Pancreatitis aguda en el escenario de hipercalcemia por hiperparatiroidismo primario / Acute pancreatitis in the setting of hypercalcemia due to primary hyperparathyroidism

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)

Hiperparatiroidismo primario por adenoma con gammagrafía negativa / Primary hyperparathyroidism due to negative scintigraphy adenoma

Introducción: El hiperparatiroidismo es una de las causas más frecuente de hipercalcemia. El 85 por ciento de los pacientes son mayores de treinta años, su frecuencia más alta se encuentra por encima de 60 años, a largo plazo puede producir una serie de complicaciones. Objetivo: Describir el caso clínico de hiperparatiroidismo primario que resultó negativa al estudio de gammagrafía con localización ectópica del adenoma. Caso Clínico: Paciente femenina de 20 años con antecedentes de litiasis renal y uretral de 7 años de evolución, la cual se sometió a 13 sesiones de litotricia y dos cirugías renales con recidivas, hace 6 meses se detectó cifras elevadas de calcio. Se realiza protocolo de estudio de hipercalcemia determinándose hiperparatiroidismo primario, ultrasonido y tomografía de cuello reportan lesión que podría corresponder a un adenoma paratiroideo paratraqueal; sin embargo, la gammagrafía con metoxi-isobutilisonitrilo y tecnecio 99 fue negativa. Se realizó cirugía de cuello convencional ya que la cirugía radioguiada no fue útil para la localización del adenoma paratiroideo intraoperatorio. Estudio histopatológico reportó adenoma paratiroideo. Conclusiones: En adenomas paratiroideo con gammagrafía negativa se puede hacer el diagnostico con estudios de imagen convencional. La localización ectópica del adenoma paratiroideo constituye una causa de gammagrafía negativa. La cirugía radioguiada no es útil en los adenomas paratiroides ectópicos con gammagrafía negativa(AU)

Introduction: Hyperparathyroidism is one of the most frequent causes of hypercalcemia. 85 percent of patients are older than thirty years, its highest frequency is over 60 years, in the long term it can produce a series of complications. Objective: To describe a clinical case of primary hyperparathyroidism that was negative in the scintigraphy study with ectopic location of the adenoma. Clinical case report: A 20-year-old female patient with a 7-year history of renal and urethral lithiasis, who had undergone 13 lithotripsy sessions and two recurrent kidney surgeries. Six months ago, elevated calcium levels were detected. A study protocol for hypercalcemia was carried out, determining primary hyperparathyroidism. Ultrasound and neck tomography report a lesion that could correspond to a paratracheal parathyroid adenoma; however, methoxy-isobutylisonitrile and technetium-99 scintigraphy was negative. Conventional neck surgery was performed since radioguided surgery was not useful for locating the intraoperative parathyroid adenoma. Histopathological study reported parathyroid adenoma. Conclusions: In parathyroid adenomas with negative scintigraphy, the diagnosis can be reached with conventional imaging studies. The ectopic location of the parathyroid adenoma is a cause of negative scintigraphy. Radioguided surgery is not helpful in scan-negative ectopic parathyroid adenomas(AU)

Utilidad de la 18f-colina pet/tc en hiperparatiroidismo primario persistente o recurrente: experiencia inicial / Usefulness of 18f-choline pet / tc in persistent or recurrent primary hyperparathyroidism: initial experience

El hiperparatiroidismo persistente/recurrente representa un desafío en la localización del tejido paratiroideo hiperfuncionante. En esta subpoblación, los métodos convencionales ofrecen un menor rédito diagnóstico. La 18F-colina PET/TC podría ser una buena alternativa dada su mejor resolución espacial, capacidad de detectar glándulas ectópicas y la conjunción de la imagen molecular y anatómica. Sin embargo, la evidencia en este subgrupo de pacientes es escasa. Objetivo: evaluar la utilidad de la 18F-colina PET/TC como método de localización en el hiperparatiroidismo persistente o recurrente. Materiales y métodos: se analizaron los pacientes con 18F-colina PET/TC para hiperparatiroidismo entre diciembre de 2015 y enero de 2018 en un centro terciario de alto volumen. Se analizaron el número de lesiones, su localización, tamaño y el Standard Uptake Value máximo (SUV max) en las imágenes tempranas y tardías. Se compararon los resultados con los métodos convencionales. Resultados: 7 de 15 pacientes habían sido operados previamente (persistentes/recurrentes). La 18F-colina PET/TC detectó 6/7 casos (83,33%), la ecografía cervical 1/4 (25%) y el SPECT de paratiroides y la resonancia nuclear magnética 2/5 (40%). El SUV max obtenido fue variable, en la mitad de los casos a los 10 minutos y en los restantes a la hora; el tamaño promedio de las lesiones fue 8,61 mm (6-12 mm). Conclusiones: la 18F-colina PET/TC muestra una alta tasa de detección en los pacientes con hiperparatiroidismo persistente/recurrente. La combinación del comportamiento biológico del PET con los hallazgos morfológicos aportados por la TC con contraste endovenoso le ofrecería ventajas sobre otros estudios que podrían posicionarlo como método de primera línea en esta subpoblación. (AU)

Persistent or recurrent hyperparathyroidism represents a challenge regarding the localization of the hyper-functioning parathyroid tissue. In this subpopulation of hyperpharathyroid patients, conventional methods have a low diagnostic yield. The 18F-choline PET /CT could be a good alternative given its better spatial resolution, ability to detect ectopic glands, and the conjunction of the molecular and anatomical image. However, the evidence in this subgroup of patients is limited. Objective: to evaluate the utility of 18F-choline PET/ CT as a localization method in persistent or recurrent hyperparathyroidism. Materials and methods: patients with 18F-choline PET / CT for hyperparathyroidism between December 2015 and January 2018 in a high-volume tertiary center were included. The number of lesions, and their location, size, and maximum Standard Uptake Value (SUV) in the early and late images were analyzed. The results were compared to conventional methods. Results: 7 of 15 patients had been previously operated (persistent/recurrent). 18F-choline PET / CT detected 6/7 cases (83,33%), cervical ultrasound 1/4 (25%) and parathyroid SPECT and magnetic resonance 2/5 (40%). The maximum SUV was variable, one half at 10 minutes and the other half at 60 minutes; the average size of the lesions was 8.61 mm (6-12 mm). Conclusions: 18F-Choline PET / CT shows a high detection rate in patients with persistent / recurrent hyperparathyroidism. The combination of the biological behavior of PET with the morphological findings provided by CT with intravenous contrast would offer advantages over other studies that could position it as a first line method in this subpopulation. (AU)

Hiperparatiroidismo persistente debido a paratiromatosis / Persistent hyperparathyroidism due to parathyromatosis

RESUMEN La paratiromatosis se describe como una dolencia en la que existen múltiples nódulos de tejido paratiroideo hiperfuncionante diseminados por cuello y el mediastino, en la cual las pruebas de imagen no son efectivas para localizar las glándulas paratiroides y requiere de la combinación de un tratamiento médico y quirúrgico que en ocasiones se presenta como un desafío. Describimos el caso de una mujer de 61 años con hiperparatiroidismo primario recurrente a la que se le extirpan un total de 9 glándulas paratiroideas en 3 cirugías diferentes, y realizamos revisión de la literatura.

ABSTRACT Parathyromatosis is described as a condition in which there are multiple nodules of hyperfunctioning parathyroid tissue disseminated by the neck and mediastinum. Imaging tests are not effective in locating the parathyroid glands and requires the combination of medical and surgical treatment that sometimes is challenging. We describe the case of a 61-year-old woman with recurrent primary hyperparathyroidism. A total of 9 parathyroid glands where removed in 3 different surgeries. We also reviewed the literature.

Síndromes de neoplasia endocrina múltiple: revisión radiológica / Multiple Endocrine Neoplasia Syndromes: Radiological Review

Los síndromes de neoplasia endocrina múltiple (MEN), incluyen una serie de enfermedades con alteraciones genéticas que se caracterizan por la presencia de tumores que afectan a dos o más glándulas endocrinas. Son síndromes con una herencia autosómica dominante e incluyen tres patrones: MEN 1 (síndrome de Wermer), MEN 2 (que incluye MEN 2A o síndrome de Sipple y MEN 2B o síndrome de Wagenmann-Froboese) y MEN 4. Los adenomas paratiroideos y el carcinoma medular tiroideo, son los tumores más frecuentes del MEN tipo 1 y 2 respectivamente. Esos síndromes son más comunes en pacientes jóvenes, con patología de afectación bilateral, múltiple o multifocal y, sobre todo, en pacientes con antecedentes familiares. Es necesario el trabajo en equipo de endocrinólogos, cirujanos, oncólogos y radiólogos para optimizar el tratamiento de esos pacientes.

Multiple endocrine neoplasia (MEN) encompasses a serial of familial genetically disorders in wich tumors simultaneusly occur in two or more endocrine organs. MEN síndromes are autosomal-dominant disorders categorized into three main patterns: MEN 1 (Wermer syndrome), MEN 2 (includes MEN 2A o Sipple syndrome and MEN 2B o Wagenmann-Froboese syndrome) and MEN 4. Parathyroid adenomas and medullary thyroid carcinoma are the most frecuent tumors in MEN 1 and MEN 2 respectively. These entities will be suspected in younger patients, bilateral, multiple or multifocal disease and, specially, in patients with family background. Cooperation between endocrinologist, surgeons, oncologists and radiologists is pivotal for optimizing patient treatment.

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.

Primary Hyperparathyroidism: The Influence of Bone Marrow Adipose Tissue on Bone Loss and of Osteocalcin on Insulin Resistance

OBJECTIVES: Bone marrow adipose tissue has been associated with low bone mineral density. However, no data exist regarding marrow adipose tissue in primary hyperparathyroidism, a disorder associated with bone loss in conditions of high bone turnover. The objective of the present study was to investigate the relationship between marrow adipose tissue, bone mass and parathyroid hormone. The influence of osteocalcin on the homeostasis model assessment of insulin resistance was also evaluated. METHODS: This was a cross-sectional study conducted at a university hospital, involving 18 patients with primary hyperparathyroidism (PHPT) and 21 controls (CG). Bone mass was assessed by dual-energy x-ray absorptiometry and marrow adipose tissue was assessed by 1H magnetic resonance spectroscopy. The biochemical evaluation included the determination of parathyroid hormone, osteocalcin, glucose and insulin levels. RESULTS: A negative association was found between the bone mass at the 1/3 radius and parathyroid hormone levels (r = -0.69; p<0.01). Marrow adipose tissue was not significantly increased in patients (CG = 32.8±11.2% vs PHPT = 38.6±12%). The serum levels of osteocalcin were higher in patients (CG = 8.6±3.6 ng/mL vs PHPT = 36.5±38.4 ng/mL; p<0.005), but no associations were observed between osteocalcin and insulin or between insulin and both marrow adipose tissue and bone mass. CONCLUSION: These results suggest that the increment of adipogenesis in the bone marrow microenvironment under conditions of high bone turnover due to primary hyperparathyroidism is limited. Despite the increased serum levels of osteocalcin due to primary hyperparathyroidism, these patients tend to have impaired insulin sensitivity.

Periapical radiolucency not requiring endodontic therapy: an unusual case.

The article is a case report of an unusual manifestation of primary hyperparathyroidism seen in the mandible. Primary hyperparathyroidism is a rare disorder that can present its first symptoms in the jaws. The pre- and post-treatment radiographic features of such cases have only rarely been reported in dental literature. This case report highlights the importance of careful clinical and radiographic examination before commencing root canal treatment.

Review of imaging of the parathyroid gland in health and disease

Primary hyperparathyroidism is usually caused by a single parathyroid adenoma and it is progressively diagnosed worldwide. The recent advancement of imaging techniques changed the surgical approach of primary hyperparathyroidism patients, from wide traditional bilateral neck exploration to limited neck exploration. Pre-operative imaging is vital in localizing ectopic adenoma to lucid the map before surgical resection The manuscript at hand is a comprehensive review of the primary hyperparathyroidism covering anatomical, physiological and pathophysiological basics, to the most recent imaging modalities and their respective tasks in patient management