RESUMO
Variations in parathyroid gland positions often cause failure in initial parathyroid adenoma surgery, especially when imaging fails to localise the adenoma. This report describes a female patient with primary hyperparathyroidism for which preoperative localisation studies did not determine the position of the hyperfunctioning gland. The initial approach with bilateral cervical exploration and intraoperative parathyroid hormone monitoring was performed unsuccessfully. A mediastinal adenoma was suspected due to meticulous negative neck exploration and repeated negative images for a neck adenoma. Subsequently, a second approach involving mediastinal exploration was performed. After the removal of remnant thymic tissue in the mediastinal space, a significant drop in intraoperative parathyroid hormone levels was achieved. The pathological result confirmed the presence of a tiny pathological parathyroid adenoma within the thymus. At 6 months follow-up, postoperative biochemical assessment was consistent with normal calcium and parathyroid hormone levels.
Assuntos
Adenoma , Hiperparatireoidismo Primário , Neoplasias das Paratireoides , Humanos , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/diagnóstico por imagem , Feminino , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/etiologia , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico , Hiperparatireoidismo Primário/diagnóstico por imagem , Adenoma/complicações , Adenoma/cirurgia , Adenoma/diagnóstico por imagem , Neoplasias do Mediastino/complicações , Neoplasias do Mediastino/diagnóstico por imagem , Neoplasias do Mediastino/cirurgia , Hormônio Paratireóideo/sangue , Pessoa de Meia-Idade , Glândulas Paratireoides/diagnóstico por imagem , ParatireoidectomiaRESUMO
Primary hyperparathyroidism (PHPT) is characterized by elevated levels of calcium and parathyroid hormone (PTH). However, the interpretation of diagnostic tests, such as serum calcium and PTH levels, is complex in pregnant women. The aim of this report is to present a case of PHTP in a pregnant adolescent, with a special emphasis on an uncommon complication, as well as diagnostic and treatment strategies. A 17-year-old pregnant female presented with hyperemesis gravidarum and neurological symptoms, leading to the diagnosis of cerebral venous thrombosis. Further investigations revealed hypercalcemia and persistently elevated PTH levels, consistent with PHPT. After localization studies, the patient underwent an emergency parathyroidectomy with a diagnosis of parathyroid adenoma. During follow-up, intrauterine growth restriction and severe preeclampsia developed, necessitating an emergency cesarean section. Both the mother and neonate had favorable outcomes. PHPT is an infrequent condition in the pregnant population, and its diagnosis can be challenging due to the overlap of symptoms with normal physiological changes during pregnancy. The occurrence of uncommon complications, such as thrombotic phenomena, highlights the need for a comprehensive approach to ensure early detection and management. In most cases, parathyroidectomy is the treatment of choice.
El hiperparatiroidismo primario (HPTP) se caracteriza por niveles elevados de calcio y hormona paratiroidea (PTH). Sin embargo, la interpretación de pruebas diagnósticas, como los niveles de calcio sérico y PTH, es compleja en mujeres embarazadas. El objetivo de este reporte es presentar un caso de HPTP en una adolescente embarazada, con especial hincapié en una complicación infrecuente, así como en las estrategias diagnósticas y de tratamiento. Una mujer embarazada de 17 años presentó hiperémesis gravídica y síntomas neurológicos, lo que llevó al diagnóstico de trombosis venosa cerebral. Posteriores investigaciones revelaron hipercalcemia y niveles persistentemente elevados de PTH, consistentes con HPTP. Tras la realización de estudios de localización, la paciente fue sometida a una paratiroidectomía de emergencia con diagnóstico de adenoma de paratiroides. Durante el seguimiento, se desarrolló restricción del crecimiento intrauterino y preeclampsia grave, lo que resultó en la necesidad de realizar una cesárea de emergencia. Tanto la madre como el neonato evolucionaron favorablemente. El HPTP es una condición infrecuente en la población embarazada y su diagnóstico puede ser desafiante por la superposición de síntomas con los cambios fisiológicos normales del embarazo. La aparición de complicaciones infrecuentes, como fenómenos trombóticos, resalta la necesidad de un abordaje integral para garantizar la detección y el manejo temprano. En la mayoría de los casos, la paratiroidectomía es el tratamiento de elección.
Assuntos
Hiperparatireoidismo Primário , Neoplasias das Paratireoides , Paratireoidectomia , Humanos , Feminino , Gravidez , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/sangue , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/diagnóstico , Adolescente , Adenoma/complicações , Adenoma/cirurgia , Adenoma/diagnóstico , Hormônio Paratireóideo/sangue , Complicações Neoplásicas na Gravidez/cirurgia , Complicações Neoplásicas na Gravidez/diagnóstico , Complicações na Gravidez/diagnóstico , Hiperêmese Gravídica/complicações , Hiperêmese Gravídica/diagnóstico , Hipercalcemia/etiologia , Hipercalcemia/sangue , Hipercalcemia/diagnóstico , CesáreaRESUMO
OBJECTIVE: Primary hyperparathyroidism (PHPT) and celiac disease (CD) are two distinct medical conditions that can affect bone health. While PHPT leads to excessive calcium levels and bone abnormalities, CD impairs calcium and vitamin D absorption due to small intestine damage. CASE REPORT: We present a case of a 49-year-old woman diagnosed with osteoporosis who was found to have both PHPT and CD. The patient underwent a successful minimally invasive parathyroidectomy, which resulted in decreased parathyroid hormone levels. CONCLUSION: This case highlights the rare coexistence of PHPT and CD and emphasizes the importance of considering secondary causes of osteoporosis in patients with low bone mass. Further studies are needed to explore the underlying mechanisms and potential links between PHPT and CD.
Assuntos
Doença Celíaca , Hiperparatireoidismo Primário , Osteoporose , Feminino , Humanos , Pessoa de Meia-Idade , Doença Celíaca/complicações , Cálcio , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/cirurgia , ParatireoidectomiaRESUMO
Hyperparathyroidism-induced hypercalcemic crisis (HIHC) is an unusual state of marked progressive primary hyperparathyroidism (PHPT). Patients have severe hypercalcemia and may have severe symptoms such as kidney failure, acute pancreatitis, and mental changes. PHPT is due to the presence of a single gland adenoma/ disease in 80 to 85%; parathyroid carcinoma is reported in <1%. Among patients with adenoma, atypical parathyroid tumor can be found infrequently. Parathyroidectomy is the only curative approach for PHPT. In this report we present three cases of HIHC due to giant parathyroid adenomas (GPAs), one of them with histopathological characteristics of an atypical parathyroid tumor, with satisfactory evolution after parathyroidectomy.
La crisis hipercalcémica inducida por hiperparatiroidismo (HIHC) es un estado inusual de hiperparatiroidismo primario progresivo y marcado (HPTP). Los pacientes tienen hipercalcemia grave y pueden tener síntomas graves como insuficiencia renal, pancreatitis aguda y cambios mentales. El HPTP se debe a la presencia de un adenoma/enfermedad de una sola glándula en 80 a 85%; el carcinoma de paratiroides se informa en <1%. Entre los pacientes con adenoma, el tumor paratiroideo atípico se puede encontrar con baja frecuencia. La paratiroidectomía es el único abordaje curativo del HPTP. En este reporte presentamos tres casos de HIHC por adenomas paratiroideos gigantes (APGs), uno de ellos con características histopatológicas de tumor paratiroideo atípico, con evolución satisfactoria luego de paratiroidectomía.
Assuntos
Adenoma , Hipercalcemia , Hiperparatireoidismo Primário , Pancreatite , Neoplasias das Paratireoides , Humanos , Hipercalcemia/etiologia , Hipercalcemia/diagnóstico , Neoplasias das Paratireoides/complicações , Neoplasias das Paratireoides/cirurgia , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico , Doença Aguda , Adenoma/complicações , Adenoma/cirurgia , Adenoma/patologiaRESUMO
Introducción: describir el caso de un paciente con pancreatitis aguda secundaria a hipercalcemia por hiperparatiroidismo prImario. Esta es una causa poco frecuente de pancreatitis, asociada a morbimortalidad significativa en caso de no ser diagnosticada oportunamente Caso clínico: un hombre de 44 años, con antecedente de pancreatitis de presunto origen biliar que había requerido previamente colecistectomía, consultó por dolor abdominal y náuseas. Los estudios complementarios fueron compatibles con un nuevo episodio de pancreatitis aguda. Presentaba hipercalcemia y hormona paratiroidea (PTH) elevada, configurando hiperparatiroidismo primario. La gammagrafía informó hallazgos compatibles con adenoma paratiroideo. Se inició tratamiento con reanimación hídrica y analgesia con adecuada disminución de calcio sérico y resolución de dolor abdominal. Después de la paratiroidectomía se logró normalizar los niveles de calcio y PTH. Discusión: la pancreatitis aguda es una condición potencialmente fatal, por lo que la sospecha de causas poco frecuentes como la hipercalcemia debe tenerse en cuenta. El tratamiento de la hipercalcemia por adenoma paratiroideo se basa en reanimación hídrica adecuada y manejo quirúrgico del adenoma, con el fin de evitar recurrencia de pancreatitis y mortalidad. (AU)
Introduction: we describe the case of a patient with acute pancreatitis secondary to hypercalcemia due to primary hyperparathyroidism. This is a rare cause of pancreatitis associated with significant morbidity and mortality if not diagnosed in time. Clinical case: a 44-year-old man with a history of pancreatitis of presumed biliary origin, which had previously required cholecystectomy, consulted for abdominal pain and nausea. The laboratory findings were compatible with a new episode of acute pancreatitis. He presented hypercalcemia and an elevated parathyroid hormone (PTH), configuring primary hyperparathyroidism. Scintigraphy was performed, yielding findings compatible with parathyroid adenoma. Treatment with fluid resuscitation and analgesia was started, resulting in an adequate decrease in serum calcium and resolution of abdominal pain. After parathyroidectomy, calcium and PTH levels were normalized. Discussion: acute pancreatitis is a potentially fatal condition; therefore the suspicion of rare causes, such as hypercalcemia, should be considered. The treatment of hypercalcemia due to parathyroid adenoma is based on adequate fluid resuscitation and surgical management of the adenoma, to avoid recurrence of pancreatitis and death. (AU)
Assuntos
Humanos , Masculino , Adulto , Pancreatite/etiologia , Neoplasias das Paratireoides/diagnóstico por imagem , Hiperparatireoidismo Primário/diagnóstico por imagem , Hipercalcemia/etiologia , Pancreatite/prevenção & controle , Neoplasias das Paratireoides/cirurgia , Neoplasias das Paratireoides/complicações , Cintilografia , Tecnécio Tc 99m Sestamibi , Hiperparatireoidismo Primário/complicações , Hipercalcemia/sangue , Hipercalcemia/terapiaRESUMO
Brown tumors represent the terminal stage of bone remodeling processes in primary hyperparathyroidism. Currently they are rare, and typically affect long bones, pelvis and ribs. Brown tumors may be not included in the initial differential diagnosis of bone disease, especially when they are present in atypical localizations. We reported two cases of oral brown tumors as the initial presentation of primary hyperparathyroidism. In the first case, a 44-year-old woman presented a painful and sessile lesion of 4 × 3 cm over the central body of the mandible which progressively increased in 4-month. The second case involved a 23-year-old woman who was referred with a 3-month history of a painful and ulcerated mass of 2 cm arising from left maxilla, episodes of gingival hemorrhage and difficulty of breathing. Both cases were solitary tumors with no evidence of palpable cervical lymphadenopathy. Incisional biopsy of oral tumors resulted in giant cell and primary hyperparathyroidism was confirmed by laboratory tests. After parathyroidectomy, histology confirmed adenoma in both cases. Although this type of clinical presentation has almost disappeared in the recent decades, brown tumors should be considered in the differential diagnosis of bone oral masses.
Los tumores pardos son raros y, por lo general, afectan huesos largos, pelvis y costillas. Pueden no estar incluidos en el diagnóstico diferencial inicial como manifestación de enfermedad esquelética, especialmente cuando se presentan en localizaciones atípicas. Comunicamos dos casos de tumores pardos orales como presentación inicial de hiperparatiroidismo primario. En el primer caso, una mujer de 44 años presentó una lesión dolorosa y sésil de 4 × 3 cm sobre el cuerpo central de la mandíbula que aumentó de tamaño progresivamente en 4 meses. El segundo caso corresponde a una mujer de 23 años que acudió por presentar una masa dolorosa y ulcerada de 2 cm en maxilar izquierdo de 3 meses de evolución, episodios de hemorragia gingival y dificultad para respirar. Todos fueron tumores solitarios sin evidencia de linfadenopatía cervical palpable. La biopsia incisional de los tumores orales resultó en células gigantes, y las pruebas de laboratorio confirmaron el hiperparatiroidismo primario. Tras la paratiroidectomía, la histología confirmó adenoma en ambos casos. Los tumores pardos representan la etapa terminal de los procesos de remodelación ósea en el hiperparatiroidismo primario. Aunque este tipo de presentación clínica casi ha desaparecido en las últimas décadas, los tumores pardos deben ser considerados en el diagnóstico diferencial de las masas óseas orales.
Assuntos
Adenoma , Hiperparatireoidismo Primário , Neoplasias Maxilomandibulares , Osteíte Fibrosa Cística , Feminino , Humanos , Adulto , Adulto Jovem , Hiperparatireoidismo Primário/complicações , Hiperparatireoidismo Primário/diagnóstico , Osteíte Fibrosa Cística/diagnóstico por imagem , Osteíte Fibrosa Cística/etiologia , Diagnóstico Diferencial , Adenoma/diagnóstico , Neoplasias Maxilomandibulares/diagnósticoRESUMO
Primary hyperparathyroidism (PHPT) is an endocrine disorder characterized by hypercalcaemia and elevated or inappropriately normal concentrations of parathyroid hormone. Remission of PHPT caused by infarction or hemorrhage of a parathyroid adenoma rarely occurs, either spontaneously or induced, not always leading to a definitive cure. We report a case of 72-year-old women with primary hyperparathyroidism who underwent fine-needle aspiration cytology (FNAC) of a parathyroid adenoma mistaken for a thyroid nodule followed by normalization of parathyroid hormone (PTH) and serum calcium levels. Parathyroid origin was confirmed by immunohistochemistry. PTH levels began to rise at 4 months after FNAC demonstrating recurrence of the PHPT. This report shows that FNAC induced hemorrhage may cause remission of PHPT. Nevertheless, patient´s levels of PTH and serum calcium should be monitored, as remission may only be transitory.
Assuntos
Hiperparatireoidismo Primário , Neoplasias das Paratireoides , Nódulo da Glândula Tireoide , Humanos , Feminino , Idoso , Nódulo da Glândula Tireoide/complicações , Biópsia por Agulha Fina , Neoplasias das Paratireoides/complicações , Hiperparatireoidismo Primário/complicações , Cálcio , Hormônio Paratireóideo , HemorragiaRESUMO
Objective: The study sought to determine the clinical features of hyperparathyroid-induced hypercalcemic crisis (HIHC) along with treatment options and outcomes. Subjects and methods: This is a retrospective analysis of our historical cohort of patients with primary hyperparathyroidism (PHPT). Patients were divided in groups according to their calcium levels and clinical presentation. HIHC (group 1) was assumed when patients had high calcium levels and needed emergency hospitalization. Group 2 was composed of patients with calcium levels above 16 mg/dL or patients who needed hospitalization for classical PHPT symptoms. Group 3 was composed of clinically stable patients with calcium levels between 14 and 16 mg/dL, who were electively treated. Results: Twenty-nine patients had calcium levels above 14 mg/dL. HIHC group had seven patients, and initial clinical measures had good response in two patients, moderate response in one patient, and poor response in four patients. All poor responders underwent immediate surgery, and one of them died due to HIHC complications. Group 2 had nine patients, and all were successfully treated during hospitalization. Group 3 had 13 patients, and all had a successful elective surgery. Conclusion: HIHC is a life-threatening condition that requires fast clinical intervention. Surgery is the only definitive treatment and should be planned for all patients. Poor response to initial clinical measures should direct treatment toward surgery to avoid disease progression and clinical deterioration.
Assuntos
Hipercalcemia , Hiperparatireoidismo Primário , Humanos , Cálcio , Estudos Retrospectivos , Paratireoidectomia , Hipercalcemia/etiologia , Progressão da Doença , Hiperparatireoidismo Primário/cirurgia , Hiperparatireoidismo Primário/complicaçõesRESUMO
Objective: This study aimed to investigate the association between 25OHD (total, bioavailable and free) with bone mass and microarchitecture among primary hyperparathyroidism (PHPT) patients and controls. Subjects and methods: Sixty-four patients in the preoperative period of PHPT and 63 matched controls, who had not taken vitamin D in the last three months. To calculate the bioavailable and free 25OHD, the genetic variants of the vitamin D-binding protein (DBP) were determined. Bone mineral density (BMD) was determined by dual-energy X-ray absorptiometry (DXA). The distributions of total, bioavailable and free 25OHD and their correlation with TBS and DXA were evaluated. Results: PHPT showed BMD and TBS values lower than CTRL in all locations (p < 0.05). There were no statistical differences in the levels of free, bioavailable and total 25OHD between the PHPT and CTRL groups [mean, min-max: 3.4 (1.4-8.6) vs. 3.1 (1.0- 9.8) pg/mL, 1.51 (0.43-3.58) vs. 1.41 (0.38-3.48) ng/mL, 22.6 (11.0-39.9) vs. 20.6 (8.9-35.3) ng/dL, respectively; (p > 0.05). The distribution of DBP haplotypes was similar between groups. DXA showed no correlation with any form of 25OHD in both groups. TBS presented a weak correlation with the total 25OHD in PHPT (r = 0.28; p = 0.02) and a moderate correlation with the total, free and bioavailable 25OHD in CTRL (r = 0.42; r = 0.42; r = 0.43; respectively, p < 0.01). Conclusion: The concentrations of total, free and bioavailable 25OHD were similar in both the PHPT and control groups. 25OHD concentrations correlated positively with TBS and not with DXA, especially in controls, suggesting that this method may be more sensitive to assessing the consequences of vitamin D deficiency on bone quality in individuals without PHPT.
Assuntos
Osso Esponjoso , Hiperparatireoidismo Primário , Humanos , Absorciometria de Fóton , Estudos Transversais , Densidade Óssea , Vitamina DRESUMO
BACKGROUND: Intraoperative parathyroid hormones have been used to establish operative success in patients with primary hyperparathyroidism. This study's aim was to assess the impact of estimated glomerular filtration rate and serum creatinine levels on the fulfillment of >50% drop and normalization of intraoperative parathyroid hormone levels. METHODS: Patients successfully treated for primary hyperparathyroidism were analyzed. The samples for parathyroid hormone were collected at baseline, 5-, 10-, and 30-minutes postexcision. The patients were classified as follows: (1) estimated glomerular filtration rate >60 mL/min, (2) estimated glomerular filtration rate <60 mL/min and serum creatinine levels <1.2 mg/dL, and (3) estimated glomerular filtration rate <60 mL/min and serum creatinine levels >1.2 mg/dL. Comparative analysis of patients achieving the >50% parathyroid hormone drop criterion and normalization of intraoperative parathyroid hormone was performed. RESULTS: One hundred-fourteen patients were distributed as follows: 88 patients (77.2%), 14 (12.3%), and 12 (10.5%) for groups 1, 2 and 3, respectively. No difference between groups in the proportion of patients fulfilling the >50% parathyroid hormone drop criterion was found. An abnormally elevated intraoperative parathyroid hormone level at 30-minute postexcision was observed in 0, 14.3, and 16.6% in groups 1, 2, and 3, respectively (P ≤ .0001). CONCLUSION: In the study, >50% parathyroid hormone drop criterion was equally achieved despite normal or reduced estimated glomerular filtration rate. When serum creatinine levels increased >1.2 mg/dL and estimated glomerular filtration rate declined <60 mL/min, the likelihood of reaching normal intraoperative parathyroid hormone levels postexcision was significantly lower.