Crisis hipercalcémica, presentación inusual de adenoma paratiroideo: reporte de 2 casos / Hypercalcemic crisis, unusual presentation of parathyroid adenoma

La crisis hipercalcémica (CH) es una emergencia endocrina inusual, definida por la presencia de calcemia > 14 mg/dl asociada a disfunción renal, alteraciones cardiovasculares, gastrointestinales y del sensorio; también podría considerarse en pacientes con síntomas graves y calcemia menor. El hiperparatiroidismo primario (HPTP) y las neoplasias malignas son las etiologías más comunes de la hipercalcemia (90% de los casos); sin embargo, rara vez el primero se presenta como CH. Debido a la alta mortalidad asociada a esta entidad, es de gran importancia establecer diagnóstico y tratamiento precoces. Presentamos dos pacientes con crisis hipercalcémica como primera manifestación del HPTP, el 1.° con bloqueo auriculoventricular (AV) completo y el 2.° con pancreatitis aguda. La anatomía patológica (AP) reveló adenoma oxifílico en ambos casos, que es una variante histológica poco frecuente y puede manifestarse clínicamente de forma grave. Conclusiones: los adenomas paratiroideos son causa poco frecuente de CH. Consideramos el tipo histológico observado (adenoma oxifílico) como probable factor condicionante. La pancreatitis y especialmente el bloqueo AV son manifestaciones poco frecuentes de la CH. Resaltamos la importancia de la determinación de los niveles de calcio dentro de la evaluación inicial de todo paciente con bloqueo AV. (AU)

Hypercalcemic crisis (HC) is an unusual endocrine emergency, defined as the presence of serum calcium > 14 mg/dl related to kidney dysfunction, cardiovascular, gastrointestinal and sensory disturbances. It could also be considered in patients with severe symptoms and lower serum calcium levels. Primary hyperparathyroidism (PHPT) and malignant neoplasms are the most common hypercalcemia etiologies (90% of cases), nevertheless, the former hardly ever occurs as HC. Due to the high mortality associated with HC, it is crucial to establish early diagnosis and treatment.We report two patients with HC as the first manifestation of PHPT; the former with atrioventricular (AV) block and the latter with acute pancreatitis. Pathology revealed oxyphilic adenoma in both cases, which is an infrequent histological variant that can have a severe clinical manifestation. Conclusions: parathyroid adenomas are a rare cause of HC. We consider the histological type observed (oxyphilic adenoma) as a probable conditioning factor. Pancreatitis and especially AV block are rare manifestations of HC. We emphasize the importance of determining calcium levels in the initial evaluation of all patients with AV block. (AU)

Manejo quirúrgico de carcinoma de paratiroides: a propósito de un caso / Surgical management of parathyroid carcinoma: a case report

Resumen Introducción: A pesar de que el carcinoma de paratiroides es uno de los cánceres menos frecuentes del mundo, es importante tenerlo en cuenta al plantear el diagnóstico diferencial del hiperparatiroidismo primario, ya que su diagnóstico temprano tiene repercusiones en el tratamiento y el pronóstico vital del paciente. Caso Clínico: A continuación, se expone un caso clínico de un paciente con sintomatología abigarrada de hiperfunción paratiroidea que, dada la sospecha clínica de carcinoma de paratiroides y la no infiltración de estructuras adyacentes, fue tratado con una paratiroidectomía. Conclusión: Esta cirugía supone una menor morbilidad, sin suponer un detrimento para la supervivencia global del paciente.

Introduction: Parathyroid carcinoma should be taken into consideration among the differential diagnosis of primary hyperparathyroidism, even though it is one of the less common malignant tumours in the world, because an early diagnosis is essential for the treatment and the prognosis of the patient. Case Report: We present the case of a patient whose symptoms were compatible with hyperfunction of parathyroid gland. Due to the malignant disease suspicion and the non-invasion of adjacent tissue, he was treated with a parathyroidectomy. Conclusión: This type of treatment supposes a lower morbidity without decrease in overall survival, according to bibliography.

Hiperparatiroidismo primario por carcinoma paratiroideo: caso problema y revisión / Primary hyperparathyroidism by parathyroid cancer: case report and review

El carcinoma paratiroideo (CP) es una neoplasia maligna con una incidencia de 0,015 cada 100.000 habitantes por año. Representa el 1% de los diagnósticos de hiperparatiroidismo primario y se manifiesta entre la 4.a y 5.a década de la vida, con una incidencia similar entre hombres y mujeres. La etiología del CP es incierta, ha sido asociada a formas esporádicas o familiares. Está caracterizado por altos niveles séricos de calcio y PTH y el desafío clínico-quirúrgico es el diagnóstico diferencial con otras entidades benignas como el adenoma o la hiperplasia de paratiroides. Aunque el diagnóstico de certeza es anatomopatológico, la sospecha clínica y el uso de métodos de baja complejidad (ecografía) con operadores avezados permite una correcta localización y abordaje pertinente del paciente para dirigir el tratamiento quirúrgico adecuado (resección en bloque) evitando persistencias y recurrencias de enfermedad. Se presenta el caso clínico de un paciente masculino que ingresa por síndrome de impregnación asociado a hipercalcemia, su abordaje diagnóstico, tratamiento y manejo interdisciplinario con discusión y revisión bibliográfica. (AU)

Parathyroid carcinoma (CP) is a malignant disease with an incidence of 0.015 per 100,000 inhabitants per year. It accounts for 1% of primary hyperparathyroidism diagnoses and occurs between the 4th and 5th decade of life, with a similar incidence between men and women. The etiology of CP is uncertain and has been associated with sporadic or family forms. CP is characterized by high serum calcium and PTH levels and the clinical-surgical challenge is the differential diagnosis with other benign entities such as parathyroid adenoma or hyperplasia. Although the diagnosis of certainty is achieved by pathological anatomy examination, the clinical suspicion and the use of low complexity methods (ultrasound) by experienced operators allows a correct localization and a patient-specific approach to direct the appropriate surgical treatment (block resection), avoiding persistence and recurrences of disease. The clinical case of a male patient admitted for severe hypercalcemia with multiple organ disfunction, the diagnostic approaches, treatment, and interdisciplinary management, together with review and discussion of the current literature are presented. (AU)

Parathyroid hemangioma

Background Hemangiomas are benign neoplasms of capillary proliferation that arise from a developmental anomaly where angioblastic mesenchyme fails to form canals. Most hemangiomas arise in the head and neck region, either superficially in the skin or deeper within endocrine organs such as the parotid gland. Parathyroid hemangiomas, however, are extremely rare, with only five cases previously reported in the literature. Case presentation Herein, we present a case of a 68-year-old man with a hemangioma almost completely replacing the right upper parathyroid gland, grossly measuring 1.3 × 1.3 × 1.2 cm and weighing 700 mg, associated with primary hyperparathyroidism. Conclusions Parathyroid gland enlargement due to vascular neoplasms such as hemangiomas can mimic, both clinically and radiographically, hyperplasias and/or adenomas. Surgeons need to be aware of the presence of this entity and should consider it in the differential diagnosis of hyperparathyroidism or parathyroid gland enlargement.

Análisis de falsos negativos en la cintigrafía SPECT de paratiroides con sestamibi en pacientes con hiperparatiroidismo primario sometidos a cirugía entre 2008-2015 en hospital universitario / False negative spect parathyroid scintigraphy with sestamibi in patients with primary hyperparathyroidism

Background: 99mTc-sestamibi parathyroid SPECT scintigraphy is a useful tool in the pre-operative study of hyperparathyroidism. False negatives (FN) have been reported in 5.7-14% of the examinations. Aim: To characterize 99mTc-sestamibi FN in cases referred for primary hyperparathyroidism (PHP) to a university hospital. Material and Methods: Descriptive retrospective analysis. We included patients with PHP, studied with SPECT scintigraphy, operated at our center between 2008 and 2015. Clinical and surgical data were recorded; biopsies of the FN were blindly reviewed by one pathologist. Results: One hundred twenty one scintigraphies fulfilled the inclusion criteria. Seven (5.8%) were negative and 114 positive. There was no difference in age, sex and PTH levels between FN and true positive scintigraphies. At surgery, one FN case had two hyperplasic glands and two cases had ectopic glands. Pathology reported adenoma in three cases, hyperplasia in three and carcinoma in one. The largest diameter of the lesion was lower in FN (1.3 and 2.1 cm respectively, p = 0.02) and the proportion of adenomas was higher in true positive cases (29% and 75% respectively; p < 0.01). The interval between scintigraphy and parathyroidectomy was greater in FN with a median of 92 days (range 20 days-3.2 years, p < 0.01). The percentage of oxyphilic cells observed was similar in both groups. Conclusions: FN parathyroid SPECT scintigraphies in PHP are uncommon. They corresponded to lesions under the equipment's resolution limit and resulted in longer time lags between scintigraphy and surgery.

Paratirotoxicosis y tumor cervical palpable: caso clínico de cáncer de paratiroides / Parathyrotoxicosis and palpable neck mass: a clinical presentation of parathyroid cancer

Introducción: El cáncer de paratiroides es poco frecuente. Suele presentarse como hiperparatiroidismo primario, en ocasiones como crisis hipercalcémica, con malestar general, náuseas, vómitos, alteraciones del ánimo y pérdida de peso. En algunos casos se presenta como osteítis fibrosa quística, osteopenia difusa, osteoporosis y fracturas patológicas. El diagnóstico suele estar dado por biopsia quirúrgica que muestra invasión a estructuras adyacentes, o metástasis locales y distantes. El tratamiento es la resección quirúrgica del tumor, sin uso de terapias adyuvantes. Su tasa de recurrencia es alta, de 25 a 100%. Algunos factores de mal pronóstico son metástasis a linfonodos en la presentación inicial, metástasis distantes y carcinomas no funcionantes. Caso clínico: Paciente masculino de 64 años que consultó por compromiso del estado general, bradipsiquia, poliuria, cefalea y masa cervical. Además presentaba hipercalcemia y gran elevación de PTH. Se realizó resección quirúrgica de la masa cervical, con biopsia rápida con atipias y mitosis 1/10, y de un nódulo hiperplásico tiroideo. Tras esto, sus niveles de PTH disminuyeron, así como también descendieron sus niveles de calcio. Discusión: Los pacientes que presentan cáncer de paratiroides suelen tener valores más elevados de calcemia y PTH. Para su diagnóstico, no se recomienda usar biopsia por punción, por riesgo de diseminación y por el bajo rendimiento de esta técnica. Conclusión: Pese a ser poco frecuente, se debe pensar en cáncer de paratiroides en el contexto de un paciente con hipercalcemia, especialmente si presenta PTH muy elevada. La sospecha clínica de este diagnóstico tiene directa relación con la posibilidad de tratamiento y pronóstico de la enfermedad.

Introduction: Parathyroid cancer is rare. Usually presented as primary hyperparathyroidism, sometimes as hypercalcemic crisis, with general malaise, nausea, vomiting, mood disturbances and low weight. In some cases it occurs as osteitis fibrosa cystica, diffuse osteopenia, osteoporosis and pathological fractures. The diagnosis is usually made by surgical biopsy shows invasion of adjacent structures, or local and distant metastases. The treatment is surgical resection of the tumor, without the use of adjuvant therapies. Their recurrence rate is high, 25 to 100%. Poor prognostic factors are lymph node metastases at initial presentation, distant metastases and nonfunctional carcinomas. Case report: Male patient consulted for 64 years in general conditions, bradypsychia, polyuria, headache and neck mass. He also had hypercalcemia and high elevation of PTH. Surgical resection of the cervical mass was performed, with rapid biopsy atypia and mitosis 1/10, and hyperplastic thyroid nodule. After this, PTH decreased levels as well as levels of calcium. Discussion: Patients with parathyroid cancer tend to have higher serum calcium and PTH of values. For diagnosis, it is not recommended to use needle biopsy, risk of spread and the poor performance of this technique. Conclusion: Despite being rare, you should think parathyroid cancer in the context of a patient with hypercalcemia, especially if you have very high PTH. The clinical suspicion of this diagnosis is directly related to the possibility of treatment and prognosis of the disease.

Evaluation of preoperative ultrasonographic and biochemical features of patients with aggressive parathyroid disease: is there a reliable predictive marker?

ABSTRACT Objective Parathyroid cancer (PC) represents < 1% of cases of PHPT. Tumors demonstrating atypical histopathologic features and don’t fulfill criteria for carcinoma are classified as atypical adenomas (APA). The purpose of this study was to determine a biochemical or ultrasonographic feature that can predict aggressive disease requiring more extensive surgery and closer follow-up. Subjects and methods Twenty eight patients operated for PHPT and diagnosed with atypical adenoma (23 patients) or carcinoma (5 patients) were enrolled in this study. The control group consisted of 102 patients operated between the same dates and diagnosed with classical PA. Classical adenomas, atypical adenomas, and carcinomas were compared according to their biochemical and ultrasonographic parameters. Results Serum Ca levels were significantly higher in the PC group compared with the APA and classical PA groups. Serum median PTH, Serum ALP and UCa was significantly higher in the APA and carcinoma groups compared to the classical PA group. ROC analysis was made to determine the best cut off values for predicting aggressive disease were 12.45 mg/dL, 265.05 pg/mL, 154.5 IU/l, 348.5 mg/day and 21.5 mm for Ca, PTH, ALP, UCa and the adenoma diameter, respectively. Multivariate analysis showed that serum Ca, ALP and isoechoic/cystic appearance were independent predictors for aggressive disease. Conclusion Preoperatively high PTH, ALP, and UCa levels and large lesions with isoechoic or cystic appearances may be predictive of atypical adenoma or carcinoma in patients being evaluated for PHPT. In such cases, surgeons may prefer en bloc parathyroidectomy to minimally invasive surgery.

Association between atypical parathyroid adenoma and neurofibromatosis

Primary hyperparathyroidism is a disease characterized by excessive production of parathyroid hormone (PTH), which is due to a parathyroid adenoma in 85% of cases. An atypical parathyroid adenoma, with some histopathological features of parathyroid carcinoma, may be found in some of the cases, although it may not fulfill all the criteria for this diagnosis. Neurofibromatosis type 1 (NF1) is an autosomal dominant systemic disease that may be associated with hyperparathyroidism. We report here the rare combination of a patient with NF1 and clinical manifestations of hyperparathyroidism due to an atypical parathyroid adenoma.

A rare case of a parathyroid adenoma inside a parathyroid cyst / Um caso raro de adenoma de paratireoide em um cisto de paratireoide

Parathyroid cysts (PCs) are rare lesions, located in the neck and anterior mediastinal region. The vast majority are non-functioning, presented as nodular cervical lesions. Large, non-functioning PCs can manifest with compressive symptoms of the surrounding tissues. Rarely, PCs produce excessive amounts of parathyroid hormone (PTH), resulting in primary hyperparathyroidism. We report a case of functional PC, describing its diagnostic and therapeutic approach.

Os cistos de paratireoide (PCs) são lesões raras, localizadas no pescoço e na região do mediastino anterior. A grande maioria é não funcionante, apresentando-se como lesões cervicais nodulares. PCs não funcionantes de grandes dimensões podem se manifestar com sintomas compressivos. Raramente, PCs produzem quantidades excessivas de hormônio da paratireoide (PTH), o que resulta em hiperparatiroidismo primário. Relatamos um caso de PC funcional, descrevendo sua abordagem diagnóstica e terapêutica.

Normocalcemic primary hyperparathyroidism: long-term follow-up associated with multiple adenomas / Hiperparatireoidismo primário normocalcêmico associado a múltiplos adenomas

Normocalcemic primary hyperparathyroidism (NPHPT) is a condition characterized by elevation of the parathyroid hormone (PTH) in the presence of normal serum calcium and the absence of secondary causes. The case described illustrates the long-term follow-up of a postmenopausal woman with NPHPT patient who progressed with multiple adenomas. This case reports a 77-year-old female who has chronic generalized pain and osteoporosis. Her initial serum PTH was 105 pg/mL, with total serum calcium of 9.6 mg/dL, albumin 4.79 g/dL, phosphorus 2.8 mg/dL, and 25OHD after supplementation was 34.6 ng/mL. The bone densitometry (BMD) results were as follows: lumbar spine: T-score -3.0, femoral neck: T-score -2.6 and distal radius: -4.2. Other causes of secondary hyperparathyroidism were ruled out and cervical ultrasound and Tc-99-Sestamibi scan were negative. She used oral alendronate and three infusions of zoledronic acid for treatment of osteoporosis. In the 10th year of follow-up, after successive negative cervical imaging, ultrasound showed a nodule suggestive of an enlarged right inferior parathyroid gland. PTH levels in fluid which was obtained during fine-needle aspiration (FNA) were over 5,000 pg/mL and a Sestamibi scan was negative. The patient underwent parathyroidectomy, and a histological examination confirmed parathyroid adenoma. Post-operatively serum PTH remained elevated in the presence of normal serum calcium levels. A follow-up cervical ultrasound showed a new solid nodule suggestive of an enlarged right superior parathyroid gland. PTH levels in the aspiration fluid were remarkably high. A second parathyroidectomy was performed, with the excision of a histologically confirmed parathyroid adenoma. In conclusion, this is an unusual presentation of NPHPT and highlights the long-term complications.

Hiperparatiroidismo primário normocalcêmico (NPHPT) caracteriza-se pela elevação do hormônio da paratiroide (PTH), na ausência da elevação dos níveis séricos de cálcio e exclusão de causas secundárias. O caso descrito ilustra o seguimento de uma mulher na pós-menopausa com NPHPT que evoluiu com múltiplos adenomas. Este caso relata uma paciente de 77 anos de idade que tem dor generalizada crônica e osteoporose. O PTH inicial foi elevado com níveis séricos de cálcio, albumina, fósforo e 25OH vitamina D normais. A densitometria óssea (DMO) evidenciou um T-SCORE da coluna lombar: -3.0, colo do fêmur: -2.6 e rádio distal: -4.2. Outras causas de hiperparatireoidismo secundário foram descartadas e a ultrassonografia cervical e varredura com Sestamibi foram negativos. Fez uso de alendronato e três infusões de ácido zoledrônico para o tratamento da osteoporose. No décimo ano de seguimento, depois de sucessivas imagens negativas, a ultrassonografia evidenciou um nódulo sugestivo de adenoma de paratireoide inferior direita. A paciente foi submetida à paratireoidectomia, e um exame histológico confirmou a hipótese. A elevação dos níveis séricos de PTH no pós-operatório se manteve com níveis normais de cálcio. A nova ultrassonografia cervical evidenciou outro nódulo sugestivo de adenoma de paratireoide superior direita. Uma segunda paratireoidectomia foi realizada, cujo histológico confirmou outro adenoma de paratireoide. Conclui-se que essa é uma apresentação incomum de NPHPT e destaca as complicações a longo prazo.

Nonadenomatous nonencapsulated thymic parathyroid tissue concomitant with primary hyperparathyroidism due to ectopic parathyroid adenoma / Tecido tímico paratireoidiano não edematoso e não encapsulado concomitante com hiperparatireoidismo devido a adenoma paratireoidiano ectópico

Primary hyperparathyroidism due to ectopic parathyroid adenoma is not infrequent. Primary hyperparathyroidism caused by unusual thymic nonadenomatous nonencapsulated parathyroid tissue has been reported before. Both can cause unsuccessful neck explorations. Here we presented for the first time a patient with hyperparathyroidism due to ectopic parathyroid adenoma concomitant to the presence of thymic nonadenomatous nonencapsulated parathyroid tissue.

O hiperparatireodismo primário devido a adenoma ectópico paratireoidiano não é raro. O hiperparatireodismo primário causado por tecido tímico paratireoidiano não edematoso e não encapsulado incomum já foi relatado anteriormente. Ambos podem levar à exploração cervical malsucedida. Apresentamos aqui, pela primeira vez, uma paciente com hiperparatireoidismo decorrente de um adenoma paratireoidiano concomitante com a presença de tecido tímico paratireoidiano não edematoso e não encapsulado.

Detección inmunohistoquímica de parafibromina en patología de paratiroides / Immunohistochemical detection of parafibromin in parathyroid pathology

Introduction: The definitive diagnosis of parathyroid cancer is extremely difficult, from the clinical approach to the molecular diagnosis. A gene mutation was detected recently in patients with parathyroid cancer. It is a suppressor tumor gene called HRPT2, which codifies for a protein that participates in PAF1 complex, the parafibromin. It has been observed that the expression of this protein it's altered in parathyroid cancer, what would serve like method of diagnosis by immunohystochemistry, with a sensitivity and specificity of 73-96 percent and 99-100 percent respectively. Material and Method: The anti-parafibromin immunohysto-chemistry staining was made in 23 parathyroids tissue samples (5 adenomas, 6 hyperplasia, 7 normal and 5 carcinomas). Results: A positive pattern is observed in almost 100 percent of benign pathology and 100 percent in normal tissue. In the cases of carcinoma only 2 of 5 had a strong positivity. Conclusions: The pathological clinical correlation does not allow the association of the loss of parafibromin immunoreactivity in some unequivocal cases of parathyroid cancer. The parafibromin immunostaining does not allow to discriminate between benign or malign pathologies.

Introducción: El diagnóstico definitivo de cáncer de paratiroides es extremadamente difícil, desde el acercamiento clínico hasta el diagnóstico molecular. Se detectó recientemente en pacientes con cáncer de paratirodes un gen supresor de tumor mutado (HRPT2), que codifica para una proteína que participa en el complejo PAF1, la parafibromina. Se ha observado que la expresión de esta proteína está alterada en los casos de cáncer de paratiroides, lo que serviría como método de diagnóstico por inmunohistoquímica, con una sensibilidad y especificidad de 73-96 por ciento y 99-100 por ciento, respectivamente. Material y Método: Se realizó tinción inmunohistoquímica anti parafibromina en 23 muestras de tejido paratiroideo (5 adenomas, 6 hiper-plasias, 7 normales y 5 carcinomas). Resultados: Se observa un patrón positivo fuerte en casi 100 por ciento de la patología benigna y 100 por ciento en tejido normal. En los casos de carcinoma sólo 2 de 5 tenían positividad fuerte. Conclusiones: La correlación clínico patológica no permite asociar la pérdida de tinción de parafibromina en algunos casos de cáncer inequívocos. La tinción de parafibromina no permite discriminar entre patología benigna y maligna.

Parathyroid carcinoma and hungry bone syndrome / Carcinoma de paratiroide e fome óssea

We hereby report two patients with parathyroid carcinoma presenting extremely high calcium and PTH levels, severe bone disease, and palpable neck mass at diagnosis. They both underwent parathyroidectomy, and one of them evolved to lung metastasis. Important hypocalcemia was observed after surgery in both: after parathyroidectomy in one patient, and only after surgical removal of the metastasis in the other. Both required intravenous calcium replacement, thus revealing hungry bone syndrome (HBS). HBS usually reflects rapid mineralization after correction of hyperparathyroidism. The more severe the bone disease before surgery, the more prone the patient is to HBS after surgery. Despite being an unfavorable outcome, HBS state suggests that surgical removal of hypersecretory parathyroid tissue was accomplished. In this study, HBS was observed in both patients, who presented severe bone disease prior to surgery. HBS would be expected post-operatively in successful parathyroid carcinoma removal.

O presente artigo descreve o relato de dois pacientes com carcinoma de paratiroide que apresentavam valores intensamente elevados de cálcio sérico e de PTH, associado a doença óssea e presença de nódulo cervical palpável ao diagnóstico. Ambos foram submetidos à paratiroidectomia, sendo que um evoluiu com metástases pulmonares. Hipocalcemia importante foi observada após a paratiroidectomia em um paciente e somente após remoção cirúrgica das metástases pulmonares em outro. Ambos necessitaram de reposição endovenosa de cálcio, revelando, assim, o estado de fome óssea (FO). A presença da FO usualmente reflete rápida mineralização óssea após correção do hiperparatiroidismo; assim, quanto mais severa a doença óssea previa à cirurgia, maior será a FO observada no pós-operatório desses pacientes. Embora inicialmente considerada um evento indesejável, a FO representa a bem-sucedida remoção cirúrgica do tecido paratiroideano hipersecretor. Fome óssea deve ser esperada no pós-operatório do tratamento cirúrgico bem-sucedido do carcinoma de paratiroide.

Adenoma weight and biochemical parameters in primary hyperparathyroidism

Primary hyperparathyroidism is autonomous production of parathyroid hormone. After removal of adenoma, one of the surgeons concern is postoperative hypocalcaemia. There is no precise method to determine if patients have hypocalcaemia postoperatively. The purpose of this study was to determine the relation between parathyroid adenoma weights, postoperative serum calcium and serum biochemical parameters in patients with primary hyperparathyroidism. In a prospective study, eighty patients with single parathyroid adenoma were enrolled. Preoperative serum levels of calcium, phosphate, PTH, as well as Postoperative serum calcium and weight of adenomas were recorded. The level of significance was set to be p < 0.05. There was no significant correlation between postoperative serum calcium, parathyroid adenoma weight [r= -0.17, p= 0.1], and parathyroid hormone level [r = -0.11, p = 0.3]. However, a weak correlation between postoperative and preoperative serum calcium levels [r = 0.23, p = 0.03] was observed. Moreover, Serum calcium decline after adenoma resection was statistically correlated with adenoma weight [r = 0.36, p= 0.001], preoperative serum calcium [r = 0.92, p- 0.0007], PTH [r- 0.54, p= 0.0005] and ALP levels [r = 0.3, p= 0.006]. Although preoperative serum markers and adenoma weight are unreliable in predicting postoperative serum calcium level, it is possible to estimate postoperative calcium decline by considering adenoma weight and preoperative serum biochemical parameters

Quiste de paratiroides asociado a carcinoma papilar de tiroides: una rara entidad / Parathiroid cyst associated to papilary thyroid carcinoma: one rare entity

Los quistes de paratiroides son lesiones raras que aparecen en el cuello y en el mediastino anterior. Menos de 300 casos han sido publicados en la literatura. Los quistes de paratiroides constituyen solo el 0,6% de todas las lesiones de tiroides y paratiroides, pero deben ser considerados como diagnóstico diferencial en pacientes con masas cervicales anteriores, particularmente asociados a hipercalcemia. No se encontraron casos reportados en la literatura médica de quistes de paratiroides asociados a carcinoma de la glandula tiroides.

Parathyroid cysts are rare lesions arising in the neck and anterior mediastinum. Fewer than 300 cases have been published in the literature. Parathyroid cysts constitute only 0.6% of all thyroid and parathyroid lesions, but should be considered in the differential diagnósis of anterior neck masses, particulary in the presence of hypercalcemia. Were not cases reported in the medical literature of parathyroid cysts associated to carcinoma of thyroid gland.

Hiperparatiroidismo primario y cáncer de paratiroides: caso clínico / Primary hyperparathyroidism and parathyroid cancer: report of one case

We report a 51 years old female operated for a primary hyperparathyroidism that relapsed in two occasions. After the first and second operation, the pathological study of the excised glands disclosed an adenoma. After the second relapse, a parathyroid gland in the right lateral and upper cervical region, located with technetium (Tc)-99m Sestamibi scintigraphy, was excised. The pathological study of the surgical piece disclosed a parathyroid carcinoma without local lymph node involvement. A postoperative 18F-fluorodexyglucose positron emission tomography did not show tumor dissemination.

Presentamos un caso de sexo femenino, operada por hiperparatiroidismo primario, con 2 recidivas posteriores. En las 2 primeras oportunidades, el diagnóstico histológico fue compatible con adenoma. La última intervención se debió realizar por una glándula ubicada, de acuerdo al Sestamibi Tc-99m, en la región cervical lateral alta derecha. Se realizó exploración supraomohiodea, efectuando la extirpación de la glándula y adenopatías peritumorales. La biopsia definitiva informó carcinoma de paratiroides y ganglios linfáticos negativos para neoplasia. Se realizó estudio de diseminación y PET- FDG de control en el postoperatorio alejado, sin nuevos hallazgos. Entre la primera y la segunda intervención, se realizó una tiroidectomía total por un bocio uninodular, cuya biopsia informó 3 paratiroides normotípicas, intratiroideas. En suma, se lograron identificar histológicamente 6 glándulas paratiroides.

Importância da complementação com SPECT e 99mTc na cintilografia das paratiroides e da correlação clínica, laboratorial, ultrassonográfica e citológica na localização pré-operatória do adenoma de paratiroide - ensaio pictórico / Importance of parathyroid SPECT and 99mTc scintigraphy, and of clinical, laboratorial, ultrasonographic and citologic correlation in the pre-operative localization of the parathyroid adenoma - pictorial assay

OBJETIVO: Diante de um paciente portador de hiperparatiroidismo primário com indicação de tratamento cirúrgico, a localização pré-operatória da paratiroide é de fundamental importância para definir a melhor abordagem cirúrgica. MATERIAIS E MÉTODOS: A realização adicional de imagens SPECT e da cintilografia com 99mTc durante a cintilografia das paratiroides com Setamibi não é rotina em nosso meio, sendo comum a aquisição apenas das imagens planas − precoce (15 minutos) e tardia (2 horas). RESULTADOS: Na nossa experiência, tem-se percebido que a realização do protocolo completo contribui de maneira decisiva na sensibilidade da localização pré-operatória da paratiroide. CONCLUSÃO: A aplicação completa de todos os métodos cintilográficos disponíveis (SPECT e 99mTc) e a análise cuidadosa das imagens em um contexto multidisciplinar podem aumentar a acurácia da cintilografia das paratiroides.

OBJECTIVE: In patients with primary hyperparathyroidism, candidates for surgical intervention, the parathyroid pre-operative localization is of fundamental importance in planning the appropriate surgical approach. MATERIALS AND METHODS: The additional acquisition of SPECT and Technetium-99m images, during parathyroid scintigraphy with Sestamibi, is not common practice. Usually, only planar image acquisition, 15 minutes prior and 2 hours after radiopharmaceutical administration, is performed. RESULTS: In our experience, the complete protocol in parathyroid scintigraphy increases the accuracy of pre-operative parathyroid localization. CONCLUSION: The complete utilization of all available nuclear medicine methods (SPECT e 99mTc) and image interpretation in a multidisciplinary context can improve the accuracy of parathyroid scintigraphy.

SPECT/CT preoperatorio, una nueva herramienta diagnóstica para localizar adenomas paratiroideos: presentación de 2 casos clínicos / Localization of functioning parathyroid adenomas by SPECT/CT: report of two cases

Single photon emission computed tomography and computed tomography (SPECT/CT), integrates a gamma camera and a CT scan and is effective for the location of parathyroid adenomas. We report a 55 years old male and a 80 years old female with primary hyperparathyroidism. In both cases the 99mTc-Sestamibi parathyroid scintigraphy detected a functioning nodule whose presence was confirmed with SPECT/CT.

El SPECT/CT integra una gamacámara y un escáner radiológico en un solo equipo híbrido que fusiona la imagen de la cintigrafía SPECT (tomografía computada de fotón único) con la imagen morfológica obtenida con un escáner de baja intensidad, sin movilizar al paciente, en una perfecta correspondencia anátomo-funcional, permitiendo identificar con exactitud la localización de un adenoma paratiroideo. Se describen dos pacientes con hiperparatiroidismo primario, con ecotomografía convencional negativa, donde el SPECT/CT demostró la localización exacta de adenomas de ubicación no habitual.

Tumor pardo como manifestación inicial de hiperparatiroidismo primario / Brown tumor as the initial manifestation of primary hyperparathyroidism

El tumor pardo es una forma localizada de osteítis fibrosa quística, parte del compromiso óseo por hiperparatiroidismo. Como primera expresión de hiperparatiroidismo es infrecuente, debido a que actualmente éste se diagnostica en estadios asintomáticos o mínimamente sintomáticos. Presentamos el caso de una paciente con un tumor pardo localizado en el maxilar superior izquierdo, como primera manifestación de hiperparatiroidismo primario causado por un adenoma paratiroideo. Posterior a la realización de una paratiroidectomía el tumor evolucionó con franca regresión, sin necesidad de ningún otro procedimiento quirúrgico local.

Brown tumor is a localized form of osteitis fibrosa cystica, being part of the hyperparathyroid bone disease. It rarely is the first manifestation of hyperparathyroidism, since nowadays, the diagnosis is made at an asymptomatic or minimally symptomatic stage. We present a case of a left superior maxillar brown tumor as the first manifestation of primary hyperparathyroidism due to a parathyroid adenoma. A parathyroidectomy was performed, and there was a regression of the bone lesion, without the need of performing other local surgical procedures.