Dataset Information

Craniofacial Brown Tumor in Patients with Secondary Hyperparathyroidism to Chronic Renal Failure: Report of Two Cases in Cipto Mangunkusumo Hospital.

ABSTRACT: Brown tumor is a bone lesion that arises in the setting of excess osteoclast activity in hyperparathyroidism. It consists of fibrous tissue, woven bone, and supporting vasculature, while contains no matrix. The characteristic of brown-colored lesion is a result of hemosiderin deposition into the osteolytic cysts. Two cases of young women aged 26 and 29 years old, respectively, are known with a history of end-stage renal disease (ESRD). Dialysis is performed two times/week over the last 7 years. Our patients presented with an intraoral mass of the hard palate since 12 months ago and decreased body height of 10?cm. The lesion causes difficulties in swallowing and talking. Laboratory workup showed elevated parathormone or PTH (3.391?pg/mL and >5.000?pg/mL). Neck ultrasound showed enlargement of the parathyroid glands. Supporting examination to diagnose brown tumor are neck ultrasound, CT of the neck, and parathyroid sestamibi scan. We performed parathyroidectomy. Pathology revealed hyperplasia of the parathyroid. The tumor regressed significantly within 2 weeks following the surgery, and we still observe tumor regression as well as reduction in PTH level. As clinicians, we should be alert to other possible causes of bony lesions. Clinical examination, laboratory finding, and imaging present important information to diagnose brown tumor.

SUBMITTER: Kartini D

PROVIDER: S-EPMC6157168 | biostudies-other | 2018

REPOSITORIES: biostudies-other

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Craniofacial Brown Tumor in Patients with Secondary Hyperparathyroidism to Chronic Renal Failure: Report of Two Cases in Cipto Mangunkusumo Hospital.

Kartini Diani D Siswiandari Maria K MK Wibisana Gunawan G Yulian Erwin D ED Kurnia Ahmad A Panigoro Sonar S SS Albar Azdi Z AZ Ramli Muchlis M

Case reports in oncological medicine 20180912

Brown tumor is a bone lesion that arises in the setting of excess osteoclast activity in hyperparathyroidism. It consists of fibrous tissue, woven bone, and supporting vasculature, while contains no matrix. The characteristic of brown-colored lesion is a result of hemosiderin deposition into the osteolytic cysts. Two cases of young women aged 26 and 29 years old, respectively, are known with a history of end-stage renal disease (ESRD). Dialysis is performed two times/week over the last 7 years. ...[more]

PMID: 30298112

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Project description:BackgroundSagliker syndrome is caused by severe secondary hyperparathyroidism in hemodialysis patients with chronic renal failure. It is mainly manifested by disturbances of calcium and phosphorus metabolism and bone changes, which eventually lead to abnormal changes in facial appearance and morphology, seriously affecting the quality of life of patients. With the improvement of management mode and technology for hemodialysis in chronic renal failure, the incidence of severe secondary hyperparathyroidism related to hemodialysis is relatively reduced. Therefore, Sagliker syndrome is more rare. How to early identify and choose the appropriate treatment for Sagliker syndrome is particularly important.Case descriptionA 34-year-old female patient with uremia, who underwent regular hemodialysis at the Blood Purification Center of Tianjin Third Central Hospital, developed Sagliker syndrome. The general data, clinical symptoms, height changes, biochemical indicators (serum calcium, phosphorus, alkaline phosphatase, parathyroid hormone, hemoglobin, and hematocrit), and parathyroid emission computerized tomography (ECT) were collected and analyzed. The patient had a history of hemodialysis with chronic renal failure for 6 years, and presented with bone pain for 2 years. Laboratory tests showed parathyroid hormone was 2,269 pg/mL and ECT showed 3 parathyroid hyperplasia. Finally, total parathyroidectomy and forearm transplantation were performed. The level of parathyroid hormone was significantly lower than that before the operation. On the first postoperative day, the level of parathyroid hormone decreased to 28.3 pg/mL. Four months later, the bone pain symptoms of the patient were significantly improved compared with those before the operation.ConclusionsSagliker syndrome is a special syndrome in maintenance hemodialysis patients with refractory secondary hyperparathyroidism. Early monitoring and standardized treatment of secondary hyperparathyroidism may prevent the occurrence of Sagliker syndrome. Early identification and diagnosis of Sagliker syndrome and the choice of appropriate treatment will have an important impact for the prognosis.

Project description:Secondary hyperparathyroidism contributes to post-transplant CKD mineral and bone disorder. Paricalcitol, a selective vitamin D receptor activator, decreased serum parathyroid hormone levels and proteinuria in patients with secondary hyperparathyroidism. This single-center, prospective, randomized, crossover, open-label study compared the effect of 6-month treatment with paricalcitol (1 μg/d for 3 months and then uptitrated to 2 µg/d if tolerated) or nonparicalcitol therapy on serum parathyroid hormone levels (primary outcome), mineral metabolism, and proteinuria in 43 consenting recipients of renal transplants with secondary hyperparathyroidism. Participants were randomized 1:1 according to a computer-generated sequence. Compared with baseline, median (interquartile range) serum parathyroid hormone levels significantly declined on paricalcitol from 115.6 (94.8-152.0) to 63.3 (52.0-79.7) pg/ml (P<0.001) but not on nonparicalcitol therapy. At 6 months, levels significantly differed between treatments (P<0.001 by analysis of covariance). Serum bone-specific alkaline phosphatase and osteocalcin decreased on paricalcitol therapy only and significantly differed between treatments at 6 months (P<0.001 for all comparisons). At 6 months, urinary deoxypyridinoline-to-creatinine ratio and 24-hour proteinuria level decreased only on paricalcitol (P<0.05). L3 and L4 vertebral mineral bone density, assessed by dual-energy x-ray absorption, significantly improved with paricalcitol at 6 months (P<0.05 for both densities). Paricalcitol was well tolerated. Overall, 6-month paricalcitol supplementation reduced parathyroid hormone levels and proteinuria, attenuated bone remodeling and mineral loss, and reduced eGFR in renal transplant recipients with secondary hyperparathyroidism. Long-term studies are needed to monitor directly measured GFR, ensure that the bone remodeling and mineral effects are sustained, and determine if the reduction in proteinuria improves renal and cardiovascular outcomes.

Dataset Information

Craniofacial Brown Tumor in Patients with Secondary Hyperparathyroidism to Chronic Renal Failure: Report of Two Cases in Cipto Mangunkusumo Hospital.

Publications

Craniofacial Brown Tumor in Patients with Secondary Hyperparathyroidism to Chronic Renal Failure: Report of Two Cases in Cipto Mangunkusumo Hospital.

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