Hypercalcium crisis and postoperative hungry bone syndrome caused by primary hyperparathyroidism: a case report / 临床耳鼻咽喉头颈外科杂志

To review the diagnosis and treatment of a case of hypercalcium crisis caused by primary hyperparathyroidism（PHPT） and prophylactic treatment of hungry bone syndrome. In a 32-year-old male with hypercalcemia, the main manifestations were loss of appetite, nausea, polyuria, polydipsia, fatigue, lethargy, etc. parathyroid hormone, serum calcium increased, thyroid function was normal, thyroid color ultrasound and MRI showed space-occupying behind the right thyroid, radionuclide examination showed abnormal imaging agent concentration in the right parathyroid area, there was a history of pathological fracture. Clinically diagnosed as hypercalcemia crisis secondary to PHPT.

Hiperparatiroidismo primario: experiencia en cirugía por mini abordaje unilateral / Primary hyperparathyroidism: experience in surgery by unilateral mini-approach

INTRODUCCIÓN: En el hiperparatiroidismo primario el origen del trastorno, como su nombre lo indica, está en la propia glándula paratiroides, la cual genera una secreción autónoma y excesiva. La cirugía de las glándulas paratiroides evolucionó en forma considerable en los últimos 30 a 40 años, pasamos de exploraciones cervicales exhaustivas, hasta una época en que gracias al desarrollo tecnológico y sobre todo medicina nuclear, podemos localizar en forma preoperatoria el tejido patológico; siendo esta a su vez la base fundamental en la realización de procedimientos más selectivos. OBJETIVO: mostrar la casuística de cirugía por mini abordaje de la glándula paratiroides en el hiperparatiroidismo primario en un centro mutual de Montevideo. MATERIAL Y MÉTODOS: Realizamos un estudio observacional descriptivo y retrospectivo. Se estudió una muestra de 18 pacientes con diagnóstico de hiperparatiroidismo primario y con sospecha de lesión única los cuales fueron intervenidos en un centro mutual de la ciudad de Montevideo entro julio de 2017 y enero de 2020. CONCLUSIÓN: La cirugía por mini abordaje de la glándula paratiroides puede ser aplicada en el hiperparatiroidismo primario en pacientes seleccionados con las ventajas de; tener un menor tiempo quirúrgico, ser ambulatoria (reintegro al hogar en pocas horas), indemnidad de la logia tiroidea contralateral, mejor resultado estético con similar tasa de éxito que la cirugía convencional.

BACKGROUND: In primary hyperparathyroidism, the origin of the disorder, as its name indicates, is in the parathyroid gland itself, which generates excessive and autonomous secretion. Parathyroid gland surgery has evolved dramatically in the last 30 to 40 years, from exhaustive cervical examinations, to nowadays when, thanks to technological development and especially nuclear medicine, we can locate pathological tissue preoperatively; this, in fact, is the fundamental basis for the performance of more selective procedures. OBJECTIVE: to show the casuistry of mini-approach surgery of the parathyroid gland in primary hyperparathyroidism in a mutual center in Montevideo. METHODS: We carried out a descriptive and retrospective observational study. We studied a sample of 18 patients diagnosed with primary hyperparathyroidism and a single suspicious lesion, who underwent surgery in a private center in the city of Montevideo from July 2017 to January 2020. CONCLUSION: Mini-approach surgery of the parathyroid gland can be applied in primary hyperparathyroidism in selected patients, with the advantages of a shorter surgical time, ambulatory (return home in a few hours), keeping the indemnity of the contralateral thyroid loggia, a better cosmetic result with a similar success rate than conventional surgery.

Delayed sampling of intraoperative parathormone may be unnecessary during parathyroidectomy in kidney-transplanted and dialysis patients / A amostragem tardia de paratormônio intraoperatório pode ser desnecessária durante paratireoidectomia em pacientes transplantados renais e em diálise

Abstract Introduction: Some authors advise in favor of delayed sampling of intraoperative parathormone testing (ioPTH) during parathyroidectomy in dialysis and kidney-transplanted patients. The aim of the present study was to evaluate the intensity and the role of delayed sampling in the interpretation of ioPTH during parathyroidectomy in dialysis patients (2HPT) and successful kidney-transplanted patients (3HPT) compared to those in single parathyroid adenoma patients (1HPT). Methods: This was a retrospective study of ioPTH profiles in patients with 1HPT, 2HPT, and 3HPT operated on in a single institution. Samples were taken at baseline ioPTH (sampling at the beginning of the operation), ioPTH-10 min (10 minutes after excision of the parathyroid glands), and ioPTH-15 min (15 minutes after excision of the parathyroid glands). The values were compared to baseline. Results: Median percentage values of ioPTH compared to baseline (100%) were as follows: 1HPT, ioPTH-10 min = 20% and ioPTH-15 min = 16%; 2HPT, ioPTH-10 min = 14% and ioPTH-15 min = 12%; 3HPT, ioPTH-10 min = 18% and ioPTH-15 min = 15%. Discussion: The reduction was equally effective at 10 minutes in all groups. In successful cases, ioPTH decreases satisfactorily 10 minutes after parathyroid glands excision in dialysis and transplanted patients, despite significant differences in kidney function. The postponed sampling of ioPTH appears to be unnecessary.

Resumo Introdução: Alguns autores aconselham a favor de se fazer uma amostragem tardia de teste de paratormônio intraoperatório (PTHIO) durante paratireoidectomia em pacientes transplantados renais e em diálise. O objetivo do presente estudo foi avaliar a intensidade e o papel da amostragem tardia na interpretação do PTHIO durante paratireoidectomia em pacientes em diálise (2HPT) e pacientes com transplante renal bem sucedido (3HPT) em comparação com aqueles em pacientes com adenoma único de paratireoide (1HPT). Métodos: Este foi um estudo retrospectivo dos perfis de PTHIO em pacientes com 1HPT, 2HPT, e 3HPT operados em uma única instituição. Foram coletadas amostras de PTHIO basal (amostragem no início da operação), PTHIO-10 min (10 minutos após a excisão das glândulas paratireoides), e PTHIO-15 min (15 minutos após a excisão das glândulas paratireoides). Os valores foram comparados aos resultados basais. Resultados: Os valores percentuais medianos do PTHIO em comparação aos basais (100%) foram os seguintes: 1HPT, PTHIO-10 min = 20% e PTHIO-15 min = 16%; 2HPT, PTHIO-10 min = 14% e PTHIO-15 min = 12%; 3HPT, PTHIO-10 min = 18% e PTHIO-15 min = 15%. Discussão: A redução foi igualmente eficaz aos 10 minutos em todos os grupos. Em casos de sucesso, o PTHIO diminui satisfatoriamente 10 minutos após a excisão das glândulas paratireoides em pacientes em diálise e transplantados, apesar das diferenças significativas na função renal. A amostragem tardia de PTHIO parece ser desnecessária.

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.

Normocalcemic with elevated post-operative parathormone in primary hyperpara-thyroidism: 9 case reports and literature review / 北京大学学报(医学版)

OBJECTIVE@#To summarize and analyze the clinical characteristics of primary hyperpara-thyroidism (PHPT) with normocalcemic parathormone elevation (NPE) after surgical treatment, so as to improve the therapeutic ability and standardized post-operative follow-up of PHPT patients.@*METHODS@#Nine patients who were diagnosed with PHPT in the Department of Endocrinology of China-Japan Friendship Hospital from August 2017 to November 2019 were selected as the subjects. They all developed NPE within 6 months after surgical treatment. The clinical features and outcomes were collected and analyzed retrospectively, in addition, the related literature was reviewed.@*RESULTS@#Clinical features: among the 9 patients, 6 were middle-aged and elderly females and 3 were male. The main clinical manifestations were bone pain, kidney stones, nausea and fatigue except for one case of asymptomatic PHPT. Pre-operative examination showed high serum calcium [(3.33±0.48) mmol/L], low serum phosphorus [0.76 (0.74, 0.78) mmol/L], high 24-hour urinary calcium [8.1(7.8, 12.0) mmol/24 h], obviously elevated intact PTH [(546.1±257.7) ng/L], vitamin D deficiency [25-hydroxyvitamin D3 (21.0±5.7) nmol/L]. Serum levels of bone alkaline phosphatase [7 patients 41.3(38.6, 68.4) μg/L, 2 patients >90 μg/L] and N-terminal midcourse osteocalcin (>71.4 μg/L) were significantly elevated. The estimated glomerular filtration rate decreased in 2 patients. Imaging examination: 7 patients had osteoporosis. Renal calculi were found in 3 patients by renal ultrasound. Imaging examination of parathyroid glands found definite lesions in all the patients, including 2 cases of multiple lesions and 7 cases of single lesions.@*TREATMENT AND OUTCOME@#two patients underwent parathyroidectomy, while other patients were treated with microwave thermal ablation. PTH increased 1 month after therapy [(255.0±101.4) ng/L], and no recurrent lesions were found by parathyroid ultrasound. After combined treatment with cal-cium and vitamin D for six months, PTH decreased significantly and the level of serum calcium remained normal at anytime during the follow-up period.@*CONCLUSION@#The occurrence of postoperative NPE may be related to the higher pre-operative PTH, vitamin D deficiency and lower creatinine clearance. However, NPE may not predict recurrent hyperthyroidism or incomplete parathyroidectomy. Adequate calcium and vitamin D supplementation after surgery seems to be beneficial for patients with NPE. Post-operative follow-up of PHPT patients should be standardized to prevent and treat post-operative NPE.

Denosumab can be used successfully as a bridge to surgery in patients with severe hypercalcemia due to primary hyperparathyroidism

SUMMARY Severe hypercalcemia is a medical emergency that requires immediate and aggressive management. Primary hyperparathyroidism (PHPT) often causes severe hypercalcemia. Volume resuscitation, parenteral salmon calcitonin, and administration of intravenous bisphosphonates are common measures used to stabilize patients. However, the use of these measures is inadequate in several patients and may even be contraindicated in individuals with renal insufficiency or severe systemic illness. This study demonstrated the efficacy and safety of denosumab in patients with severe hypercalcemia due to PHPT, when immediate surgery was not feasible. We present four patients with severe hypercalcemia due to PHPT. Immediate surgery was not feasible because the patients had severe systemic illness, such as seizures and altered sensorium (case 1); acute severe pancreatitis (cases 2 and 3); or coronavirus disease 2019 pneumonia (case 4). Intravenous normal saline and parenteral salmon calcitonin were inadequate for controlling hypercalcemia. Intravenous bisphosphonates were avoided because of severe systemic illness in all cases and impaired renal function in three cases. Denosumab was administered to control hypercalcemia and allow the stabilization of patients for definitive surgical management. Following denosumab administration, serum calcium levels normalized, and general condition improved in all patients. Three patients underwent parathyroidectomy after two weeks and another patient after eight weeks. The use of denosumab for the management of severe hypercalcemia due to PHPT is efficacious and safe in patients when immediate surgical management is not feasible due to severe systemic illness.

Medición de hormona paratiroidea intraoperatoria y su relación con el riesgo de enfermedad persistente post-paratiroidectomía por hiperparatiroidismo primario / Measurement of intraoperative parathyroid hormone and its relation to the risk of persistent post-parathyroidectomy disease due to primary hyperparathyroidism

Introducción: La monitorización con hormona tiroidea intraoperatoria (IOPTH) es utilizada en la práctica médica para predecir la cura bioquímica durante la paratiroidectomía del hiperparatiroidismo primario. Sin embargo, existen diversos criterios utilizados para predecir la normocalcémica luego de una paratiroidectomía. El propósito de este estudio es determinar cuál criterio es aquel que se corresponde con el menor riesgo de presentar un hiperparatiroidismo persistente luego de una paratiroidectomía por hiperparatiroidismo primario. Material y métodos: Se han analizado publicaciones realizadas en los últimos 10 años tomando como fuente de recopilación la base de datos de Pub-Med, trabajos publicados en Journal of the American Medical Associatiion y World Journal of Surgery. Conclusión: El criterio de IOPTH final en valores por debajo de los 65 pg/mL, y en especial, por debajo de los 40 pg/mL parece comportarse como mejor predictor de bajo riesgo de enfermedad persistente en comparación con el criterio de disminución de IOPTH por debajo del 50% del valor basal (AU)

Introduction: Monitoring with intraoperative thyroid hormone (IOPTH) is used in medical practice to predict the biochemical cure during the parathyroidectomy of primary hyperparathyroidism. However, there are several criteria used to predict normocalcemia after a parathyroidectomy. The purpose of this study is to determine which criterion is the one that correspons to the lowest risk of presenting a persistent hyperparathyroidism after a parathyroidectomy due to primary hyperparathyroidism. Materials and methods: Publications made in the last 10 years have been analyzed taking as a source of compilations the PubMed database, works published in Journal of the American Medical Association and World Journal of Surgery. Conclusiion: The final IOPTH criterion in values below 65 pg/mL, and especially below 40 pg/mL, seems to be the best predictor of low risk of persistent disease compared to the IOPTH decrease criterior Below 50% of the basal value (AU)

Granuloma Periférico de Células Gigantes e Hiperparatiroidismo. Presentación de un Caso / Peripheral Giant Cell Granuloma and Hyperparathyroidism. Case Report

RESUMEN: Se presenta el caso de un paciente sexo femenino 31 años, insuficienciente renal crónica en hemodiálisis, hipertensa secundaria, consulta por aumento de volumen oral en relación a encía marginal vestibular izquierda, 6 meses de evolución. Al examen se observa aumento de volumen de 15 mm en relación a piezas. 3.6 y 3.7. Radiografía muestra compromiso periodontal pieza 3.7. Se realiza biopsia excisional y exodoncia pieza 3.7. Histopatología informa granuloma periférico de células gigantes. Se solicitan exámenes de laboratorio para estudio de hiperparatiroidismo (PTH elevada, calcemia fosfatemia normales). Evoluciona favorablemente. Sin signos de recidiva a los 3 meses. El diagnóstico histopatológico de lesión de células gigantes debe ser complementado con la clínica, exámenes de laboratorio e imagenología, el objetivo final es definir si tal diagnóstico corresponde a un tumor pardo, a un granuloma central de células gigantes o a un granuloma periférico de células gigantes. En la literatura los reportes de asociación entre granuloma periférico de células gigantes e hiperparatiroidismo son escasos. Este caso corresponde a granuloma periférico células gigantes, asociado a hipersecreción de paratohormona.

ABSTRACT: A 31-year-old female patient with chronic renal insufficiency in hemodialysis and secondary hypertension consulted due to an enlarging intraoral lesion in relation to the left marginal vestibular gingival, with 6 months of evolution. A 15mm mass in relation to mandibular left molars was noted. Radiograph showed periodontal involvement of mandibular left second molar. Excisional biopsy and tooth 3.7 extraction were performed. Histopathology reported a peripheral giant cell granuloma. Laboratory tests were requested for suspected hyperparathyroidism (aiding in the diagnosis) (high PTH, normal phosphatemia and calcemia). Evolution was favorable, without signs of reappearance after 3 months. The histopathological diagnosis of giant cell lesions should be complemented with clinical, laboratory and imaging tests. The final objective is to make the differential diagnosis between brown tumor, central giant cell granuloma and peripheral giant cell granuloma because the treatment varies considerably. There are few reports in the literature about the association between peripheral giant cell granuloma and hyperparathyroidism. This case corresponds to peripheral giant cell granuloma, possibly associated with parathormone hypersecretion.

Prediction of bone mass changes after successful parathyroidectomy using biochemical markers of bone metabolism in primary hyperparathyroidism: is it clinically useful?

ABSTRACT Objective To measure type 1 serum amino-terminal propeptide procollagen (P1NP) and type 1 cross-linked C-terminal telopeptide collagen (CTX) before parathyroidectomy (PTX) in PHPT patients, correlating these measurements with bone mineral density (BMD) changes. Subjects and methods 31 primary hyperparathyroidism (HPTP) were followed from diagnosis up to 12-18 months after surgery. Serum levels of calcium, parathyroid hormone (PTH) vitamin D, CTX, P1NP, and BMD were measured before and 1 year after surgery. Results One year after PTX, the mean BMD increased by 8.6%, 5.5%, 5.5%, and 2.2% in the lumbar spine, femoral neck (FN), total hip (TH), and distal third of the nondominant radius (R33%), respectively. There was a significant correlation between BMD change 1 year after the PTX and CTX (L1-L4: r = 0.614, p < 0.0003; FN: r = 0.497, p < 0.0051; TH: r = 0.595, p < 0.0005; R33%: r = 0.364, p < 0.043) and P1NP (L1-L4: r = 0,687, p < 0,0001; FN: r = 0,533, p < 0,0024; TH: r = 0,642, p < 0,0001; R33%: r = 0,467, p < 0,0079) preoperative levels. The increase in 25(OH)D levels has no correlation with BMD increase (r = -0.135; p = 0.4816). On linear regression, a minimum preoperative CTX value of 0.331 ng/mL or P1NP of 37.9 ng/mL was associated with a minimum 4% increase in L1-L4 BMD. In TH, minimum preoperative values of 0.684 ng/mL for CTX and 76.0 ng/mL for P1NP were associated with a ≥ 4% increase in BMD. Conclusion PHPT patients presented a significant correlation between preoperative levels of turnover markers and BMD improvement 1 year after PTX.

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.

Cirugía mayor ambulatoria de la glándula tiroides y paratiroides: resultados tras 6 años de experiencia / Major outpatient surgery of the thyroid and parathyroid glands: results after 6 years experience

Objetivo: La cirugía mayor ambulatoria (CMA) y con estancia inferior a 24 horas (overnight stay) se ha consolidado en muchas patologías aunque en cirugía tiroidea y paratiroidea resulta controvertido. El objetivo es valorar nuestros resultados en cirugía del tiroides y paratiroides en régimen de CMA durante un periodo de 6 años. Material y Métodos: Estudio observacional prospectivo de pacientes con indicación de hemitiroidectomía o paratiroidectomía enfocada en régimen CMA entre enero de 2011 y diciembre de 2016. Los pacientes cumplían criterios de CMA. En los primeros años se excluyeron los nodulos tiroideos mayores de 3 cm y los pacientes ASA III. Los pacientes ingresaron la mañana de la intervención, tras el acto quirúrgico pasaron a la Unidad de Recuperación Posquirúrgica y posteriormente a sala de hospitalización. Tras 8 h, si cumplían criterios, fueron dados de alta. Resultados: Se intervinieron 270 pacientes, la tasa de aplicabilidad global fue del 59%, la tasa de aceptabilidad global del 83,6% y el índice de sustitución del 49,2%. El índice de ingresos no deseados fue del 10,4% para la paratiroidectomía y del 17,6% en la cirugía del tiroides. Ningún paciente presentó complicaciones mayores en su domicilio. El grado de satisfacción fue alto o muy alto en el 94% de los pacientes. Conclusiones: La paratiroidectomía enfocada y la hemitiroidectomía realizada por cirujanos expertos en pacientes seleccionados, es segura y efectiva en régimen ambulatorio. Es posible mejorar el índice de sustitución ambulatorio aumentando la tasa de aplicabilidad y aceptabilidad.

Objetive: Ambulatory or overnight stay surgery have been consolidated in many different procedures. However, its use in thyroid and parathyroid surgery is still controversial. The aim of this report is to present the results of 6 years of ambulatory patients undergoing surgery of the thyroid or parathyroid glands. Material and Methods: Prospective observational study of patients who underwent hemithyroidectomy or selective parathyroidectomy in the ambulatory program from January 2011 to December 2016. All patients included met the general criteria of ambulatory surgery. During the first years nodules bigger than 3 cm and patients classified as ASA III were excluded. Patients arrive at hospital the morning of surgery. After the operation, patients pass to the post-anesthesia care unit and then to the hospitalization room. 8 hours after surgery patients are discharged home if they meet the criteria. Results: 270 patients were operated, 159 of them met the inclusion criteria. The overall applicability rate was 59%. The acceptance rate was 83.6% and the substitution index was 49.2%. The unwanted hospital admission was 10.4% for the parathyroidectomy and 17.6% for the hemithyroidectomy. Any patient presented major complications at home. The satisfaction rate was high or very high for 94% of the patients. Conclusion: Selective parathyroidectomy and hemithyroidectomy performed by experienced surgeons in selected patients can be safely and effectively carried out in ambulatory surgery (outpatient). It would be possible to improve the substitution index by increasing the application and acceptability ratios.

Paratiroidectomía transoral endoscópica por acceso vestibular oral: cirugía de paratiroides sin cicatriz: primer caso realizado en Sudamérica / Transoral endoscopic parathyroidectomy vestibular approach (TOEPVA)

Introducción: El abordaje de elección de la glándula tiroides y paratiroides, desde su descripción, ha sido la cervicotomía transversal. En el hiperparatiroidismo, con la optimización de los métodos de localización preoperatoria de las glándulas anómalas, se han creado técnicas abiertas mínimamente invasivas, con un mejor resultado estético y un menor tiempo de recuperación. Con el objetivo de eliminar las cicatrices externas se ha implementado una nueva técnica quirúrgica que utiliza abordajes endoscópicos a través de orificios naturales (NOTES). En los últimos años se ha hecho conocida una técnica de tiroidectomía y paratiroidectomía transoral endoscópica a través de incisiones en el vestíbulo oral. Caso clínico: Paciente de 65 años con hiperparatiroidismo primario con nódulo hiperfuncionante superior derecho que se somete a una paratiroidectomía parcial transoral endoscópica por abordaje vestibular. Resultados: El procedimiento se realiza sin incidentes con buena evolución posoperatoria de la paciente. Discusión: La paratiroidectomía transoral endoscópica por abordaje vestibular (TOEPVA) ha demostrado ser una técnica bastante segura, con resultados similares a los de la técnica abierta, pero sin dejar cicatrices visibles.

Introduction: The approach of choice of the thyroid and parathyroid glands, since its description, has been the transverse cervicotomy. In hyperparathyroidism, with the optimization of the preoperative localization methods of the anomalous glands, minimally invasive open techniques have been created, with a better aesthetic result and a shorter recovery time. With the aim of eliminating external scars, a new surgical technique has been implemented that uses endoscopic approaches through natural orifices (NOTES). In recent years, a transoral endoscopic thyroidectomy and parathyroidectomy technique through incisions in the oral vestibule has been known. Clinical case: A 65-year-old patient with primary hyperparathyroidism with right upper hyperfunctioning nodule who underwent partial transoral endoscopic parathyroidectomy due to vestibular approach. Results: The procedure is performed without incidents with good postoperative evolution of the patient. Discussion: Transoral endoscopic parathyroidectomy by vestibular approach (TOEPVA) has proven to be a fairly safe technique, with results similar to open technique, but without leaving visible scars.

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.

Hiperparatiroidismo primario en adolescente. Reporte de caso / Primary hyperparathyroidism in adolescent. Case report

INTRODUCTION: The hypercalcemia is infrequent in pediatrics, its clinical is diverse, and its etiology is determined by age. Among the dependent causes of parathormone (PHT) is the hyperthyroidism, state of hypersecretion of PHT by parathyroid glands (PG). The primary hyperparathyroidism (PHPT) is rare in children, there are 200 reported cases. In older children, the causes for PHPT correspond to parathyroid adenoma, multiglandular disease and parathyroid carcinoma. OBJECTIVE: Report a case of an 11 years old male adolescent. He presents three months symptoms of constipation, anorexia, vomiting and weight loss. Urgent consultation due to an increase of his symptoms, in exams stand out: calcemia 16.67 mg/dl (NV 8.8-10.8 mg/dl), phosphatemia 2.21 mg/dl (NV 4.5-5.5 mg/dl), parathormone (PHT) 308.7 pg/ml (NV 15-68.3 pg/ml), calciuria/creatininuria 0.56 (NV < 0.2). He was hospitalized to manage his severe hypercalcemia, it was indicated hyperhydration, monopotassium phosphate, intravenous hydrocortisone and furosemide. In his study was performed a cervical ultrasound which showed a solid node in the right parathyroid gland, hypoechogenic and scintigram parathyroid compatible with right superior parathyroid adenoma. In the waiting for surgery was necessary the administration of intravenous pamidronate. In the post-operatory, he evolved with hipocalcemia that was corrected with intravenous calcium carbonate, overlapping to oral calcium and calcitriol. CONCLUSION: The PHPT is a pathology of low prevalence in pediatrics, one hundred times less than adults. In the adolescent, the most frequent cause is the parathyroid adenoma. It should be considered as a differential diagnosis in cases of symptomatic hypercalcemia in this age group

Resultados del tratamiento quirúrgico del hiperparatiroidismo primario en un país en vías de desarrollo / Outcomes of Primary Hyperparathyroidism Surgical Treatment in a Low-Middle Income Country

Introducción: El hiperparatiroidismo primario es una enfermedad común y con una distribución similar en todo el mundo. El propósito del estudio fue establecer si la presentación clínica y bioquímica, así como los resultados de su tratamiento quirúrgico, difieren en un país en vías de desarrollo, que no cuenta con todos los recursos diagnósticos y terapéuticos recomendados actualmente. Metodología: Análisis retrospectivo de pacientes operados por un mismo equipo, entre 1992 y 2015. Se obtuvo información sobre presentación clínica, resultados de estudios preoperatorios, procedimientos quirúrgicos, diagnóstico histopatológico y evolución postoperatoria. Resultados: Se operaron 55 pacientes con edad promedio de 45 años, 78% mujeres. El 65% eran sintomátcos. El valor promedio preoperatorio de calcio sérico fue 11.2 mg/dl, PTH 167.1 pg/ml, fósforo 2.6 mg/ dl, 25-hidroxi vitamina D 17.3 ng/ml y calcio urinario de 24 horas 294.7 mg. Al 59% se realizó estudios de localización preoperatoria. La sensibilidad del ultrasonido fue 57.14% y 75% para centellografa con tecnecio sestamibi. La positividad de los estudios de localización determinó el tipo de exploración quirúrgica (p=0.02). Se practcaron 27 (49%) exploraciones unilaterales y 28 (51%) bilaterales, resecando 47 (85.5%) adenomas solitarios y 3 ½ glándulas en 7 (12.7%) casos de hiperplasias. En 27 (36%) coexista patología tiroidea. Las tasas de curación, persistencia y recurrencia fueron 94.5%, 5.5% y 3.6% respectivamente. Conclusiones: La mayoría de nuestros pacientes operados son jóvenes y sintomátcos. La estrategia quirúrgica fue condicionada por los estudios de localización. Nuestras tasas de curación, persistencia y recurrencia son comparables a las reportadas.

Background: Primary hyperparathyroidism (HPTP) is a common disease with widespread distribution around the world. The aim of this study was to establish if clinical and biochemical disease characteristics and long term results differ in patents with HPTP in a low-middle income country without all recommended diagnostc and therapeutc resources. Methods: Retrospective collection of clinical diagnosis, biochemical, operative details, histology and long term results of all surgically treated patents with HPTP, from 1992 to 2015, by the same surgical team. Results: 55 patents with HPTP were analyzed. Average age is 45 years old with 78% of female patents. Sixty five percent were symptomatic. The mean preoperative serum calcium level was 11.2 mg/dl, PTH 167.1 pg/ml, phosphorus 2.6 mg/ dl, vitamin D 17.3 ng/ml and 24 hour urinary calcium 294.7 mg. Fifty nine percent of the patents had preoperative imaging. Ultrasound and sestamibi scan sensitivity was 57.1% and 75% respectively. Unilateral localization in preoperative imaging determined surgical exploration (p=0.02). Unilateral approach was used in 27 (49%) patents and bilateral in 28 (51%); 47 (85.5%) solitary adenomas and 7 (12.7%) 3 ½ gland resections of hyperplastic glands were performed. Thyroid pathology co-existed in 27 (36%) patients. Cure, persistence and recurrence rates were 94.5%, 5.5% y 3.6% respectively. Conclusions: In this study most of the patents were young and symptomatc. Surgical strategy was determined by preoperatve imaging. Cure, persistence and recurrence rates were comparable to published literature.

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)

Risk factors for intraoperative hypertension during surgery for primary hyperparathyroidism

Objective:To investigate the incidence and identify risk factors for the occurrence of intraoperative hypertension [IOH] during surgery for primary hyperparathyroidism [pHPT]

Subjects and Methods:The study included 269 patients surgically treated between January 2008 and January 2012 for pHPT. IOH was defined as an increase in systolic blood pressure >20% compared to baseline values which lasted for 15 min. The investigated influence were demographic characteristics, surgical risk score related to physical status [based on the American Society of Anesthesiologists [ASA] classification], comorbidities, type and duration of surgery, and duration of anesthesia on IOH occurrence. The investigated factors were obtained from the patients' medical history, anesthesia charts, and the daily practice database. Logistic regression analysis was done to determine the predictors of IOH

Results: Of the 269 patients, 153 [56.9%] had IOH. Based on the univariate analysis, age, body mass index, ASA status, duration of anesthesia, and preoperative hypertension were risk factors for the occurrence of IOH. Multivariate analysis showed that independent predictors of IOH were a historyof hypertension [OR - 2.080, 95% Cl: 1.102-3.925, p = 0.024] and age [OR - 0.569,95% Cl: 0.360-0.901, p = 0.016]

Conclusion: In this study, a high percentage [56%] of the patients developed IOH during surgery for pHPT, which indicates that special attention should be paid to these patients, especially to the high-risk groups: older patients and those with a history of hypertension. Further, this study showed that ad-vanced age and hypertension as a coexisting disease prior to parathyroid surgery were independent risk factors for the Occurrence Of IOH

Hiperparatiroidismo primario: manejo quirúrgico / Primary hyperparathyroidism: surgical treatment

Background: Primary hyperparathyroidism (HPTP) is the most common cause of hypercalcaemia in the ambulatory setting. This condition affects between 0.1 -2 percent of the population. Surgery is the only curative treatment. Objective: The aim of the study is to present our 10-year surgical experience. Methods: Analytical-descriptive study. We included all patients diagnosed with HPTP treated with surgery in our hospital between June 2003 and June 2013. Results: 173 patients underwent surgery for HPTP. The mean age was 57.5 +/- 13.5 years. Asymptomatic in 98 cases (56.6 percent). Resection the abnormal parathyroid glands was performed in all cases with intraoperative PTH monitoring. In 167 patients (96.5 percent) intraoperative PTH declined ≥ 50 percent. The median follow-up was 61 months (1-117 months). Cure was achieved in 169 patients (97.7 percent). Four cases (2.3 percent) had recurrence. Conclusions: Surgery is effective and safe for the treatment of HPTP. A decline in intraoperative PTH > 50 percent predicts cure in 97.7 percent of cases.

Introducción: El hiperparatiroidismo primario (HPTP), es una entidad frecuente que afecta entre el 0,1 y 2 por ciento de la población. La cirugía es el único tratamiento curativo. Objetivo: Evaluar la eficacia y riesgos del tratamiento quirúrgico del HPTP. Material y Métodos: Estudio analítico-descriptivo. Se incluyó a todos los pacientes con diagnóstico de HPTP referidos para cirugía al Hospital Clínico de la Pontificia Universidad Católica de Chile entre junio de 2003 y junio de 2013. Resultados: 173 pacientes fueron operados en nuestra institución por HPTP. El promedio de edad de los pacientes fue de 57,5 +/- 13,5 años. La forma de presentación fue asintomática en 98 casos (56,6 por ciento) y sintomática en 75 casos (43,4 por ciento). Todos los pacientes fueron tratados con resección de la o las glándulas hiperfuncionantes con medición de PTH intraoperatoria. En 167 pacientes (96,5 por ciento) se logró caída de PTH intraoperatoria > 50 por ciento. Catorce pacientes (8,1 por ciento) presentaron complicaciones post operatorias. La mediana de hospitalización fue de 2 días (1 a 23 días). La mediana de seguimiento fue de 61 meses (6 a 117 meses). Se logró curación en 169 pacientes (97,7 por ciento). Cuatro casos (2,3 por ciento) presentaron recidiva. Conclusiones: La cirugía es eficaz y segura para el tratamiento del hiperparatiroidismo primario. Una caída de PTH intraoperatoria > 50 por ciento del valor basal predice curación en 97,7 por ciento de los casos.