Hypercalcemic crisis caused by a parathyroid adenoma with hemorrhage and cystic degeneration: A case report / 中华内分泌代谢杂志

This paper reported a rare case of hypercalcemic crisis caused by a parathyroid adenoma with hemorrhage and cystic degeneration. Preoperative imaging examination of the patient was unable to determine the histological origin of the cervical cystic lesion. Despite aggressive medical treatment and hemodialysis, hypercalcemic crisis could not be relieved. Therefore, surgical exploration and excision of the cervical lesion were performed, and final diagnosis of parathyroid adenoma with hemorrhage and cystic degeneration was confirmed by pathology. Blood calcium level and renal function returned to normal after the surgery.

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

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

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

Injuria renal aguda secundaria a crisis hipercalcémica: a propósito de un caso / Acute renal injury secondary to hypercalcemic crisis: a case report / Lesão ou injúria renal aguda secundária a crise hipercalcêmica: relatório de caso clínico

La injuria renal aguda es una entidad clínica compleja, caracterizada por la disminución abrupta de la función renal. La hipercalcemia como etiología de la misma es poco frecuente. Los mecanismos involucrados en su desarrollo son múltiples y poco estudiados. Se presenta el caso de un paciente varón de 59 años que desarrolló un cuadro severo de falla renal aguda como complicación de crisis hipercalcémica por un adenoma de paratiroides. Se observó alteración en los marcadores de daño y función renal. La bioquímica urinaria mostró una necrosis tubular aguda. Los niveles de calcio, parathormona y calciuria se asociaron a endocrinopatía. La ecografía, el centellograma y la biopsia paratiroidea mostraron la presencia de un adenoma. Se presentaron otras complicaciones sistémicas concomitantes como pancreatitis y complicaciones cardíacas. El tratamiento paliativo fue la hemodiálisis y el definitivo la paratiroidectomía. El síndrome de hueso hambriento se presentó como una complicación postquirúrgica. Tras el alta, la recuperación de la función renal nunca fue total. El daño renal agudo asociado a disfunción sistémica por hipercalcemia puede llevar a una recuperación parcial de la función renal. Se debe considerar el desarrollo de enfermedad renal crónica posterior a la falla renal aguda por hipercalcemia como complicación de la misma.

Acute renal injury is a complex clinical entity, characterized by the abrupt worsening in renal function. Hypercalcemia as its etiology is rare. The mechanisms involved in its development are multiple and rarely studied. The case of a 59-year-old male patient who developed a severe acute renal failure as a complication of an hypercalcemic crisis due to a parathyroid adenoma is presented here. Alterations in markers of damage and renal function were observed. Urinary biochemistry showed acute tubular necrosis. Calcium, parathormone and urine calcium levels were associated with endocrinopathy. The ultrasound, the scintigraphy and the parathyroid biopsy showed the presence of an adenoma. There were other concomitant systemic complications such as pancreatitis and cardiac complications. Hemodialysis was the palliative treatment, while the definitive treatment was parathyroidectomy. The hungry bone syndrome occurred as a postoperative complication. After discharge, recovery of renal function was never complete. Acute renal damage associated with systemic dysfunction due to hypercalcemia can lead to a partial recovery of renal function. The development of chronic kidney disease after acute renal failure due to hypercalcemia should be considered one of its complications.

A Lesão renal aguda é uma entidade clínica complexa, caracterizada pela diminuição abrupta da função renal. A hipercalcemia como etiologia da mesma não é muito frequente. Os mecanismos que participam no seu desenvolvimento são múltiplos e pouco estudados. Apresenta-se o caso de um paciente, homem, de 59 anos, que desenvolveu um quadro severo de insuficiência renal aguda como complicação de crise hipercalcêmica por um adenoma da paratireóide. Foi observada alteração nos marcadores de dano e função renal. A bioquímica urinária mostrou uma necrose tubular aguda. Os níveis de cálcio, paratormona e calciúria foram associados a endocrinopatia. A ultra-sonografia, a cintilografia, e a biópsia da paratireóide mostraram a presença de um adenoma. Apresentaram-se outras complicações sistêmicas concomitantes como pancreatite e cardíacas. O tratamento paliativo foi hemodiálise e o definitivo, a paratireoidectomia. A síndrome do osso faminto apresentou-se como uma complicação pós-operatória. Após a alta, a recuperação da função renal nunca foi total. O dano renal agudo associado à disfunção sistêmica por hipercalcemia pode levar para uma recuperação parcial da função renal. Deve ser considerado o desenvolvimento da doença renal crônica posterior à insuficiência renal aguda por hipercalcemia como complicação da mesma.

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)

Pancreatitis in patients with primary hyperparathyroidism / 中华内分泌外科杂志

Objective To investigate the clinical characters of pancreatitis in patients with primary hyperparathyroidism (PHPT).Methods The clinical data of patients with PHPT undergoing parathyroidectomy from Jan.2009 to May.2014 in Peking Union Medical College Hospital were retrospectively analyzed for coexistent pancreatitis.Results 571 patients received parathyroidectomy due to PHPT during this period.Thirteen patients (2.3％)with PHPT were confirmed with coexistent pancreatitis by clinical manifestation and abdominal imaging evaluation,including acute pancreatitis in 9 patients and chronic pancreatitis in 4 patients.PHPT with pancreatitis was associated with preoperative serum intact parathyroid hormone(iPTH)higher than 500 pg/ml (P=0.025)and occurrence of hypercalcemic crisis (P=0.013).The age and sex of patients,preoperative serum calcium,gallstones,urinary calculi and bone fracture were not related with pancreatitis in this group.The average follow-up period for these patients was 34(ranging from 13 to 68)months.All patients were free from hyperparathyroidism recurrence.Only one young lady with chronic pancreatitis suffered from repeated pancreatitis after surgery.Conclusions Serum iPTH higher than 500 pg/ml and occurrence of hypercalcemic crisis were possible risk factor of pancreatitis in patients with PHPT.Parathyroidectomy can help to prevent the recurrence of pancreatitis in these patients.

Functional parathyroid cystic adenoma: A rare cause of hypercalcemic crisis with primary hyperparathyroidism.

We discuss a case of primary hyperparathyroidism caused by a giant cystic parathyroid adenoma presenting with neck swelling and hypercalcemic crisis. Fine‑needle aspiration cytology of presumed thyroid swelling from one of the two sites aspirated yielded clear fluid but was not attributed to parathyroid pathology. Elevated serum calcium and intact parathormone (iPTH) levels suggested preoperative parathyroid pathology. Ultrasound neck and sestamibi scan for parathyroid localization were not conclusive. Due to resistant hypercalcemia, the patient underwent emergency bilateral neck exploration and excision of the identified left superior parathyroid cyst along with total thyroidectomy. Monitoring of intra‑operative iPTH helped complete removal of hyperfunctioning parathyroid tissue. Histopathological examination confirmed the parathyroid cyst. Cystic parathyroid adenoma should be considered in the differential diagnosis of cystic neck lesions.

Hypercalcemic Crisis – A Fatal Case of Primary Hyperparathyroidism

We describe the clinical presentation, investigation and management of an eventually fatal case of hypercalcemic crisis due to primary hyperparathyridism (PHPT). A 60 year-old lady with history of urolithiasis presented with worsening generalized bone pain, spinal scoliosis and a limp. Laboratory data showed hypercalcemia and raised alkaline phosphatase. Left hip x-ray revealed a subcapital femoral neck fracture. Intact parathyroid hormone was elevated, 187.6 pmol/L (1.6 – 6.9) and ultrasound showed an enlarged right parathyroid gland. Despite initial reduction of serum calcium with saline infusion and multiple doses of intravenous pamidronate, her calcium increased to 4.14 mmol/L a week following application of Buck’s traction for persistent left hip pain. She succumbed eventually with serum calcium peaking at 6.28 mmol/L despite multiple therapeutic interventions.

The efficacy of disodium Pamidronate in the treatment of primary hyperparathyroidism complicated by hypercalcemic crisis / 中华急诊医学杂志

Objective To study the efficacy of disodium pamidronate in the treatment of patients with primary hyperparathyroidism (PHPT) complicated by hypercalcemic crisis.Methods A total of 9 patients (12 cases) admitted into our hospital were diagnosed as PHPT complicated by hypercalcemic crisis.Of them,4 patients had parathyroid carcinoma,3 patients had parathyroid adenoma,2 patients had parathyroid hyperplasia.The intravenous disodium pamidronate was given for 12 times in all 9 patients.Serum calcium were monitored before and after treatment.Results Before the treatment,the serum calcium levels was 3.75 (3.66,3.99) mmol/L.On the second and third day after the treatment with disodium pamidronate,the serum calcium levels were 3.44 (2.95,3.78) mmol/L and 2.79 (2.50,3.14) mmol/L.The lowest level of serum calcium after treatment was 2.25 (2.00,2.55) mmoL/L.There were significant differences in the level of serum calcium of different days (P ＜ 0.05).On the second and third day,the change of serum calcium were 0.46 (0.10,0.88) mmol/L,0.60 (0.25,0.75) mmol/L,there were no differences.After intravenous disodium pamidronate,the serum calcium level decreased below 3.5 mmol/L in (2.27 ± 0.65) days,and were kept below 3.5mmol/L for 18.45 ± 12.30 days.Conclusions Disodium pamidronate can decrease serum calcium levels in hypercalcemic crisis caused by primary hyperparathyroidism effectively with mild adverse events.

Two Cases of Hyperparathyroidism Presenting as Acute Pancreatitis / 대한내분비학회지

The relationship between hypercalcemic crisis and pancreatitis, first described in patients with hyperparathyroidism, still remains controversial. Acute pancreatitis may complicate the clinical course of hyperparathyroidism, particularly when the degree of hypercalcemia is severe. The incidence of hyperparathyroidism presenting as acute pancreatitis appears to be steadily decreasing, possibly reflecting the earlier diagnosis of asymptomatic hyperparaparathyroidism due to widespread application of screening methods. Here, we report two patients with primary hyperparathyroidism manifesting clinically as acute pancreatitis. One patient died of progessive pancreatitis and uncontrolled sepsis. The other patient was fullly recovered by emergent parathyroidectomy followed by medical management of hypercalcemia.