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1.
Osteoporos Int ; 35(1): 195-199, 2024 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-37644196

RESUMO

Bone lytic lesions are a possible complication of pseudohypoparathyroidism type 1B, in undertreated adult patients. Whole body [18F] F-fluorocholine PET/CT is a useful imaging tool to assess brown tumor progression in this context. We describe the case of a 33-year-old woman, referred for the diagnostic evaluation of lytic bone lesions of the lower limbs, in the context of asymptomatic pseudohypoparathyroidism. She had been treated with alfacalcidol and calcium during her childhood. Treatment was discontinued at the age of 18 years old because of the lack of symptoms. A femur biopsy revealed a lesion rich in giant cells, without malignancy, consistent with a brown tumor. Laboratory tests showed a parathyroid level at 1387 pg/ml (14-50). Whole-body Fluorocholine PET/CT revealed hypermetabolism of bone lesions. The final diagnosis was brown tumors related to hyperparathyroidism complicating an untreated pseudohypoparathyroidism. Genetic testing confirmed PHP type 1B. Pseudohypoparathyroidism with radiographic evidence of hyperparathyroid bone disease, is a very rare condition due to parathyroid hormone resistance in target organs, i.e., kidney resistance, but with conserved bone cell sensitivity. It has been reported in only a few cases of pseudohypoparathyroidism type Ib. Long-term vitamin D treatment was required to correct bone hyperparathyroidism. With this rationale, the patient was treated with calcium, alfacalcidol, and cholecalciferol. One-year follow-up showed complete resolution of pain, improvement in serum calcium, and regression of bone lesions on [18F]F-fluorocholine PET/CT. This case illustrates the usefulness of [18F]F-fluorocholine PET/CT for the imaging of brown tumors in pseudohypoparathyroidism type 1B, and emphasizes the importance of calcium and vitamin D treatment in adult patients, to avoid the deleterious effects of high parathyroid hormone on skeletal integrity.


Assuntos
Doenças Ósseas , Colina/análogos & derivados , Hiperparatireoidismo , Neoplasias , Osteíte Fibrosa Cística , Pseudo-Hipoparatireoidismo , Humanos , Adulto , Feminino , Criança , Adolescente , Cálcio/uso terapêutico , Tomografia por Emissão de Pósitrons combinada à Tomografia Computadorizada , Osteíte Fibrosa Cística/complicações , Pseudo-Hipoparatireoidismo/complicações , Hormônio Paratireóideo , Hiperparatireoidismo/complicações , Vitaminas , Vitamina D/uso terapêutico
2.
BMC Nephrol ; 25(1): 15, 2024 01 05.
Artigo em Inglês | MEDLINE | ID: mdl-38182973

RESUMO

BACKGROUND: Tertiary hyperparathyroidism describes the autonomous and excessive secretion of parathyroid hormone (PTH) by the parathyroid glands after longstanding secondary hyperparathyroidism in chronic kidney disease. Brown tumors are a sign of uncontrolled hyperparathyroidism. In this case, we have reported a refractory and destructive hyperparathyroidism storm. Also, it presented with atypical onset and unexpected adenoma location. CASE PRESENTATION: A 37-year-old man was diagnosed with end-stage kidney disease 22 years ago. He has been undergoing dialysis treatment since that time. Recently, he was admitted to the ophthalmology department due to the unilateral anterior bulging of the right eye and drooping of the eyelid. Magnetic resonance imaging exhibited an extraconal mass lesion located in the right orbital posterior superolateral position. Computerized tomography scans considered expansile bone lesion with peripheral calcification and originating from the sphenoid wing. The bone mass lesion was resected via craniotomy due to the compressive effect. The pathological findings were consistent with brown tumors. Plasma intact PTH level was 4557 pg/mL. The patient informed that he underwent parathyroidectomy and two leg fractures operation in a medical query. Parathyroid scintigraphy determined three distinct foci consistent with adenomas and one of them was in mediastenum. Second parathyroidectomy was recommended to the patient but the patient refused surgery. Despite his medication and dialysis regimen being revised, PTH levels were maintained at higher levels in follow-up. CONCLUSIONS: We presented a hyperparathyroidism case that was resistant to all treatments and exhibited all the severe complications in a long-term dialysis patient. Furthermore, this case has revealed the importance and difficulty of secondary hyperparathyroidism management.


Assuntos
Hiperparatireoidismo Secundário , Neoplasias , Osteíte Fibrosa Cística , Masculino , Humanos , Adulto , Diálise Renal , Hiperparatireoidismo Secundário/complicações , Hiperparatireoidismo Secundário/diagnóstico por imagem , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Glândulas Paratireoides/diagnóstico por imagem
3.
Arch Orthop Trauma Surg ; 144(7): 2927-2934, 2024 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-38795187

RESUMO

INTRODUCTION: Brown tumors are reactive osteolytic lesions caused by hyperparathyroidism. These rare lesions are non-neoplastic processes that result from bone resorption. The purpose of this study was to retrospectively review a 34-year experience with brown tumors in our institution. MATERIALS AND METHODS: We retrospectively analyzed the records of 26 consecutive patients with brown tumor who were treated in our institution between May 1988 and October 2020, with a mean follow-up of 36,1 months. RESULTS: 17 male (65,4%) and 9 female (34,6%) patients with a mean age of 41,6 were included in the study. Localized bone pain was present in 13 cases (50,0%) as the first presenting symptom. 3 patients (11,5%) presented with diffuse bone pain. 7 patients (26,9%) were diagnosed with brown tumor while being investigated for pathological fractures. The other 3 patients (11,5%) were diagnosed while being evaluated for hypercalcemia symptoms. 7 patients (26,9%) had solitary lesions, while 19 patients (73,1%) had multiple lesions. Pelvis, femur, ribs, tibia, proximal humerus and mandible were the most common sites of localization. 23 patients (88,5%) were diagnosed with primary hyperparathyroidism, while the other 3 patients (11,5%) had secondary hyperparathyroidism. A total of the 65 lesions, 23 (35.4%) underwent orthopedic surgery, and 42 (64.6%) were followed up conservatively after parathyroidectomy. Orthopedic surgery was performed in 21 of 26 patients, the other 5 cases were followed up conservatively. Intralesional curettage was performed in 19 cases (82,6%). The resulting cavity was filled with bone cement in 11 cases (47,8%). Bone grafting was applied in 8 cases (34,8%). No recurrence was observed in any of the patients. CONCLUSION: The diagnosis of brown tumor begins with clinical suspicion. Endocrinology and general surgery consultation is important before surgery. Treatment of brown tumors requires a multidisciplinary approach.


Assuntos
Hiperparatireoidismo , Humanos , Masculino , Estudos Retrospectivos , Feminino , Adulto , Pessoa de Meia-Idade , Adulto Jovem , Idoso , Adolescente , Hiperparatireoidismo/cirurgia , Hiperparatireoidismo/complicações , Hiperparatireoidismo/etiologia , Osteíte Fibrosa Cística/etiologia
4.
BMC Endocr Disord ; 23(1): 214, 2023 Oct 08.
Artigo em Inglês | MEDLINE | ID: mdl-37807045

RESUMO

BACKGROUND: Primary hyperparathyroidism (PHPT) is an uncommon disorder characterised by hypercalcemia with an increased parathyroid hormone level. We reported a PHPT familial case with two subjects, a father and a daughter, and both of them had suffered from the brown tumor. CASE PRESENTATION: The proband, a 43-year-old patient, developed parathyroid adenomas at the age of 15; a histologically confirmed right parathyroid adenoma was removed by parathyroidectomy; and after six months follow-up, the serum calcium level was normalised. At the age of thirty-three, a CT scan of his head and neck revealed a mass in the right maxilla, as well as PHPT (i.e., left inferior parathyroid adenoma). Then, he underwent a biopsy of an exophytic lesion in the right maxilla and was diagnosed by pathology as a brown tumor, with the serum calcium and PTH levels at 2.78 mmol/L and 221 pg/mL, respectively. Subsequently, the patient took a left inferior parathyroid microwave ablation with ultrasound guidance. After three months of follow-up, the serum calcium and PTH levels returned to normal, and the brown tumor was resolved. After three years, it mineralised as revealed in a CT scan. By the time he was 43 years old, during the 28-year follow-up period, the serum calcium and PTH levels were still within the normal range, and there was no discomfort reported. He has consistently taken calcium supplements throughout the 28 years. Since the initial diagnosis, his blood indicators of kidney function have been normal, and ultrasound showed renal calculus in the right kidney and a normal left kidney. The proband's daughter, a 15-year-old girl, experienced left upper extremity pain for ten months. CT scan revealed a mass in the distal left radius, and a giant cell tumor was suspected. A surgical internal fixation was performed, and the pathology showed a brown tumor. Laboratory tests revealed a serum parathyroid hormone (PTH) level of 1554pg/mL, calcium level of 3.14 mmol/L, phosphorus level of 0.72 mmol/L, and alkaline phosphatase level of 1892 U/L. Given the osteitic changes and elevated levels of calcium and PTH, ultrasonography was performed, after which a mass was detected measuring 19 × 9 × 7 mm mixed with solid components and cystic fluid in the right thyroid gland. The results of 99mTc-MIBI scintigraphy confirmed the abnormal accumulation of 99mTc-MIBI in the right thyroid gland but not seen in the bilateral parathyroid glands. The patient underwent thyroidectomy, and the postoperative pathology report indicated an intra-thyroid ectopic parathyroid adenoma. The serum calcium and PTH levels became normal at 4 h after surgery. One to three months after operation, the serum calcium level was low, while the serum PTH level was high. Then, the patient was advised to take calcium supplements. Until the sixth month after the operation, the serum calcium level and serum PTH level returned to normal, and the bone pain was relieved. The patient's blood tests for kidney function remained normal. There was no evidence of bilateral kidney disease (such as nephrolithiasis or nephrocalcinosis) detected by ultrasound scan. There were several similarities in the state of illness between these two subjects. Both the father and the daughter developed parathyroid adenomas at the age of 15, and there was no lesion in other endocrine glands. And genetic testing revealed mutations in the CDC73 genes in both father and daughter. On the other hand, there were also a few differences. The father's first signs of brown tumor were in the right maxilla, while the daughter's appeared in the distal left radius. The father presented pathological changes in the left and right parathyroid glands, whereas the daughter presented with an ectopic parathyroid adenoma in the right thyroid gland. CONCLUSION: We report a familial case in which father and daughter were diagnosed to have brown tumors due to parathyroid adenoma and ectopic parathyroid adenoma, and genetic testing revealed CDC73 gene mutations in both. Therefore, in the diagnostic and differential process of young patients having bone disease, clinicians should not only focus on the clinical manifestations of the skeleton, but also implement a comprehensive analysis of systemic symptoms, considering the possibility that the patient has familial PHPT.


Assuntos
Hiperparatireoidismo Primário , Osteíte Fibrosa Cística , Neoplasias das Paratireoides , Masculino , Feminino , Humanos , Adolescente , Adulto , Hiperparatireoidismo Primário/complicações , Cálcio , Neoplasias das Paratireoides/patologia , Hormônio Paratireóideo , Dor
5.
Cancer Invest ; 40(6): 544-553, 2022 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-35275786

RESUMO

Multiple myeloma and its precursor and variant types represent some of the most common hematologic malignancies in adults. These plasma cell dyscrasias are well-known in modern medicine. There are well-established clinical, laboratory, and pathologic criteria for diagnosis and staging. There is debate about the diagnosis of some of the earliest cases of myeloma described in the literature. We present a critical review of one such case.


Assuntos
Mieloma Múltiplo , Osteíte Fibrosa Cística , Adulto , Humanos , Mieloma Múltiplo/diagnóstico
6.
BMC Endocr Disord ; 22(1): 66, 2022 Mar 14.
Artigo em Inglês | MEDLINE | ID: mdl-35287634

RESUMO

BACKGROUND: Brown tumour is a rare tumour-like lesion of the bone, which is considered as an end-stage lesion of abnormal bone metabolism caused by persistently high parathyroid hormone (PTH) levels. Brown tumour can be found in any part of the skeleton; in some cases, it can occur in multiple bones and can be easily misdiagnosed as a metastatic tumour. CASE PRESENTATION: We report the case of a 44-year-old man who presented to the Department of Oncology in our hospital with a 2-month history of local pain in his left shoulder joint. The initial diagnosis was an aneurysmal bone cyst by biopsy, for which the patient underwent tumour resection surgery. The diagnosis of a malignant tumour was made again following postoperative pathological examination. The pathological sections and all clinical data were sent to the Department of Pathology of the First Affiliated Hospital of Sun Yat-sen University; the diagnosis made there was brown tumour. His blood PTH level was 577 pg/ml (15-65 pg/ml). Colour Doppler ultrasonography of the parathyroid gland suggested a parathyroid adenoma. For further treatment, the left parathyroid adenoma was removed by axillary endoscopic resection. Postoperatively, a pathologic examination was performed, and the diagnosis of a parathyroid adenoma was confirmed. One year after the surgery, the left humerus was completely healed, and the left shoulder joint had a good range of movement. CONCLUSIONS: In summary, histopathological diagnosis is not sufficient for the diagnosis of brown tumours. A comprehensive analysis combining clinical symptoms with findings of imaging and laboratory tests is also required. Generally, the treatment of brown tumour includes only partial or complete resection of the parathyroid glands. However, when the tumour is large, especially when it involves the joint, surgery is indispensable.


Assuntos
Hiperparatireoidismo Primário/diagnóstico , Osteíte Fibrosa Cística/diagnóstico , Adulto , Erros de Diagnóstico , Humanos , Hiperparatireoidismo Primário/complicações , Masculino , Osteíte Fibrosa Cística/etiologia
7.
BMC Musculoskelet Disord ; 23(1): 322, 2022 Apr 04.
Artigo em Inglês | MEDLINE | ID: mdl-35379205

RESUMO

BACKGROUND: Osteitis fibrosa cystica is a rare, benign and osteolytic lesion attributed to hyperparathyroidism. The high level of parathyroid hormone cause rapid bone loss. CASE PRESENTATION: The patient is a 50-year-old male complaining of severe and persistent pain in the right knee joint. Imaging studies were suspicious for a benign tumor in the right distal femur. Biopsy under CT guidance showed numerous osteoclast aggregation and hemosiderin deposition around the bone trabeculae. Blood tests disclosed significantly elevated parathyroid hormone, serum calcium, serum alkaline phosphatase. Parathyroid ultrasonography and CT scan showed a solid mass in front of the trachea at the thoracic entrance plane. After resection of the mass, the clinical symptoms were relieved and the radiological results were significantly improved, which further confirmed the diagnosis. CONCLUSIONS: Metabolic diseases-associated bone lesions require a comprehensive diagnosis of multiple inspection items. An interprofessional team approach to the diagnosis and treatment of osteitis fibrosa cystica will provide the best outcome.


Assuntos
Neoplasias Ósseas , Hiperparatireoidismo , Osteíte Fibrosa Cística , Neoplasias das Paratireoides , Neoplasias Ósseas/diagnóstico , Neoplasias Ósseas/diagnóstico por imagem , Fêmur/diagnóstico por imagem , Fêmur/patologia , Fêmur/cirurgia , Humanos , Hiperparatireoidismo/complicações , Masculino , Pessoa de Meia-Idade , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Neoplasias das Paratireoides/diagnóstico , Neoplasias das Paratireoides/diagnóstico por imagem
8.
Osteoporos Int ; 32(1): 205-208, 2021 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-32772143

RESUMO

We present the case of a 65-year-old man with brown tumors due to secondary hyperparathyroidism. Magnetic resonance imaging of the pelvis showed multiple lesions with expansive bone appearance. Additionally, prostate cancer was diagnosed during this time. For this reason, differential diagnosis was performed through biopsy of the right iliac bone lesion. Brown tumors are caused by osteoclastic activity and fibroblast proliferation; the differential diagnosis of these bone lesions includes giant tumors, metastases, Paget's disease, and paraneoplastic syndrome with high levels of parathyroid hormone-related peptide (PTHrP). This case report describes the coexistence of two pathologies that could explain these images. In this report, we present a case of a 65-year-old man with brown tumors due to secondary hyperparathyroidism and prostate cancer. In this setting, histologic confirmation is recommended.


Assuntos
Hiperparatireoidismo Secundário , Osteíte Deformante , Osteíte Fibrosa Cística , Neoplasias da Próstata , Idoso , Diagnóstico Diferencial , Humanos , Hiperparatireoidismo Secundário/complicações , Masculino , Osteíte Deformante/complicações , Osteíte Fibrosa Cística/complicações , Osteíte Fibrosa Cística/diagnóstico , Neoplasias da Próstata/complicações
9.
BMC Nephrol ; 22(1): 298, 2021 09 03.
Artigo em Inglês | MEDLINE | ID: mdl-34479496

RESUMO

BACKGROUND: No publications have reported on osteomalacia in patients receiving intermittent cyclical therapy with etidronate (a bisphosphonate) and undergoing long-term hemodialysis (HD). CASE PRESENTATION: We report on a 46-year-old Japanese man admitted to our hospital for further examination of left forearm pain. Maintenance HD was started at age 24 years, and the man had been on HD since then. At age 38 years, surgical parathyroidectomy was performed for secondary hyperparathyroidism; iliac crest bone biopsy performed at the same time showed osteitis fibrosa. The active vitamin D3 preparation calcitriol was started, and intermittent cyclical etidronate therapy was introduced 2 years later for osteoporosis. At age 45 years, the patient stopped taking calcitriol because of hypercalcemia but continued with etidronate. At age 46 years, a pseudofracture with a Looser zone occurred in the left ulna, and left femur bone biopsy revealed osteomalacia. Etidronate was discontinued, and calcitriol was restarted; open reduction and internal fixation with an angular stability plate were performed. Union of the bone was achieved 10 months after the operation. At age 49 years, a lumber bone biopsy confirmed improved bone morphometry. CONCLUSIONS: We believe that intermittent cyclical etidronate therapy without administration of active vitamin D3 during long-term HD might have induced osteomalacia, resulting in the ulna insufficiency fracture. Therefore, we propose that administration of active vitamin D3 is essential to prevent osteomalacia in patients on long-term HD who are receiving bisphosphonates and have potential vitamin D3 deficiency.


Assuntos
Conservadores da Densidade Óssea/efeitos adversos , Ácido Etidrônico/efeitos adversos , Osteomalacia/induzido quimicamente , Diálise Renal , Conservadores da Densidade Óssea/uso terapêutico , Osso e Ossos/diagnóstico por imagem , Calcitriol/uso terapêutico , Colecalciferol/uso terapêutico , Ácido Etidrônico/uso terapêutico , Humanos , Ílio/patologia , Masculino , Pessoa de Meia-Idade , Osteíte Fibrosa Cística/tratamento farmacológico , Deficiência de Vitamina D/complicações , Deficiência de Vitamina D/tratamento farmacológico
10.
BMC Musculoskelet Disord ; 22(1): 479, 2021 May 25.
Artigo em Inglês | MEDLINE | ID: mdl-34034731

RESUMO

BACKGROUND: We report a case of osteitis fibrosa cystica, a rare benign resorptive bone lesion caused by hyperparathyroidism, that presented on imaging as an aggressive bone tumor. CASE PRESENTATION: The patient is a 51-year-old male complaining of severe sustained pain of the right hip region. Imaging studies were suspicious for a malignant tumor of the right iliac bone. Biopsy under CT guidance was performed and showed remodeled bone trabeculae with numerous osteoclasts, excluding bone tumor and raising the possibility of osteitis fibrosa cystica. Complementary tests disclosed elevated blood level of parathyroid hormone and a partially cystic enlarged left inferior parathyroid gland consistent with adenoma. After parathyroidectomy, the clinical symptoms were relieved and the radiological findings were significantly improved, which confirmed the diagnosis. CONCLUSIONS: Metabolic diseases-associated bone lesions should always be considered in the differential diagnosis of bone tumors, to avoid unnecessary surgeries and treatments.


Assuntos
Neoplasias Ósseas , Hiperparatireoidismo , Osteíte Fibrosa Cística , Neoplasias Ósseas/diagnóstico por imagem , Diagnóstico Diferencial , Humanos , Hiperparatireoidismo/diagnóstico , Hiperparatireoidismo/cirurgia , Masculino , Pessoa de Meia-Idade , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Hormônio Paratireóideo , Paratireoidectomia
11.
BMC Musculoskelet Disord ; 22(1): 443, 2021 May 14.
Artigo em Inglês | MEDLINE | ID: mdl-33990191

RESUMO

BACKGROUND: Osteitis fibrosa cystica is the classic manifestation of primary hyperparathyroidism (PHPT), occurs after prolonged exposure of bone to high serum parathyroid hormone (PTH) level. It has become increasingly rare due to early detection of PHPT. CASE PRESENTATION: A 37-year-old woman was referred to our institution for fixation of multiple fractures of upper and lower extremities that had been reoccurring in the past 5 years. Her medical history showed right-shoulder, left-elbow, and right-femur fractures after a fall 5 years previously. One month ago, she sustained fractures of the right distal humerus, left tibia, and left femur without history of trauma. Upon arrival to our hospital, a thorough review of her plain radiographs demonstrated brown tumors at multiple sites, along with a salt-and-pepper appearance of the skull and a rugger-jersey spine, compatible with osteitis fibrosa cystica. Patient was diagnosed with PHPT, confirmed by high-corrected serum calcium (13.6 [8.6-10.0] mg/dl), low serum phosphate (2.2 [2.5-4.5] mg/dL), high serum alkaline phosphatase (1482 [35-105] U/L), and significantly elevated parathyroid hormone (PTH 3850 [15-65] pg/mL). A histologically confirmed, 2.5-cm parathyroid adenoma was removed by parathyroidectomy. Ten days later, closed reduction and internal fixation of the left proximal femoral shaft was performed. Pain and ambulation were significantly improved 6 months postoperatively. At the 1.5-year follow-up, fracture unions and complete mineralization of brown tumors were noted; the patient could ambulate with neither pain nor an assistive device. CONCLUSIONS: PHPT has become more asymptomatic in countries where routine calcium screening is performed. Nevertheless, the classic skeletal involvement, osteitis fibrosa cystica, should not be overlooked, particularly in young patients who present with a low-energy fracture.


Assuntos
Fraturas Espontâneas , Hiperparatireoidismo Primário , Osteíte Fibrosa Cística , Neoplasias das Paratireoides , Adulto , Feminino , Fraturas Espontâneas/diagnóstico por imagem , Fraturas Espontâneas/etiologia , Fraturas Espontâneas/cirurgia , Humanos , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico por imagem , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Hormônio Paratireóideo
12.
J Pak Med Assoc ; 71(4): 1266-1269, 2021 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-34125787

RESUMO

Brown tumour (BT) is the pathological expression of osteitis fibrosa cystica which is caused due to primary and secondary hyperparathyroidism (HPT). It is a rare benign lesion of skeletal system that usually affects the facial bones, clavicles, ribs, pelvis and extremities. The purpose of this case report is to present the clinical, pathological and radiological findings of BT - rarely seen in adults - originating from the giant parathyroid adenoma and emerging as the first clinical sign of HPT. The patient underwent a successful parathyroidectomy and was discharged on the first postoperative day without any complications. Histopathologically, it was confirmed as parathyroid adenoma. With the increased use of biochemical tests, HPT is diagnosed and treated early and so the frequency of symptomatic HPT due to parathyroid adenoma has decreased in developed countries, but we still sometimes encounter cases of primary hyperparathyroidism (PHPT) occurring as BT. With multidisciplinary preoperative evaluation, surgical resection of bones is refrained from in order to prevent unnecessary morbidity and mortality, unless these osteolytic bone lesions cause a pathological fracture.


Assuntos
Fraturas Espontâneas , Hiperparatireoidismo Primário , Osteíte Fibrosa Cística , Neoplasias das Paratireoides , Adulto , Humanos , Hiperparatireoidismo Primário/diagnóstico , Hiperparatireoidismo Primário/etiologia , Hiperparatireoidismo Primário/cirurgia , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Neoplasias das Paratireoides/diagnóstico , Neoplasias das Paratireoides/diagnóstico por imagem , Paratireoidectomia
13.
BMC Endocr Disord ; 20(1): 6, 2020 Jan 13.
Artigo em Inglês | MEDLINE | ID: mdl-31931802

RESUMO

BACKGROUND: Primary hyperparathyroidism is characterized by hypercalcemia and elevated or inappropriately normal serum levels of parathyroid hormone. Brown tumor of bone is a rare non-neoplastic lesion resulted from abnormal bone metabolism in hyperparathyroidism. However, nowadays, skeletal disease caused by primary hyperparathyroidism is uncommon. We report a case of brown tumor in the mandible as the initial exhibition of primary hyperparathyroidism associated with an atypical parathyroid adenoma. CASE PRESENTATION: The patient was a 49-year-old female, she had a pain mass on the right mandible a year ago and was treated with root canal therapy and marginal resection. After seven months, the mass recurred and enlarged. Enhanced CT scan, laboratory examination, Ultrasonography, 99mTc-MIBI SPECT-CT scintiscan and pathological examination were used to confirm the diagnosis of brown tumor. The patient's symptom improved after parathyroidectomy. CONCLUSIONS: 99mTc-MIBI SPECT/CT scintigraphy is a highly sensitive examination of the localization diagnosis of hyperparathyroidism. Brown tumors should be considered in the differential diagnosis of osteolytic lesions to avoid unnecessary and harmful interventions.


Assuntos
Hiperparatireoidismo Primário/diagnóstico , Neoplasias Mandibulares/diagnóstico , Osteíte Fibrosa Cística/diagnóstico , Diagnóstico Diferencial , Feminino , Humanos , Hiperparatireoidismo Primário/cirurgia , Neoplasias Mandibulares/cirurgia , Pessoa de Meia-Idade , Osteíte Fibrosa Cística/cirurgia , Paratireoidectomia , Prognóstico
14.
Endocr Pract ; 26(9): 983-989, 2020 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-33471703

RESUMO

OBJECTIVE: Clinically overt symptomatic bone disease in primary hyperparathyroidism (PHPT) is rarely seen today, and osteoporosis is the dominant finding. Subperiosteal bone resorption in the fingers and skull mottling are typical bone PHPT findings, the contemporary prevalence of which is unknown. We evaluated these mild lesions and investigated the impact of their occurrence on PHPT clinical management. METHODS: We evaluated retrospectively a monocentric series of 363 PHPT patients classified in Group 1 (n = 100) or Group 2 (n = 263) according to the presence or absence of bone involvement, respectively. Patients belonging to Group 1, in turn, were subdivided into Group 1A, with severe and symptomatic lesions (n = 48), and Group 1B, with milder signs of osteitis fibrosa cystica (OFC) without brown tumors or fractures (n = 52). RESULTS: Serum total and ionized calcium, parathyroid hormone, osteocalcin, alkaline phosphatase, and its bone fraction levels were higher in Group 1 than in Group 2, while no gender or age differences were observed between the groups. Osteoporosis prevalence was similar in Group 1B and Group 2 patients but lower than in Group 1A. Mild OFC findings did not modify the surgical indication in any patient of Group 1B. CONCLUSION: Minor radiologic signs of OFC are not uncommon in the modern PHPT phenotype and occur in patients with more active disease. These signs could identify PHPT patients in an earlier stage of typical bone involvement. However, these features do not seem to upgrade either the clinical classification of asymptomatic PHPT patients or the propensity toward surgical choice.


Assuntos
Hiperparatireoidismo Primário , Osteíte Fibrosa Cística , Densidade Óssea , Cálcio , Humanos , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico por imagem , Hiperparatireoidismo Primário/epidemiologia , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/epidemiologia , Osteíte Fibrosa Cística/etiologia , Hormônio Paratireóideo , Prevalência , Estudos Retrospectivos
15.
BMC Pediatr ; 20(1): 547, 2020 12 05.
Artigo em Inglês | MEDLINE | ID: mdl-33278878

RESUMO

BACKGROUND: Primary hyperparathyroidism may present in a myriad of manners, varying from an incidental asymptomatic biochemical finding to gastrointestinal, psychiatric, renal, and bone manifestations. While hyperparathyroidism remains a rare diagnosis in the pediatric population, the initial approach to diagnosis and management of hypercalcemia in children is imperative for the general pediatrician. Herein, we describe an adolescent who presented with a lytic bone lesion and severe, symptomatic hypercalcemia due to primary hyperparathyroidism. CASE PRESENTATION: A 14-year-old male presented with vomiting, constipation, abdominal pain, and lethargy. He had an elevated total corrected calcium of 4.3 mmol/L. He was found to have a large pelvic lytic tumor consistent with a brown tumor due to primary hyperparathyroidism. He received pharmacologic therapy for stabilization of his hypercalcemia, including intravenous saline, intravenous bisphosphonates, and calcitonin. He subsequently received definitive therapy via parathyroidectomy and his post-operative course was complicated by hungry bone syndrome. Long-term follow-up has found full resolution of the lytic lesion and restored calcium homeostasis. CONCLUSIONS: We present this case to highlight the possible presentations of hypercalcemia and hyperparathyroidism that are essential for a general pediatrician to recognize to ensure prompt diagnosis and management. Evaluation for hypercalcemia should be considered in patients with suggestive symptoms and physical exam findings. To our knowledge, this patient represents the first reported pediatric case of a pelvic brown tumor in an adolescent. While the multi-systemic complications of hyperparathyroidism may be quite severe, swift and appropriate management may mitigate these clinical outcomes.


Assuntos
Hipercalcemia , Hiperparatireoidismo Primário , Osteíte Fibrosa Cística , Neoplasias das Paratireoides , Adolescente , Criança , Humanos , Hipercalcemia/diagnóstico , Hipercalcemia/etiologia , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico , Masculino , Osteíte Fibrosa Cística/diagnóstico , Osteíte Fibrosa Cística/etiologia , Osteíte Fibrosa Cística/cirurgia , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/diagnóstico , Neoplasias das Paratireoides/cirurgia , Paratireoidectomia
17.
BMC Nephrol ; 20(1): 425, 2019 11 21.
Artigo em Inglês | MEDLINE | ID: mdl-31752733

RESUMO

BACKGROUND: Pregnancy is rare in women on hemodialysis. Recommendations for the treatment of secondary hyperparathyroidism (sHPT) and preservation of bone health in pregnant dialysis patients are lacking. CASE PRESENTATION: We present the case of a young woman with end-stage kidney disease (ESKD) due to lupus nephritis, who developed multiple brown tumors while on hemodialysis during her second pregnancy. During her first pregnancy sHPT was well controlled and no skeletal complications occurred. Before the second pregnancy she developed severe sHPT. During pregnancy, dialysis time was increased to 24 h per week, the patient was given oral calcitriol, and the dialysate calcium concentration was set at 1.5 mmol/l. In week 20 the patient complained about bone pain in her left hip. Magnetic resonance imaging revealed a cystic lesion compatible with a brown tumor. The baby was delivered in the 36th week by cesarean section. Further assessment identified multiple brown tumors of her skeleton, including the acetabulum, tibia, ribs, skull, thoracic spine and thumb. She required multiple orthopedic surgeries. Three months after pregnancy, etelcalcetide was started, which brought about a gradual improvement in her sHPT. CONCLUSIONS: This case demonstrates that the combination of pregnancy and severe sHPT in dialysis patients can have deleterious consequences for bone health.


Assuntos
Hiperparatireoidismo Secundário/complicações , Falência Renal Crônica/terapia , Nefrite Lúpica/complicações , Osteíte Fibrosa Cística/etiologia , Complicações na Gravidez , Diálise Renal/efeitos adversos , Adulto , Cálcio/sangue , Feminino , Humanos , Hiperparatireoidismo Secundário/sangue , Hiperparatireoidismo Secundário/tratamento farmacológico , Falência Renal Crônica/sangue , Falência Renal Crônica/etiologia , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/cirurgia , Hormônio Paratireóideo/sangue , Peptídeos/uso terapêutico , Gravidez , Complicações na Gravidez/sangue , Complicações na Gravidez/terapia
18.
Br J Neurosurg ; 33(6): 684-686, 2019 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-29160114

RESUMO

Brown tumours affecting the cervical spine are a rare but recognised complication of renal failure-related secondary hyperparathyroidism. We present a case of a 26 year-old female with radiculopathy who was managed successfully with 360° cervical spine fixation and parathyroidectomy.


Assuntos
Vértebras Cervicais/cirurgia , Hiperparatireoidismo Secundário/complicações , Osteíte Fibrosa Cística/cirurgia , Neoplasias da Coluna Vertebral/cirurgia , Adulto , Feminino , Humanos , Hiperparatireoidismo Secundário/cirurgia , Osteíte Fibrosa Cística/complicações , Paratireoidectomia/métodos , Radiculopatia/etiologia , Radiculopatia/cirurgia , Neoplasias da Coluna Vertebral/complicações
19.
J Craniofac Surg ; 30(6): e551-e553, 2019 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-30939562

RESUMO

A 28-year-old African American female with end-stage renal disease on dialysis secondary to preeclampsia presented to the office as a referral for large multifocal tumors of maxilla and mandible. Surgical pathology and laboratory findings were supportive of secondary hyperparathyroidism leading to multifocal brown tumors. She underwent osteoplasty after using virtual surgical planning to create stereolithic models to visualize the tumor and fabricate cutting guides to minimize the risk of injury to adjacent nerves and teeth. Brown tumors can be resistant to medical management with unreliable regression in size. With the advent of customized surgical guides, more precise and judicious surgery can be performed on these types of tumors safely.


Assuntos
Osteíte Fibrosa Cística/cirurgia , Procedimentos de Cirurgia Plástica , Adulto , Feminino , Humanos , Mandíbula/cirurgia , Maxila/cirurgia
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