and death. The χ~2 test were used to compare proportions or the Fisher exact test for small sample frequencies. Student t-test was used to detect differences between the two groups regarding continuous variables.RESULTS Fifty-three patients(66.4%) had parathyroid nodules with higher levels of parathyroid hormone, calcium and phosphorus. Sixteen patients underwent parathyroidectomy and had higher levels of phosphorus and calcium × phosphorus product(P = 0.03 and P = 0.006, respectively). They also had lower mortality(32% vs 68%, P = 0.01) and lower incidence of cardiovascular or cerebrovascular events(27% vs 73%, P = 0.02). Calcium × phosphorus product above 55 mg^2/dL^2 [RR 1.48(1.06, 2.08), P = 0.03], presence of vascular calcification [1.33(1.01, 1.76), P = 0.015] and previous occurrence of vascular events [RR 2.25(1.27, 3.98), P < 0.001] were risk factors for mortality in this population. There was no association between the occurrence of nodules and mortality.CONCLUSION The identification of nodules at ultrasonography strengthens the indication for parathyroidectomy in patients with secondary hyperparathyroidism due to renal failure.

Brown tumor of the femur and ulna in a woman with hyperparathyroidism

Objective A typical brown tumor caused by hyperparathyroidism(HPT) is rare. In this report, we describe our pathological findings along with a review of the literature to enhance understanding of the disease and prevent misdiagnosis, as well as to provide evidence for treatment and prognosis.Methods We present a case of brown tumor of the left proximal femur and pelvis in a 57-year-old woman who was admitted to our hospital(Dalian Municipal Central Hospital, Dalian, China). Pelvic computed tomography(CT) showed cystic expansile lesions in the left proximal femur and pelvis. Lung and abdominal CT also revealed multiple lytic lesions in the ribs and lumbar spine. X-ray of the left ulna and radius showed that the middle of the left ulna had a fracture caused by a brown tumor. A bone biopsy from the left proximal femur showed focal distribution of giant cells, with hemorrhage and fibrin hyperplasia. Results The patient underwent internal fixation of the left intertrochanteric fracture, and postoperative bone biopsy showed focal distribution of giant cells with hemorrhage and fibrin hyperplasia. The patient had a parathyroidectomy 5 months after discharge. Two weeks later, the patient developed a fracture in the right femoral neck and pain in the left forearm. X-ray of the left ulna and radius showed that the middle of the left ulna was affected by a pathological fracture caused by a brown tumor. The patient was debilitated and declined surgical treatment. The patient and her family chose discharge.Conclusion Brown tumor of bone, also called osteitis fibrosa cystica, is a rare non-neoplastic lesion that reflects abnormal bone metabolism in patients with HPT. However, with fine needle aspiration cytology in combination with biochemical tests, a correct diagnosis can be reached. The increase in osteoclast activity leads to decalcification and dissolution of bone, and formation of a cystic bone defect with hyperplastic fibrous tissue. This eventually becomes a brown tumor, with deformed and bleeding fibrous tissue. The patient had a typical brown tumor, as well as osteoporosis, anemia, and pathological fractures.

Diagnosis Performance of 99mTc-MIBI and Multimodality Imaging for Hyperparathyroidism

This study aimed to examine the diagnosis performance of^（99m）Tc-methoxyisobutylisonitrisonitrile （^（99m）Tc-MIBI） and multimodality imaging [ultrasound, single-photon emission computed tomography/computed tomography（SPECT/CT）] for hyperparathyroidism（HPT）. From Nov. 2009 to Dec. 2015, clinical data of a total of 43 HPT patients（16 males and 27 females; 26–70 years old, average age： 51.60±10.66 years old） were retrospectively analyzed. Among them, 19 patients with primary hyperparathyroidism（PHPT） underwent ^（99m）Tc-MIBI planar imaging, 24 [15 with PHPT and 9 with secondary hyperparathyroidism（SHPT）] underwent SPECT/CT hybrid imaging, and 41（33 with PHPT and 8 with SHPT） had neck ultrasound imaging. Final diagnosis was determined by pathological examination after surgery. The positive rate was compared between different imaging modalities, and the correlation analysis was conducted between imaging results and lesion size or serum parathyroid hormone（PTH） level. The results showed that the total positive rates of^（99m）Tc-MIBI imaging, ultrasound, and the two combined imaging in the 43 HPT cases were 90.70%（39/43）, 58.54%（24/41）, and 100%（41/41）, respectively. According to lesion numbers, the positive rates were 79.10%（53/67）, 53.23%（33/62）, and 88.71%（55/62）, respectively. SPECT/CT hybrid images were positive in all the 24 patients who underwent this examination. The mean maximum diameters of the lesions in ^（99m）Tc-MIBI positive and negative patients were 1.96±0.95 cm and 1.36±0.67 cm respectively, with statistically significant difference noted（P=0.03）. The T/NT of ^（99m）Tc-MIBI imaging at the early phase was correlated positively with serum PTH level（r=0.40, P=0.01）. The T/NT of ^（99m）Tc-MIBI imaging at both the early phase and the delay phase was correlated positively with lesion size（r=0.51, and r=0.45, respectively; P〈0.01 for both）. It was concluded that ^（99m）Tc-MIBI imaging presents significant value for location diagnosis of HPT, especially when combined with SPECT/CT hybrid imaging or ultrasound. The ^（99m）Tc-MIBI uptake correlates positively with serum PTH level and lesion size.

New options for the management of hyperparathyroidism after renal transplantation

The persistence and severity of hyperparathyroidism(HPT) post-renal transplantation is relatively frequent and primarily associated with the timing and its magnitude in the pre-transplant period and with the presence of parathyroid adenomas. HPT after renal transplantation is clinically manifested with hypercalcemia, hypophosphatemia, bone pain, fractures, and in more serious cases with cardiovascular calcifications that affect the survival. The primary clinical objective for patients with secondary HPT after renal transplantation is to obtain a level of parathyroid hormone(PTH) adequate to the renal transplanted function and to normalize levels of calcium, phosphorus and vitamin D. In many cases during this period, the development of hypercalcemiaand/or hypophosphatemia makes it necessary to take different therapeutic measures. The use of vitamin D or its analogues has been extrapolated from the management of pre-transplant HPT obtaining variable outcomes, although its use is limited by its capacity to produce hypercalcemia. Calcimimetics are drugs that have proven be effective in reducing PTH levels in patients with HPT on dialysis and has been effective in reducing up to 50% PTH levels in moderate to severe HPT in post-renal transplantation.When HPT persists after renal transplantation and does not respond to medical treatment, invasive management by percutaneous ethanol injection therapy of parathyroid glands or parathyroidectomy should be considered. The emergence of new methods for the management of HPT expands the availability of therapeutic tools for transplant patients.

Huge brown tumor of the rib in an unlocatable hyperparathyroidism patient with “self-recovered” serum calcium and parathyroid hormone: A case report

BACKGROUND The brown tumor is a kind of complication of hyperparathyroidism(HPT). The ultimate therapy usually is the resolution of HPT. We herein report an unlocatable HPT patient who received resection of a huge brown tumor of the rib, and experienced "self-recovery" of serum calcium and parathyroid hormone.CASE SUMMARY A 34-year-old female patient who suffered from a gradually increasing mass of the left chest wall since 2007 came to our hospital for treatment. The patient had a history of serum Ca and parathyroid hormone(PTH) increasing since June 2015 and received zoledronic acid treatment for 17 mo. When she came to our hospital in November 2017 after discontinuing medical treatment for 3 mo, the serum Ca and PTH levels were within normal ranges. The patient had no imaging abnormalities of parathyroid ultrasound or 99 m Tc-methoxyisobutyl isonitrile.Enhanced computed tomography revealed a local soft tissue mass of 96 mm × 113 mm with bone erosion of the left 8 th rib, and the mass presented irregular enhancement with an unclear boundary between the mass and spleen. The mass was thought to likely be caused by HPT, but a malignancy could not be ruled out.Resection of the mass was performed, and the pathology proved that the mass was a brown tumor. A diagnosis of unlocatable HPT was considered. Since the serum Ca and PTH levels were both normal pre-and post-operation, the patient did not receive exploratory surgery for HPT, and received regular follow-up.CONCLUSION The huge brown tumor of the rib and "self-recovered" serum PTH and Ca levels are relatively rare in HPT patients. An exploratory operation may be deferred for these patients, and long-term follow-up should be performed.

Primary hyperparathyroidism presenting as acute pancreatitis: An institutional experience with review of the literature

BACKGROUND Acute pancreatitis(AP)presenting as an initial manifestation of primary hyperparathyroidism(PHPT)is uncommon,and its timely diagnosis is crucial in preventing recurrent attacks of pancreatitis.AIM To determine the clinical,biochemical,and radiological profile of PHPT patients presenting as AP.METHODS This is a retrospective observational study,51 consecutive patients admitted with the diagnosis of PHPT during January 2010 and October 2021 at a tertiary care hospital in Puducherry,India was included.The diagnosis of AP was established in the presence of at least two of the three following features:abdominal pain,levels of serum amylase or lipase greater than three times the normal,and characteristic features at abdominal imaging.RESULTS Out of the 51 consecutive patients with PHPT,twelve(23.52%)had pancreatitis[5(9.80%)AP,seven(13.72%)chronic pancreatitis(CP)].PHPT with AP(PHPT-AP)was more common among males with the presentation at a younger age(35.20±16.11 vs 49.23±14.80 years,P=0.05)and lower plasma intact parathyroid hormone(iPTH)levels[125(80.55-178.65)vs 519.80(149-1649.55,P=0.01)]compared to PHPT without pancreatitis(PHPT-NP).The mean serum calcium levels were similar in both PHPT-AP and PHPT-NP groups[(11.66±1.15 mg/dL)vs(12.46±1.71 mg/dL),P=0.32].PHPT-AP also presented with more gastrointestinal symptoms like abdominal pain,nausea,and vomiting with lesser skeletal and renal manifestations as compared to patients with PHPT-NP.CONCLUSION AP can be the only presenting feature of PHPT.Normal or higher serum calcium levels during AP should always draw attention towards endocrine causes like PHPT.

Vitamin D and Secondary Hyperparathyroidism in HIV Infected Patients Taking Antiretroviral Therapy

Objective: Due to the lack of studies assessing hypovitaminosis D and secondary hyperparathyroidism in Brazilian HIV-infected population, especially in the northeastern population, this study aimed to determine the profile of these conditions in patients infected with HIV and its correlation with immuno-virological, sociodemographic data and associated comorbidities. Methods: Comparison studies were obtained from routine clinical samples of HIV infected patients submitted for 25-OH Vitamin D, PTH and alkaline phosphatase determination. Results: A total of 78 patients were included, 42 (53.8%) males, mean age 45.7 years. Antiretroviral regimens most used in this study were Zidovudine/Lamivudine/Efavirenz 17.9%, Tenofovir/Lamivudine/Efavirenz 17.9%,Tenofovir/Lamivudine/Atazanavir-r 15.4%. The mean value CD4 count was 592.1 ± 247.2 cells/mm3, CD8 cell count was 1026.5 ± 467.3 cells/mm3, mean detectable viral load was 2220 ± 15703 copies and CD4/CD8 ratio was 0.63 ± 0.33. A total of 34 vitamin D dosages were collected with 41.2% representing sufficient amount and 58.8% insufficient. Alkaline Phosphatase (ALP) dosage was elevated in 49.3% (N=35) of the patients. Parathormone (PTH) was elevated in 18% (N = 11). Among patients with elevated PTH levels, 81.9% had elevated levels of ALP (p = 0.01). In the group of patients with high levels of ALP, 45.7% had a CD4 count 3 (p = 0.02). There was no significant difference in vitamin D related to gender (p = 0.21), age (p = 0.23), CD4 count (p = 0.26), suppressed viral load (p = 0.44) or blood glucose (p = 0.45). Conclusions: This study evidenced a high prevalence of Vitamin D insufficiency in Northeast Brazil, which suggests HIV infection correlation. A high prevalence of Hyperparathyroidism was detected and related with inflammatory condition persistence and low CD4 count. We suggest improve vitamin D follow up and measurements in this population with better CD4 count control to avoid future osteoarticular complications of HIV treatment.

Sagliker syndrome: A case report of a rare manifestation of uncontrolled secondary hyperparathyroidism in chronic renal failure

BACKGROUND Sagliker syndrome(SS)resulting from uncontrolled secondary hyperparathyroidism(SHPT)in chronic renal failure(CRF)is seldom reported.CASE SUMMARY A 24-year-old woman presented with asymmetric facial deformity and stature shortening.She was diagnosed with SS,SHPT,CRF,and thyroid cancer.The patient underwent a total parathyroidectomy and thyroidectomy with central lymph node dissection.The patient’s condition was stable and was discharged from the hospital.CONCLUSION Undergoing dialysis vintage,presenting high serum phosphate levels,and female gender may be risk factors for SS.Intramembranous ossification in the craniomaxillofacial region is possibly activated in this special pathophysiological condition.What’s more,the choice of surgery mainly depends on the treatment goal and the experience of the individual surgeon.