1.Persistent primary hyperparathyroidism secondary to an ectopic mediastinal adenoma in a young adult: A case report
Karl Homer Nievera ; Rebecca Alba
Journal of the ASEAN Federation of Endocrine Societies 2023;38(2):145-148
Primary hyperparathyroidism commonly affects elderly women. When present in the young population, it is usually asymptomatic, most frequently due to a parathyroid adenoma and the definitive management is surgical excision. Uncommonly, 5-10% of patients fail to achieve long-term cure after initial parathyroidectomy and 6-16% of them is due to an ectopic parathyroid adenoma that will require focused diagnostic and surgical approaches. We report a 21-year-old male who had bilateral thigh pain. Work-up revealed bilateral femoral fractures, brown tumors on the arms and multiple lytic lesions on the skull. Serum studies showed hypercalcemia (1.83 mmol/L), elevated parathyroid hormone [(PTH) 2025.10 pg/mL], elevated alkaline phosphatase (830 U/L), normal phosphorus (0.92 mmol/L) and low vitamin D levels (18.50 ng/mL). Bone densitometry showed osteoporotic findings. Sestamibi scan showed uptake on the left superior mediastinal region consistent with an ectopic parathyroid adenoma. Vitamin D supplementation was started pre-operatively. Patient underwent parathyroidectomy with neck exploration; however, the pathologic adenoma was not visualized and PTH levels remained elevated post-operatively. Chest computed tomography with intravenous contrast was performed revealing a mediastinal location of the adenoma. A repeat parathyroidectomy was done, with successful identification of the adenoma resulting in a significant drop in PTH and calcium levels. Patient experienced hungry bone syndrome post-operatively and was managed with calcium and magnesium supplementation. A high index of suspicion for an ectopic adenoma is warranted for patients presenting with hypercalcemia and secondary osteoporosis if there is persistent PTH elevation after initial surgical intervention. Adequate follow-up and monitoring is also needed starting immediately in the post-operative period to manage possible complications such as hungry bone syndrome.
Hyperparathyroidism
;
Reoperation
;
Hypercalcemia
2.Hypercalcium crisis and postoperative hungry bone syndrome caused by primary hyperparathyroidism: a case report.
Mengdi ZHANG ; Yifei ZENG ; Lei WANG ; Yian SUN ; Jingwei LI
Journal of Clinical Otorhinolaryngology Head and Neck Surgery 2023;37(5):389-392
To review the diagnosis and treatment of a case of hypercalcium crisis caused by primary hyperparathyroidism(PHPT) and prophylactic treatment of hungry bone syndrome. In a 32-year-old male with hypercalcemia, the main manifestations were loss of appetite, nausea, polyuria, polydipsia, fatigue, lethargy, etc. parathyroid hormone, serum calcium increased, thyroid function was normal, thyroid color ultrasound and MRI showed space-occupying behind the right thyroid, radionuclide examination showed abnormal imaging agent concentration in the right parathyroid area, there was a history of pathological fracture. Clinically diagnosed as hypercalcemia crisis secondary to PHPT.
Male
;
Humans
;
Adult
;
Hypercalcemia/diagnosis*
;
Hyperparathyroidism, Primary/surgery*
;
Parathyroid Hormone
;
Hypocalcemia/complications*
;
Thyroid Gland
;
Calcium
3.The influencing factors of renal response in newly diagnosed multiple myeloma patients with renal impairment.
Yu Hang SONG ; Fu Jing ZHANG ; Rong Rong HU ; Miao CHEN ; Chen YANG ; Wei WANG ; Yan QIN ; Dao Bin ZHOU ; Jun Ling ZHUANG
Chinese Journal of Hematology 2023;44(2):141-147
Objective: To investigate the causative factors of renal function in newly diagnosed multiple myeloma (MM) patients with renal inadequacy. Methods: 181 MM patients with renal impairment from August 2007 to October 2021 at Peking Union Medical College Hospital were recruited, whose baseline chronic kidney disease (CKD) stage was 3-5. Statistical analysis was performed based on laboratory tests, treatment regimens, hematological responses, and survival among various renal function efficacy groups. A logistic regression model was employed in multivariate analysis. Results: A total of 181 patients were recruited, and 277 patients with CKD stages 1-2 were chosen as controls. The majority choose the BCD and VRD regimens. The progression-free survival (PFS) (14.0 months vs 24.8 months, P<0.001) and overall survival (OS) (49.2 months vs 79.7 months, P<0.001) of patients with renal impairment was considerably shorter. Hypercalcemia (P=0.013, OR=5.654) , 1q21 amplification (P=0.018, OR=2.876) , and hematological response over a partial response (P=0.001, OR=4.999) were independent predictive factors for renal function response. After treatment, those with improvement in renal function had a longer PFS than those without (15.6 months vs 10.2 months, P=0.074) , but there was no disparity in OS (56.5 months vs 47.3 months, P=0.665) . Conclusion: Hypercalcemia, 1q21 amplification, and hematologic response were independent predictors of the response of renal function in NDMM patients with renal impairment. MM patients with CKD 3-5 at baseline still have worse survival. Improvement in renal function after treatment is attributed to the improvement in PFS.
Humans
;
Multiple Myeloma/drug therapy*
;
Bortezomib/therapeutic use*
;
Hypercalcemia
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Prognosis
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Chromosome Aberrations
;
Kidney/physiology*
;
Renal Insufficiency, Chronic
;
Retrospective Studies
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Antineoplastic Combined Chemotherapy Protocols
4.An atypical presentation of primary Hyperparathyroidism with multiple spontaneous Tendon Ruptures: A case report and literature review on the management of primary Hyperparathyroidism
Journal of the ASEAN Federation of Endocrine Societies 2022;37(2):76-82
Primary hyperparathyroidism (PHPT) is a common endocrine condition, increasingly presenting asymptomatically and detected on routine laboratory examination in developed countries. Multiple spontaneous tendon ruptures as the initial presentation of PHPT is extremely rare. We present the case of a 28-year-old male diagnosed with severe hypercalcemia secondary to PHPT after presenting with complications of multiple spontaneous tendon ruptures,and discuss the management issues in PHPT for this patient.
Hyperparathyroidism, Primary
;
Hypercalcemia
5.Infantile hypercalcemia with novel compound heterozygous mutation in SLC34A1 encoding renal sodium-phosphate cotransporter 2a: a case report
Seok Jin KANG ; Rosie LEE ; Heung Sik KIM
Annals of Pediatric Endocrinology & Metabolism 2019;24(1):64-67
Idiopathic infantile hypercalcemia is characterized by hypercalcemia, dehydration, vomiting, and failure to thrive, and it is due to mutations in 24-hydroxylase (CYP24A1). Recently, mutations in sodium-phosphate cotransporter (SLC34A1) expressed in the kidney were discovered as an additional cause of idiopathic infantile hypercalcemia. This report describes a female infant admitted for evaluation of nephrocalcinosis. She presented with hypercalcemia, hypercalciuria, low intact parathyroid hormone level, and high 1,25-dihydroxyvitamin D3 level. Exome sequencing identified novel compound heterozygous mutations in SLC34A1 (c.1337G>A, c.1483C>T). The patient was treated with fluids for hydration, furosemide, a corticosteroid, and restriction of calcium/vitamin D intake. At the age of 7 months, the patient's calcium level was within the normal range, and hypercalciuria waxed and waned. Renal echogenicity improved on the follow-up ultrasonogram, and developmental delay was not noted. In cases of hypercalcemia with subsequent hypercalciuria, DNA analysis for SLC34A1 gene mutations and CYP24A1 gene mutations should be performed. Further studies are required to obtain long-term data on hypercalciuria and nephrocalcinosis.
Calcitriol
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Calcium
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Dehydration
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DNA
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Exome
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Failure to Thrive
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Female
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Follow-Up Studies
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Furosemide
;
Humans
;
Hypercalcemia
;
Hypercalciuria
;
Hypophosphatemia
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Infant
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Kidney
;
Nephrocalcinosis
;
Parathyroid Hormone
;
Reference Values
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Sodium-Phosphate Cotransporter Proteins
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Ultrasonography
;
Vitamin D
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Vitamin D3 24-Hydroxylase
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Vomiting
6.Primary hyperparathyroidism versus familial hypocalciuric hypercalcemia: a challenging diagnostic evaluation in an adolescent female
Mustafa TOSUR ; Monica E LOPEZ ; David L PAUL
Annals of Pediatric Endocrinology & Metabolism 2019;24(3):195-198
Primary hyperparathyroidism (PHPT) and familial hypocalciuric hypercalcemia (FHH) have significantly different treatment approaches, so physicians must be careful to differentiate these 2 diseases. Herein, we report a 14-year-old female who presented with symptomatic hypercalcemia (12 mg/dL; reference range, 9.2–10.7 mg/dL), elevated intact parathyroid hormone (iPTH) (236 pg/mL; reference range, 9–69 pg/mL), and vitamin D deficiency (6 ng/mL; reference range, ≥ 20 ng/mL). On numerous occasions, her 24-hour urine calcium was more than 4 mg/kg/day, consistent with PHPT, but her fractional excretion of calcium on 24-hour urine collection was consistently below 1%, in line with FHH. ⁹⁹mTc-Sestamibi scan failed to detect any abnormalities. However, a 4-dimensional computed tomography scan of the neck revealed a right superior parathyroid adenoma which was excised with a focused parathyroidectomy. Although the patient’s calcium and iPTH levels normalized, her nonspecific symptoms persisted. This case illustrates both the challenges of differentiating PHPT from FHH and the limitations of a first-line imaging tool in identifying a parathyroid adenoma.
Adolescent
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Calcium
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Female
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Humans
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Hypercalcemia
;
Hyperparathyroidism, Primary
;
Neck
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Parathyroid Hormone
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Parathyroid Neoplasms
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Parathyroidectomy
;
Reference Values
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Urine Specimen Collection
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Vitamin D Deficiency
7.Polymeric Nanoparticles Containing Both Antigen and Vitamin D₃ Induce Antigen-Specific Immune Suppression
Ho Hyun JUNG ; Sang Hyun KIM ; Jun Hyeok MOON ; Seong Un JEONG ; Sundong JANG ; Chan Su PARK ; Chong Kil LEE
Immune Network 2019;19(3):e19-
The active form of vitamin D3, 1,25-dihydroxyvitamin D₃ (aVD₃), is known to exert beneficial effects in the treatment of autoimmune diseases because of its immunosuppressive effects. However, clinical application of aVD₃ remains limited because of the potential side effects, particularly hypercalcemia. Encapsulation of aVD₃ within biodegradable nanoparticles (NPs) would enhance the delivery of aVD₃ to antigen presenting cells, while preventing the potential systemic side effects of aVD₃. In the present study, polymeric NPs containing ovalbumin (OVA) and aVD₃ (NP[OVA+aVD₃]) were prepared via the water-in-oil-in-water double emulsion solvent evaporation method, after which their immunomodulatory effects were examined. Bone marrow-derived immature dendritic cells (DCs) treated with NP(OVA+aVD₃) did not mature into immunogenic DCs but were converted into tolerogenic DCs, which express low levels of co-stimulatory molecules and MHC class II molecules, produce lower levels of pro-inflammatory cytokines while increasing the production of IL-10 and TGF-β, and induce the generation of Tregs. Intravenous injection with NP(OVA+aVD₃) markedly suppressed the generation of OVA-specific CTLs in mice. Furthermore, OVA-specific immune tolerance was induced in mice orally administered with NP(OVA+aVD₃). These results show that biodegradable NPs encapsulating both antigen and aVD₃ can effectively induce antigen-specific immune suppression.
Animals
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Antigen-Presenting Cells
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Autoimmune Diseases
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Cholecalciferol
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Cytokines
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Dendritic Cells
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Hypercalcemia
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Immune Tolerance
;
Injections, Intravenous
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Interleukin-10
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Methods
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Mice
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Nanoparticles
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Ovalbumin
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Polymers
;
T-Lymphocytes, Regulatory
;
Vitamins
8.A Case of Hypercalcemia and Antibiotic-Related Acute Kidney Injury Following Implantation of Antibiotic Impregnated Calcium Sulfate Beads
Douglas CHALLENER ; Omar ABU SALEH
Annals of Laboratory Medicine 2019;39(5):507-508
No abstract available.
Acute Kidney Injury
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Calcium Sulfate
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Calcium
;
Hypercalcemia
9.Real-world Treatment Pattern and Outcomes of Hypercalcemia among Solid Tumor Patients
Da Eun SHIN ; Seol Hee PARK ; Sung Hwan KIM ; Sung Yun SUH ; Yun Hee JO ; Yoon Sook CHO ; Seock Ah IM ; Ju Yeun LEE
Korean Journal of Clinical Pharmacy 2019;29(3):166-172
BACKGROUND: Hypercalcemia is an important metabolic emergency condition in cancer patients. Bisphosphonate is the treatment of choice for hypercalcemia, whereas calcitonin and hydration with furosemide are recommended for acute supportive therapy. However, data regarding real-world treatment patterns and outcomes of pharmacological treatments are limited. Therefore, we aimed to investigate the treatment patterns and clinical outcomes of hypercalcemia treatment in solid tumor patients. METHODS: Electronic medical records of 123 adults with solid cancers and albumin-corrected calcium levels >10.5 mg/dL or ionized calcium levels >1.35 mmol/L were reviewed. We retrospectively analyzed the pharmacological treatment and recovery rate according to the severity of hypercalcemia. RESULTS: A total of 177 cases were identified, of which 49 were not treated and 30 were treated with hydration only. In moderate-to-severe cases, 86.5% received pharmacological treatment. Thirty-four cases (19.2%) were treated with bisphosphonate alone and 58 cases (32.8%) were treated with bisphosphonate and calcitonin. In mild hypercalcemia cases, the recovery rate was higher for those receiving hydration only or pharmacological treatment (79.7%) than for those receiving no treatment (61.4%, p = 0.041). Most moderate-to-severe cases were treated with medication and of those treated, 56.3% recovered. The recovery rate was lower in those treated with bisphosphonate alone (38.2%) than in those who underwent calcitonin combination treatment (73.7%, p = 0.001). CONCLUSIONS: Bisphosphonate combined with calcitonin was found to be more effective than bisphosphonate alone for the treatment of moderate-to-severe hypercalcemia. Considering the current shortage of calcitonin, further efforts are required to ensure its stable supply.
Adult
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Calcitonin
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Calcium
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Electronic Health Records
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Emergencies
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Furosemide
;
Humans
;
Hypercalcemia
;
Retrospective Studies
10.Parathyroid Carcinoma: Analysis of patient characteristics and outcomes in a retrospective review of eight cases seen in a single center
Shamira Shahar ; Kim Piow Lim ; Masni Mohamad
Journal of the ASEAN Federation of Endocrine Societies 2019;34(2):229-232
Eight cases of parathyroid carcinoma were identified (8 females; median age 45 years, range 28-72). Half of whom were diagnosed preoperatively. Hypercalcemic symptoms were seen in 87.5% of the patients and the main complication was nephrolithiasis. At presentation, the median calcium was 3.675 mmol/L, median phosphate of 0.68 mmol/L, median intact parathyroid hormone (iPTH) was 211 pmol/L. Five patients had regional nodes metastasis and 1 had distant metastasis to the lungs. Parathyroid gland invasion to adjacent structures was seen in 62.5% of cases while another 62.5% showed capsular or vascular infiltration on histology with median tumour size of 3.2 cm. Recurrent hypercalcemia occurred in 50% of the patients with median time of recurrence of 21 months. In this case series, we found that patients with severe hypercalcemia and high iPTH also exhibited a high index suspicion of PC.
Parathyroid Neoplasms
;
Carcinoma
;
Hypercalcemia


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