Hypercalcemic crisis as a presentation of primary hyperparathyroidism / Crisis hipercalcémica como presentación de hiperpara-tiroidismo primario

Abstract Hyperparathyroidism-induced hypercalcemic crisis (HIHC) is an unusual state of marked progressive pri mary 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 parathy roid 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.

Resumen La crisis hipercalcémica inducida por hiperparatiroi dismo (HIHC) es un estado inusual de hiperparatiroidis mo 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 paratiroidec tomía 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.

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)

Hipercalcemia asociada a hiperparatiroidismo primario: enfoque y manejo a partir de un caso clínico

Introducción: el calcio es el electrolito más abundante del cuerpo humano y la hipercalcemia es el trastorno común causado normalmente por el hiperparatiroidismo primario o malignidad, su manejo depende de la presentación y causa subyacente. Además, una proporción de casos se presentan como una emergencia, lo que conlleva a una mortalidad significativa. Objetivo: mostrar un caso inusual de presentación clínica de hipercalcemia asociada a hiperparatiroidismo primario y, asimismo, dar una breve revisión acerca del enfoque y el manejo de esta patología. Presentación del caso: paciente femenina de 32 años, antecedente de adenoma paratiroideo no resecado y pancreatitis, asiste por tres días de dolor abdominal de tipo cólico y de moderada intensidad, acompañado de episodios eméticos de contenido alimentario, paraclínicos iniciales con hipercalcemia severa, electrocardiograma con bloqueo auriculoventricular grado I, gases arteriales con alcalosis respiratoria aguda e hiperlactatemia. También se le practicó un TAC de abdomen donde este apareció con tumores pardos. Se ingresó a la UCI para la administración de líquidos endovenosos, diuréticos de asa y cinacalcet, pero no presentó mejoría, por lo que se indicó terapia de hemodiálisis. Discusión y conclusión: la hipercalcemia es un hallazgo frecuente. El hiperparatiroidismo primario y la neoplasia maligna son las dos causas más frecuentes de aumento de los niveles de calcio sérico y, en conjunto, representan alrededor del 90 % de todos los casos, donde los valores en suero varían entre el calcio total (8,5 y 10,5 mg/dl) y el iónico (1,16-1,31). La concentración sérica de Ca 2+ está estrechamente relacionada por las acciones de la hormona paratiroidea y el calcitriol, donde el hiperparatiroidismo primario ocurre como resultado de adenomas, hiperplasias y carcinoma. Las manifestaciones clínicas y la severidad van a estar correlacionadas con el tiempo de duración de la enfermedad, los niveles de calcio y de PTH. Dentro del tratamiento, este será guiado por su causa, sin embargo, es posible clasificarlo en tratamiento urgente y no urgente. Además, el enfoque de la hipercalcemia aguda severa es un reto diagnóstico dadas las múltiples causas que pueden llevar a este trastorno hidroelectrolítico y la rápida instauración de tratamiento que se requiere cuando es detectada.

Background: Calcium is the most abundant electrolyte in the human body, hypercalcemia is a common disorder usually caused by primary hyperparathyroidism or malignancy. A proportion of cases presenting as an emergency, leading to significant mortality. The management of hypercalcemia depends on the presentation and underlying cause. Purpose: to present an unusual case of clinical presentation of hypercalcemia associated with primary hyperparathyroidism, as well as to give a brief review about the approach and management of this pathology. Case presentation: A 32-year-old female patient, with a history of unresected parathyroid adenoma and pancreatitis, attended for 3 days of abdominal pain, moderate intensity, accompanied by emetic episodes of food content, initial paraclinical findings showed severe hypercalcemia, electrocardiogram with block grade I atrioventricular, arterial gases with acute respiratory alkalosis and hyperlactatemia. CT of the abdomen with brown tumors. She was admitted to the ICU for administration of intravenous fluids, loop diuretics, and cinacalcet without showing any improvement, so hemodialysis therapy was indicated. Discussion and conclusion: hypercalcemia is a frequent finding. Primary hyperparathyroidism and malignancy are the two most common causes of elevated serum calcium levels, together accounting for about 90 % of all cases. Serum values vary between total calcium 8.5 and 10.5 mg/dl and ionic 1.16- 1.31. Serum Ca 2+ concentration is closely related by the actions of parathyroid hormone and calcitriol. Primary hyperparathyroidism occurs as a result of adenomas, hyperplasias, or carcinoma. The clinical manifestations and severity will be correlated with the duration of the disease, calcium and PTH levels. Within the treatment, this will be guided by its cause, however, it is possible to classify it into urgent and non-urgent treatment. The approach to severe acute hypercalcemia is a diagnostic challenge given the multiple causes that can lead to this hydroelectrolyte disorder and the rapid establishment of treatment that is required when it is detected.

Radiofrequency ablation for thyroid and parathyroid disease.

Radiofrequency ablation is a well-known, safe, and effective method for treating benign thyroid nodules and recurring thyroid cancer as well as parathyroid adenomas that has yielded promising results in recent years. Since the Korean Society of Thyroid Radiology introduced the devices and the basic techniques for radiofrequency ablation in 2012, radiofrequency ablation has been approved all over the world and both the devices and techniques have improved. This review aims to instruct interventional radiologists who are doing or intend to start doing radiofrequency ablation of thyroid and parathyroid lesions, as well as thyroid and parathyroid specialists who provide pre- and post-operative care, in the training, execution, and quality control for radiofrequency ablation of thyroid nodules and parathyroid adenomas to optimize the efficacy and safety of the treatment.

Ablación por radiofrecuencia en la enfermedad tiroidea y paratiroidea / Radiofrequency ablation for thyroid and parathyroid disease

La ablación por radiofrecuencia (ARF) es un método bien conocido, seguro y eficaz para tratar los nódulos tiroideos benignos, los cánceres tiroideos recurrentes, así como los adenomas de paratiroides, con resultados prometedores en los últimos años. Los dispositivos empleados y las técnicas básicas para la ARF fueron introducidos por la Sociedad Coreana de Radiología de Tiroides (KSThR) en 2012, si bien la ARF se ha aprobado en todo el mundo, con avances posteriores tanto en dispositivos como en técnica.El objetivo de esta revisión es instruir a los radiólogos intervencionistas que pretendan realizar, o que ya estén realizando, intervenciones de ARF, así como especialistas en tiroides y paratiroides que brinden atención pre y postoperatoria, acerca de la capacitación, la ejecución y el control de calidad de la ARF de los nódulos tiroideos y adenomas paratiroideos, para optimizar la eficacia del tratamiento y la seguridad del paciente.(AU)

Radiofrequency ablation is a well-known, safe, and effective method for treating benign thyroid nodules and recurring thyroid cancer as well as parathyroid adenomas that has yielded promising results in recent years. Since the Korean Society of Thyroid Radiology introduced the devices and the basic techniques for radiofrequency ablation in 2012, radiofrequency ablation has been approved all over the world and both the devices and techniques have improved.This review aims to instruct interventional radiologists who are doing or intend to start doing radiofrequency ablation of thyroid and parathyroid lesions, as well as thyroid and parathyroid specialists who provide pre- and post-operative care, in the training, execution, and quality control for radiofrequency ablation of thyroid nodules and parathyroid adenomas to optimize the efficacy and safety of the treatment.(AU)

Paraganglioma del cuerpo carotídeo asociado a un adenoma paratiroideo: reporte de caso

Introducción: Los paragangliomas son tumores raros, y más raros aun cuando se presentan con otros tumores endocrinos. Objetivo: Presentar el caso de un paraganglioma del cuerpo carotídeo asociado con adenomas paratiroideos. Materiales y Método: Se presenta la evaluación clínica, imagenológica y fotográfica del caso. Resultados: Se presenta el caso de una paciente de 34 años con una masa cervical en región cervical de un año de evolución y que, durante los estudios de extensión, se encontró que correspondía a un paraganglioma en la bifurcación carotídea izquierda, asociada además con dos adenomas paratiroideos, que fueron resecados sin complicaciones. Discusión: Se discute la fisiopatología, el diagnóstico y manejo en relación con el caso presentado. Conclusión: La presentación de paragangliomas del cuerpo carotídeo asociadas con adenomas paratiroideos es rara, y su evaluación clínica deberá ser individualizada, dado que, si bien el manejo será en su mayoría quirúrgico, el abordaje dependerá de cada caso.

Introduction: Paragangliomas are rare tumors, and they are even rarer when they present with other endocrine tumors. Aim: To present a clinical case of a carotid body paraganglioma associated with parathyroid adenomas. Materials and Method: There are shown the clinical evaluation, images, and photos of the case. Results: We present the case of a 34 years old female patient with a cervical mass, which has grown for a year, and, after extension studies, it was found that the mass corresponded to a paraganglioma located in the left carotid bifurcation, and it was associated with two parathyroid adenomas, all the neoplasms were resected with no complications. Discussion: It is discussed physiopathology, diagnosis and management based on the presented case Conclusion: Carotid body paragangliomas associated with parathyroid adenomas are rare, and the clinical evaluation must be individual, given that, most of the management is surgical, however, the approach will depend on each case.

Safety and diagnostic performance of parathyroid hormone assay in fine-needle aspirate in suspicious parathyroid adenomas.

OBJECTIVE: To evaluate the safety and diagnostic performance of parathyroid hormone assay in fine-needle aspirate (PTH-FNA) in patients with primary hyperparathyroidism and suspicious parathyroid adenomas. METHODOLOGY: A retrospective observational study was performed in 47 patients (57.7 ±â€¯11.2 years of average age, 74% women) attending an endocrinology clinic for primary hyperparathyroidism (average calcemia: 11.6 ±â€¯1.6 mg/dl and PTH: 276 ±â€¯477 pg/mL) in which PTH-FNA was made. Sensibility, specificity, positive predictive value and negative predictive value were calculated in all surgical patients. RESULTS: Forty-seven lesions were punctured (mean adenoma maximum diameter: 1.8 ±â€¯2.6 cm): negative image in the sestamibi scan (26 patients); the discordance between ultrasonography and the sestamibi scan (6 patients); possible intrathyroidal adenomas (4 patients); a positive sestamibi scan in 2 or more localizations (4 patients); ectopic adenoma (3 patients); persistent primary hyperparathyroidism (2 patients) and atypical adenomas (2 patients). Mean PTH-FNA was 2853 ±â€¯3957 pg/mL and 68% were considered positive (PTH-FNA ≥ 100 pg/mL). No complications were detected during or after the puncture. Thirty-seven patients were operated on, 95% were cured and no parathyromatosis cases were detected. PTH-FNA ≥ 100 pg/mL as a diagnostic test had a sensitivity of 93.7%, a specificity of 100%, a positive predictive value of 100% and an negative predictive value of 71.4%. CONCLUSION: PTH-FNA is an easy and safe diagnostic test and has a high sensitivity and specificity for differentiating between parathyroid adenomas and other cervical masses in patients with primary hyperparathyroidism.

Factors associated with negative 99mTc-MIBI scanning in patients with primary hyperparathyroidism.

OBJECTIVE: The objective was to evaluate the clinical and biological factors associated with negative 99mTc-MIBI scanning in patients with primary hyperparathyroidism (PHPT). METHODS: A retrospective observational study was designed in 195 patients (mean age: 59.2 ±â€¯13.0 years; 77% woman) with PHPT (calcium: 11,3 ±â€¯1,1 mg/dl and PTH: 218 ±â€¯295 pg/ml) studied in endocrinology setting between 2013 and 2020. An univariate and multivariate analysis was made to evaluate the clinical and biological factors associated with negative 99mTc-MIBI scanning. RESULT: 50 patients (26%) with negative 99mTc-MIBI scanning had lower PTH levels (146 ±â€¯98 vs. 244 ±â€¯334; p < 0,001), adenomas with smaller sonographic dimensions (maximum diameter: 1,2 ±â€¯0,4 vs. 1,7 ±â€¯0,9 cm; p = 0,001 and volume: 0,36 ±â€¯0,43 vs. 1,7 ±â€¯4,1 cm3; p < 0,001), localized more frequently in upper parathyroid glands (37% vs 14%; p = 0,005) and associated more frequently to thyroid nodules (72% vs 57%; p = 0,045) than patients with positive scanning. 116 patients were operated and parathyroid adenomas were smaller (maximum diameter: 1,3 ±â€¯0,5 vs. 1,9 ±â€¯1,1 cm; p = 0,008 and volume: 0,30 ±â€¯0,20 vs. 1,2 ±â€¯1,1 cm3; p < 0,001), less heavy (567 ±â€¯282 vs. 1470 ±â€¯1374 mgr.; p = 0,030) and were localized more frequently in upper situation (65% vs 16%; p < 0,001) than patients with positive scanning. In the multivariate analysis an independent association between negative 99mTc-MIBI scanning and size of removed adenoma ≤1 cm (OR: 5,77; IC 95: 1,46-22,71) and upper adenoma localization were observed (OR: 8,05; IC 95%: 2,22-29,16). CONCLUSIONS: One in four patients studied for PHPT had a negative 99mTc-MIBI scanning and were independent associated with size of adenoma ≤1 cm and upper adenoma localization.

Escisión transcervical de un adenoma paratiroideo mediastínico gigante / Transcervical Excision of a Giant Mediastinal Parathyroid Adenoma

Resumen El hiperparatiroidismo primario (HPTP) es el tercer trastorno neuroendocrino más común, cuya principal causa es el adenoma paratiroideo. Dentro de la clasificación aquellos adenomas que pesan más de 3.5 g se clasifican como adenomas paratiroideos gigantes (APG), se asocian a una peor evolución y malignidad. El propósito de este artículo es presentar nuestro enfoque diagnóstico y terapéutico, en el abordaje de una paciente con HPTP asociado a un APG mediastínico. Una búsqueda en la literatura internacional de los últimos 10 años reveló que solo otros 8 centros han informado la extracción de un APG de localización y dimensiones similares al nuestro. Presentación del caso: Mujer de 66 años con antecedente de bocio multinodular, se solicitan perfil tiroideo y medición de parathormona (PTH) con reporte de 350.60 pg/mL. Se decidió realizar una gammagrafía tiroidea/paratiroidea con 99mTc sestaMIBI que reportó persistencia de una zona focal de la concentración localizada en topografía de polo inferior del lóbulo tiroideo derecho. Se complementó dicho estudio con SPECT-CT, donde se reportó imagen de paratiroides inferior derecha hiperfuncionante compatible con adenoma y se diagnosticó a la paciente con HPTP. Finalmente, se realizó resección obteniendo tumoración de 7.0 × 4.5 × 2.0 cm, con un peso de 24.5 g. Conclusiones: Nuestro caso destacó el uso de los estudios de imagen para facilitar la localización y lograr el diagnóstico aunado a la presentación clínica y el perfil bioquímico. El tratamiento selectivo se logró de manera segura a través de una técnica transcervical mínimamente invasiva, combinada con la medición de los niveles de PTH.

Abstract Primary hyperparathyroidism (HPTP) is the third most com- mon neuroendocrine disorder, its main cause is parathyroid adenoma. Within the classification, those adenomas that weigh more than 3.5 g are classified as giant parathyroid adenomas (APG), and are associated with a worse evolution and malignancy. The purpose of this article is to present our diagnostic and therapeutic approach of a patient with HPTP associated with a mediastinal APG. A search in the international literature of the last 10 years revealed that only 8 other centers have reported the extraction of an APG of similar dimensions to ours and in a similar location. Case presentation: A 66-year-old woman with a history of multinodular goiter, thyroid profile and parathormone (PTH) measurement were requested, with a report of 350.60 pg/ mL. A thyroid/parathyroid scintigraphy was performed with 99mTc sestaMIBI which reported persistence of a focal area of the concentration located in the topography of the lower pole of the right thyroid lobe. This study was complemented with SPECT-CT, which reported an image of hyperfunctioning right inferior parathyroid compatible with adenoma and the patient was diagnosed with PTH. Finally, resection was performed, obtaining a tumor measuring 7.0 × 4.5 × 2.0 cm, with a weight of 24.5 g. Conclusions: Our case highlights the use of imaging studies to facilitate localization and achieve diagnosis together with clinical presentation and biochemical profile. Selective treatment was achieved safely through a minimally invasive transcervical technique, combined with the measurement of PTH.

Hiperparatiroidismo primario por adenoma gigante de paratiroides. Abordaje quirúrgico selectivo / Primary hyperparathyroidism caused by giant parathyroid adenoma. Selective surgical approach / Hiperparatireoidismo primário devido a um adenoma gigante da paratireoide. Abordagem cirúrgica seletiva

Resumen: Introducción: el hiperparatiroidismo primario por un adenoma gigante de paratiroides es infrecuente. Los adenomas de mayor tamaño reportados ocurrieron sobre paratiroides ectópicas (mediastinales). Comparte con el carcinoma su gran tamaño y elevadas cifras de calcemia y de hormona paratiroidea, hecho que dificulta el diagnóstico. Su tratamiento quirúrgico es la paratiroidectomía mediante una cervicotomía transversa centrada en la región infrahioidea. Objetivo: presentar un caso clínico de hiperparatiroidismo primario por un adenoma gigante de paratiroides tratado quirúrgicamente mediante un abordaje selectivo. Caso clínico: paciente de 53 años, sexo femenino, con antecedentes de litiasis ureteral, dolores óseos y tumoración infrahiodea de 4 cm de diámetro que imagenológicamente presentó las características de un adenoma paratiroideo inferior izquierdo. La valoración funcional confirmó hiperparatiroidismo. Con diagnóstico de hiperparatiroidismo primario por adenoma gigante se trató quirúrgicamente a través de una incisión pequeña y centrada en la tumoración, realizándose la paratiroidectomía inferior izquierda con la cual remitió la sintomatología y normalizó la funcionalidad paratiroidea. Discusión: el hiperparatiroidismo primario por adenoma gigante de paratiroides tiene indicación quirúrgica y es curativo. El caso presentado demuestra la factibilidad y seguridad de un abordaje selectivo a lo que suma una menor afectación cosmética, dejando indemne la logia tiroidea contralateral ante futuras cirugías sobre ésta.

Summary: Introduction: primary hyperparathyroidism caused by giant parathyroid adenoma is a rather unusual condition. Reported large adenomas occurred in ectopic parathyroid glands (mediastinal). Just like carcinomas, they are large, present high calcemia and parathyroid hormone values, what complicates diagnosis. Surgical treatment consists in parathyroidectomy by means of transversal cervicotomy around the infrahyoid region. Objective: the study presents the clinical case of primary hyperparathyroidism caused by giant parathyroid adenoma that was treated by selective surgery approach. Clinical case: 53 year-old female patient with a history of uretheral lithiasis, bone pain and 4-cm-diameter infrahyoid tumor. Imagenology studies revealed the characteristics of lower left parathyroid adenoma. Functional assessment confirmed hyperparathyroidism. Upon the diagnosis of primary hyperparathyroidism caused by giant parathyroid adenoma it was surgically addressed by means of a small cut around the tumour and performing a lower left parathyroidectomy, what resulted in the remission of symptoms and normalized parathyroid functionality. Discussion: primary hyperparathyroidism caused by giant parathyroid adenoma has an indication for surgery and is therapeutic. The case presented shows the feasibility and safety of a selective approach, as well as its smaller cosmetic harm, managing to keep the contralateral thyroid loggia intact, in view of future surgeries involving it.

Resumo: Introdução: o hiperparatireoidismo primário devido a adenoma de paratireoide gigante é raro. Os maiores adenomas relatados ocorreram em paratireoides ectópicas (mediastinais). Compartilha com o carcinoma seu grande tamanho e altos níveis de cálcio e hormônio da paratireoide, o que torna o diagnóstico difícil. Seu tratamento cirúrgico é a paratireoidectomia por meio de cervicotomia transversa com foco na região infra-hióidea. Objetivo: apresentar um caso clínico de hiperparatireoidismo primário por adenoma gigante da paratireoide tratado cirurgicamente por abordagem seletiva. Caso clínico: paciente do sexo feminino, 53 anos, com história de litíase ureteral, dor óssea e tumor infra-hióideo de 4 cm de diâmetro que apresentava características de imagem de adenoma de paratireoide inferior esquerdo. A avaliação funcional confirmou hiperparatireoidismo. Com diagnóstico de hiperparatireoidismo primário por adenoma gigante, foi tratada cirurgicamente por meio de pequena incisão focada no tumor, realizando paratireoidectomia inferior esquerda com remissão dos sintomas e normalização da funcionalidade da paratireoide. Discussão: o hiperparatireoidismo primário devido ao adenoma gigante da paratireoide tem indicação cirúrgica e é curativo. O caso apresentado demonstra a viabilidade e segurança de uma abordagem seletiva que apresenta um menor envolvimento estético, deixando o espaço contralateral da tireoide sem danos para futuras cirurgias.

Paciente con fractura patológica de cúbito lado derecho

RESUMEN El hiperparatiroidismo es una enfermedad eminentemente clínica que se presenta cuando existe un aumento en la producción de hormona paratiroidea, superior a las necesidades que el organismo requiere, dando lugar a hipercalcemia, trastornos óseos y renales en un grado variable. Se presenta el caso de un paciente de 64 años con hiperparatiroidismo secundario a un adenoma paratiroideo que fue diagnosticado por hallazgo casual, posterior a que el paciente venía presentando dolores óseos frecuentes de más de un año de evolución. Durante su internación se obtuvo el diagnóstico de adenoma paratiroideo por lo que fue intervenido quirúrgicamente con exéresis y tratamiento ambulatorio.

ABSTRACT Hyperparathyroidism is an eminently clinical disease that occurs when there is an increase in the production of parathyroid hormone, greater than the needs that the body requires, leading to hypercalcemia, bone and kidney disorders to a variable degree. We present the case of a 64-year-old patient with hyperparathyroidism secondary to a parathyroid adenoma that was diagnosed by chance finding, after the patient had been presenting frequent bone pain of more than a year of evolution. During his hospitalization, a diagnosis of parathyroid adenoma was obtained, for which he underwent surgery with excision and outpatient treatment.

Tumor pardo del paladar, presentación atípica de hiperparatiroidismo primario en adolescente: buena evolución y normalización de la masa ósea tras paratiroidectomía / Brown tumor of the palate, atypical presentation of primary hyperparathyroidism in an adolescent: good evolution and bone mass normalization after parathyroidectomy

El hiperparatiroidismo primario (HPTP) es poco frecuente en niños y adolescentes. Hay escasos datos para el manejo de estos pacientes en pediatría. Las glándulas paratiroideas son glándulas endocrinas que secretan hormona paratiroidea (PTH) y regulan el metabolismo del calcio y del fósforo. La sobreexpresión de PTH se llama hiperparatiroidismo, que se clasifica en primario, secundario y terciario. En los adolescentes, 80 a 92% de los hiperparatiroidismos primarios se deben a adenoma paratiroideo. Presentamos el caso clínico de una adolescente con una primera manifestación atípica de HPTP, la presencia de un tumor pardo del paladar, presentación rara de adenoma paratiroideo, acompañado de hipercalcemia, marcada elevación de PTH y varias lesiones óseas. (AU)

Primary hyperparathyroidism (PHPT) in children and adolescents is uncommon. Guidelines for management in pediatric patients are limited. Parathyroid glands are endocrine glands that secrete parathyroid hormone (PTH) and regulate calciumphosphate metabolism. The overexpression of PTH is called hyperparathyroidism, and is classified as primary, secondary, and tertiary. In adolescents, 80 to 92% of PHPT cases are due to a parathyroid adenoma. We present here a case report of an adolescent with a brown tumor of the palate as the first manifestation of the disease, atypical and rare presentation of parathyroid adenoma in an adolescent. She had hypercalcemia, marked elevation of PTH and bone lesions. (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)

Tumor de células gigantes (osteoclastoma) multicentrico, diagnóstico incidental de adenoma paratiroideo reporte de caso / Multicenter giant cell tumor (osteoclastoma), incidental diagnosis of parathyroid adenoma case report

Los tumores de células gigantes (TDG) llamados también osteoclastomas o tumores pardos cuando se hallan dentro la esfera endocrinológica, son uno de los tumores menos frecuentes, más controversial y menos predecible en su comportamiento. Se producen como consecuencia del exceso de la actividad osteoclastica, como ocurre en el caso del hiperparatiroidismo, mismo que es un desorden endocrino común, por lo general asintomático y diagnosticado por el hallazgo fortuito de hipercalcemia. El diagnóstico de los osteoclastomas suele ser un reto, el alto índice de sospecha es esencial y la biopsia es el estándar de oro para el diagnóstico. Presentamos el caso de un hombre de 42 años quien presentó fracturas patológicas de radio derecho y tibia izquierda, gammagrama óseo con Tc ­ 99m con múltiples lesiones óseas , hormona paratiroidea (PTH) elevada, hipercalcemia, gammagrama de paratiroides con MIBI con presencia de adenoma paratiroideo, la biopsia de las lesiones óseas con presencia de células gigantes multinucleadas correspondientes a osteoclastomas; se llevó a cabo paratiroidectomia y el examen histopatológico confirmó la presencia de un adenoma paratiroideo.

Giant cell tumors (TDG), also called osteoclastomas or brown tumors when they are within the endocrinological sphere, are one of the least frequent, most controversial and least predictable tumors in their behavior. They occur as a consequence of excess osteoclastic activity, as occurs in the case of hyperparathyroidism, which is a common endocrine disorder, generally asymptomatic and diagnosed by the fortuitous finding of hypercalcemia. Diagnosing osteoclastomas is usually challenging, the high index of suspicion is essential, and biopsy is the gold standard for diagnosis. We present the case of a 42-year-old man who presented pathological fractures of the right radius and left tibia, a bone scan with Tc-99m with multiple bone lesions, elevated parathyroid hormone (PTH), hypercalcemia, parathyroid scan with MIBI with the presence of parathyroid adenoma, the biopsy of the bone lesions with the presence of multinucleated giant cells corresponding to osteoclastomas; parathyroidectomy was performed and histopathological examination confirmed the presence of a parathyroid adenoma.

Identification of Occult Adenomas in Primary Hyperparathyroidism With 18F-fluorocholine PET/CT. / Identificación de adenomas ocultos en hiperparatiroidismo primario con PET-TC 18F-fluorocolina.

INTRODUCTION: Single parathyroid adenomas are the most common cause of primary hyperparathyroidism (PHP) in our population. Parathyroidectomy is still the only potentially curative treatment and requires preoperative localization imaging studies to perform selective surgery. In patients with negative results on conventional tests, PET/CT has demonstrated higher sensitivity rates. METHODS: A prospective cohort study was designed, including 34 patients diagnosed with PHP between 2017 and 2019, candidates for surgery with negative preoperative localization tests with scintigraphy and MIBI SPECT/CT. All patients underwent PET/CT with 18F-Fluorocholine. The clinical, biochemical and postoperative outcome results were compared with a control group of 30 patients with positive standard tests. RESULTS: Hyperfunctional parathyroid tissue was detected in 85% of the patients that had undergone choline PET/CT. The selective resection of the adenoma identified in these patients achieved curative criteria in 87% of the cases without undergoing bilateral cervical surgical exploration. The preoperative levels of PTH, calcemia and gland weight were significantly lower in this group compared to the control group. No differences were identified in cure criteria or approach between the 2groups. CONCLUSION: In our study, choline PET/CT showed higher detection rates compared to the gold standard. The increase provides the opportunity to perform unilateral selected adenoma resection, especially in patients with smaller adenomas associated with lower calcemia and PTH levels and patients with previous cervical surgery.

High capacity of ultrasound for locating parathyroid adenomas in endocrinology (the ETIEN 4 study). / Elevada capacidad de localización ecográfica de adenomas paratiroideos en endocrinología (estudio ETIEN 4).

OBJECTIVE: To assess the diagnostic performance of neck ultrasound examination performed by endocrinologists to locate parathyroid adenomas in patients with primary hyperparathyroidism (PHPT). METHODOLOGY: A retrospective observational study in 135 patients (mean age, 60.0±12.3 years; 74.8% females) seen at endocrinology for PHPT (mean calcium level, 11.3±1.2mg/dL mean PTH level, 240.4±346.8pg/mL) who underwent neck ultrasound examinations at the endocrinology department. 99mTc-MIBI parathyroid scintigraphy was performed before surgery in all patients. Sensitivity, specificity, positive predictive value, and negative predictive value were calculated in surgical patients. RESULTS: Ninety-eight patients underwent surgery for PHPT and 97.8% were cured at 6 months. Parathyroid ultrasound had a sensitivity of 85% (95% CI: 75.7%-91.2%) and a positive predictive value (PPV) of 95.2% (95% CI: 87.5%-98.4%) to locate parathyroid adenomas 1.7±0.9cm in maximum diameter (69.4% in smaller glands), showing a high correlation (r=0.661 and r=0.716) with maximum diameter and volume of the excised adenoma. Sixty percent of patients had nodular thyroid disease (64.2% bilateral nodules with mean maximum diameter of 1.5±0.9cm), and thyroidectomy was performed in 31.6%. The highest diagnostic performance was seen with a combination of neck ultrasound and 99mTc-MIBI scintigraphy (sensitivity: 96.8% and PPV: 95.8%). CONCLUSION: In our area, parathyroid adenoma localization with ultrasound performed by endocrinologists has a high diagnostic yield and allows for detecting nodular thyroid disease in 60% of patients.

Adenoma paratiroideo mediastínico como causa de hiperparatiroidismo primario / Mediastinal parathyroid adenoma as a cause of primary hyperparathyroidism

Resumen Las glándulas paratiroides participan en la regulación de las concentraciones de calcio al producir parathormona como respuesta a la hipocalcemia. Los adenomas paratiroideos, que rara vez se encuentran en mediastino, son la causa más importante de hiperparatiroidismo primario, una enfermedad rara con manifestaciones clínicas graves y que se diagnostica por la com binación de concentraciones elevadas de calcio sérico y paratohormona. Se considera que el adenoma paratiroideo ectópico se deriva de la migra ción de las glándulas paratiroides al mediastino y se conoce como una causa importante del fracaso de la exploración primaria del hiperparatiroi dismo primario. Se presenta un caso de adenoma paratiroideo mediastíni co en una mujer de 48 años que llevó a la aparición de hiperparatiroidismo primario. El adenoma se extirpó mediante esternotomía media total.

Abstract Parathyroid glands participate in regulation of calcium concentrations by producing parathormone in response to hypocalcemia. Parathyroid adeno mas, which are rarely found in the mediastinum, are the most important cause of primary hyperparathyroidism, which is a rare disease with serious clinical manifestations and is diagnosed by combination of high concentrations of serum calcium and parathormone. It is considered that ectopic parathyroid adenoma is derived from the migration of the parathyroid glands to the mediastinum and is known as an important cause of the failure of primary exploration of primary hyperparathyroidism. We present a case of medias tinal parathyroid adenoma in a 48-year-old woman that led to appearance of primary hyperparathyroidism. Adenoma was removed by total median sternotomy.

Hyperparathyroidism during pregnancy. Is it a diagnostic and therapeutic challenge?

BACKGROUND: Primary hyperparathyroidism (PHPT) is characterized by an increase in parathyroid hormone (PTH) and hypercalcemia, which, when present during pregnancy, increases both maternal and fetal morbidity and mortality. OBJECTIVE: Emphasize the importance of surgical intervention in primary hyperparathyroidism during pregnancy. CLINICAL CASE: A 27-year-old female with a pregnancy of 27.2 weeks of gestation, with a diagnosis of symptomatic primary hyperparathyroidism secondary to parathyroid adenoma, a history of nephrolithiasis and severe acute pancreatitis, surgery was decided upon finding intrathyroid right parathyroid adenoma, post-surgical course with adequate evolution and remission of hyperparathyroidism. CONCLUSIONS: Parathyroidectomy in primary hyperparathyroidism during pregnancy is safe.

INTRODUCCIÓN: El hiperparatiroidismo primario (HPTP) se caracteriza por un aumento de la hormona paratiroidea (PTH) e hipercalcemia, que aumenta la morbimortalidad materna y fetal cuando se presenta durante el embarazo. OBJETIVO: Enfatizar la importancia de la intervención quirúrgica en el hiperparatiroidismo primario durante el embarazo. CASO CLÍNICO: Paciente femenino de 27 años de edad con embarazo de 27.2 semanas de gestación, con diagnóstico de hiperparatiroidismo primario sintomático secundario a adenoma paratiroideo, antecedentes de nefrolitiasis y pancreatitis aguda grave; al decidir realizar un procedimiento quirúrgico se identificó un adenoma paratiroideo derecho intratiroideo; el curso posquirúrgico mostró adecuada evolución y remisión del hiperparatiroidismo. CONCLUSIÓN: La paratiroidectomía en el hiperparatiroidismo primario durante el embarazo es segura.

Autofluorescencia: un método innovador en la identificación intraoperatoria de adenomas paratiroideos / Autofluorescence: an innovative method in the intraoperative identification of parathyroid adenomas

RESUMEN Introducción El tratamiento definitivo del hiperparatiroidismo primario es la resección quirúrgica de la glándula paratiroidea anómala. Su identificación resulta un desafío aun para cirujanos expertos. Hasta el momento no se han descripto métodos inocuos y efectivos para la identificación intraoperatoria de las glándulas. Tenemos como objetivo reportar la experiencia del uso de autofluorescencia en la identificación de las glándulas paratiroideas. Método Se incluyeron pacientes con hiperparatiroidismo primario evaluados preoperatoriamente con laboratorio, ecografía cervical y centellografía con Tc-99 MIBI. Durante el acto operatorio se utilizó un método de autofluorescencia (VINFLUO-P) para identificar las glándulas paratiroides (GP). Se analizó la intensidad lumínica de las (GP) normales y anómalas (AP) y distintas covariables. Se dosó PTH ultra rápida post resección del AP y se evaluó la histopatología de la pieza intraoperatoriamente. Resultados Se incluyeron 59 pacientes. La ecografía preoperatoria predijo la ubicación correcta en el 68% y el centellograma Tc-99 MIBI el 75% de los AP. La localización más frecuente fue inferior derecha (29%). El VINFLUO-P facilitó la visualización de las GP y los AP en el 100% de los pacientes con un aumento del 27% respecto a la luz blanca. Se evidenció un descenso postoperatorio de PTH del 76,44% y de la calcemia en 1,8 mg/dl. La intensidad de la luz reflejada por los AP fue mayor que la de las GP normales (p <0,001). Se observó una relación lineal entre PTH e intensidad lumínica de AP. (CC = 0,448; p = 0,045). El patrón arquitectural sólido de los AP evidenció una asociación negativa (CC = -0,4709 p = 0,03). Conclusión La utilización del VINFLUO-P demostró ser efectivo para la identificación de las GP normales y patológicas. Las glándulas anómalas resultaron con mayor fluorescencia que los tejidos normales.

ABSTRACT Introduction The treatment of primary hyperparathyroidism consists on the resection of the abnormal parathyroid gland (PG). Identification of PGs is challenging even for expert surgeons. Currently, there are no effective and harmless methods for intraoperative identification of PGs. The aim of this study is to report our experience with the identification of PGs using autofluorescence. Materials and methods Patients with diagnosis of primary hyperparathyroidism were included in the study. Patients were preoperatively worked up with labs [parathyroid hormone (PTH), serum calcium], neck ultrasound (US) and Technetium (99mTc) sestamibi. The parathyroid gland Intraoperative fluorescent visualization (PG-IFV) method was used during the surgery to identify PGs. The fluorescent intensity ratio of normal PGs and parathyroid adenomas (PA) was analyzed and correlated to different variables. All patients underwent a post-resection rapid PTH analysis and frozen section. Results Fifty-nine patients were included in the study. The US accurately predicted the location of the PA in 68% of the cases, while 99mTc sestamibi was accurate in 75% of the cases. The most frequently reported localization of the adenoma was right inferior (29%). PG-IFV facilitated the visualization of the PGs in 100% of the cases, with a 27% increase in the visualization of the PGs when compared to white light. The postoperative PTH decreased 76.4% and the calcium 1.8 mg/dl. The fluorescent intensity ratio of the PAs was significantly higher than normal PGs (44.4 vs 27.2, p <0.001). There was positive correlation between the PTH and the fluorescent intensity ratio of the PAs [Spearman's correlation coefficient (SCC) = 0.448; p = 0.045]. The solid histoarchitectural pattern of the PAs presented a negative correlation with fluorescent intensity ratio (SCC = -0.4709, p = 0.03). Conclusion The use of PG-IFV is an effective method for intraoperative identification of normal and abnormal PGs. The fluorescent intensity ratio of abnormal PGs was significantly higher than normal PGs.

Crisis hipercalcémica causada por adenoma paratiroideo: reporte de un caso clínico / Hypercalcemic crisis due to parathyroid adenoma: report of a clinical case

La hipercalcemia es un trastorno común que representa aproximadamente el 0,6% de todas las admisiones médicas agudas. El hiperparatiroidismo primario (HPTP) y las neoplasias malignas son las dos causas más comunes de elevación de los niveles séricos de calcio; constituyen, en conjunto, alrededor del 90% de todos los casos. La presentación sintomática clásica de la hipercalcemia se observa con relativa poca frecuencia en el mundo desarrollado; la presentación más común es la detección asintomática en las pruebas bioquímicas. Sin embargo, en casos raros, el HPTP puede desarrollar hipercalcemia aguda, grave y sintomática, llamada crisis hipercalcémica (CH). Esta condición se asocia a alteraciones profundas en el estado mental y las funciones cardíaca, renal y gastrointestinal en presencia de concentraciones marcadamente elevadas de calcio sérico y paratohormona (PTH). Mientras que algunas elevaciones en el calcio sérico pueden ser bien toleradas, los síntomas de la CH son severos. Si el tratamiento se retrasa, la CH puede provocar la muerte. Describimos el caso de un paciente masculino que ingresa en la unidad de cuidados críticos por una CH secundaria a un HPTP por adenoma paratiroideo. (AU)

Hypercalcaemia is a most common disorder, accounting for approximately 0,6% of all acute medical admissions. Primary hyperparathyroidism (PHPT) and malignancy are the two most common causes of increased serum calcium levels, together accounting for about 90% of all cases. The classical symptomatic presentation of hypercalcaemia is seen relatively rarely in the developed world, the most common presentation being asymptomatic and detected following on biochemical testing. However, in rare cases HPTP can result in acute, severe and symptomatic hypercalcemia, called hypercalcemic crisis (HC). This condition is associated with profound disturbances in mental status, and cardiac, renal, and gastrointestinal function in the presence of markedly increased serum calcium and parathyroid hormone (PTH) concentrations. While some elevations in serum calcium can be well tolerated, symptoms of HC are severe. If treatment is delayed, HC can result in death. We describe herein a case of a male patient who was admitted to the intensive care unit as a consequence of HC resulting from elevated PTH, secondary to a parathyroid adenoma. We describe the case of a male patient who was admitted to the critical care unit for a HC mediated by PTH secondary to a parathyroid adenoma. (AU)