Characterization of a cohort of patients with hypercalcemia in a tertiary pediatric hospital. / Caracterización de una cohorte de pacientes con hipercalcemia en un centro pediátrico de tercer nivel.

Introduction. Hypercalcemia is infrequent in pediatrics, of diverse etiology, and with multiorgan morbidity. Objective. Describe the etiology, biochemistry, clinical, and treatment in pediatric patients with hypercalcemia. Population and methods. Retrospective and descriptive study of a cohort of patients with hypercalcemia between 2008 and 2022. They were classified into three groups (G): hypercalcemia of iatrogenic cause (G1), parathyroid hormone (PTH) independent (G2), or PTH-dependent (G3). Results. One hundred forty-seven patients were included; 57% were male, with a median age of 3.7 years, median calcemia of 11.8 mg/dl, and mean phosphatemia of 4.9 mg/dl. Symptoms were present in 29% of patients, and 28.6% required additional treatments to those of the first line. In G1, 76 patients (51.7%) were included; in G2, 58 (39.4%), and in G3, 13 (8.8%). Median calcemia was lower in G1 vs. G2 and G3 (11.6 mg/dl, 12.6 mg/dl, and 12.3 mg/dl), and mean phosphatemia was lower in G3 vs. G1 and G2 (3.7 mg/dl, 5.3 mg/dl, and 4.9 mg/dl). Most of the patients with hypercalcemia were asymptomatic and did not require additional treatments. The percentage of symptomatic patients and the percentage requiring additional treatment were lower in G1 than in the other two groups. Conclusions. Iatrogenesis was the most frequent cause, presenting lower calcemia, while PTH-dependent causes presented the lowest phosphatemia. PTH-independent causes represented a diagnostic and therapeutic challenge due to lacking a characteristic biochemical profile.

Introducción. La hipercalcemia es infrecuente en pediatría, de etiología diversa y con morbilidad multiorgánica. Objetivo. Describir etiología, bioquímica, clínica y tratamiento en pacientes pediátricos con hipercalcemia. Población y métodos. Estudio retrospectivo y descriptivo de una cohorte de pacientes con hipercalcemia entre 2008 y 2022. Se clasificaron en tres grupos (G): hipercalcemia de causa iatrogénica (G1), paratohormona (PTH) independiente (G2) o PTH dependiente (G3). Resultados. Se incluyeron 147 pacientes; el 57 % eran varones, edad mediana de 3,7 años, calcemia mediana 11,8 mg/dl y fosfatemia media 4,9 mg/dl. El 29,9 % de los pacientes fueron sintomáticos y el 28,6 % requirió tratamientos adicionales a los de la primera línea. En G1 se incluyeron 76 pacientes (51,7 %); en G2, 58 (39,4 %), y en G3, 13 (8,8 %). La calcemia mediana fue menor en G1 vs. G2 y G3 (11,6 mg/dl, 12,6 mg/dl y 12,3 mg/dl). La fosfatemia media fue menor en G3 vs. G1 y G2 (3,7 mg/dl, 5,3 mg/dl y 4,9 mg/dl). La mayoría de los pacientes con hipercalcemia fueron asintomáticos sin requerimientos de tratamientos adicionales. El porcentaje de pacientes sintomáticos y el de requerimiento de tratamientos adicionales fue menor en G1 que en los otros dos grupos. Conclusiones. La iatrogenia fue la causa más frecuente, y se presentó con calcemias más bajas; mientras que las causas PTH dependientes presentaron las fosfatemias más bajas. Las causas PTH independientes representaron un desafío diagnóstico y terapéutico por la falta de un perfil bioquímico característico.

Life-threatening hypercalcemia in a child with vitamin D intoxication due to parental self-medication: A case report.

Vitamin D is essential for bone metabolism and has gained popularity since the general population is now more aware of its benefits. Unfortunately, the availability of unregulated vitamin D formulations without prescription increases the risk of inadvertently ingesting excessive doses of vitamin D. Reports of pediatric cases of vitamin D toxicity are scarce in the world literature. We present the case of a 4-years 9-months old boy from a rural town with vitamin D intoxication secondary to ingestion of seven oral vials containing each of them 600,000 Units of cholecalciferol for a period of 8 months. It is important to educate general population about the risks of ingesting vitamin D without medical prescription. In our patient, the most effective treatment strategy was the use of pamidronate.

Primary hyperparathyroidism in pregnancy: a case report highlighting uncommon complication / Hiperparatiroidismo primario en el embarazo: reporte de caso destacando una complicación infrecuente

Abstract Primary hyperparathyroidism (PHPT) is characterized by elevated levels of calcium and parathyroid hormone (PTH). However, the interpretation of diagnostic tests, such as serum calcium and PTH levels, is complex in pregnant women. The aim of this report is to present a case of PHTP in a pregnant adolescent, with a special emphasis on an uncommon complication, as well as diagnostic and treatment strategies. A 17-year-old pregnant female presented with hyper emesis gravidarum and neurological symptoms, leading to the diagnosis of cerebral venous thrombosis. Further investigations revealed hypercalcemia and persistently elevated PTH levels, consistent with PHPT. After local ization studies, the patient underwent an emergency parathyroidectomy with a diagnosis of parathyroid ad enoma. During follow-up, intrauterine growth restric tion and severe preeclampsia developed, necessitating an emergency cesarean section. Both the mother and neonate had favorable outcomes. PHPT is an infrequent condition in the pregnant population, and its diagnosis can be challenging due to the overlap of symptoms with normal physiological changes during pregnancy. The occurrence of uncom mon complications, such as thrombotic phenomena, highlights the need for a comprehensive approach to ensure early detection and management. In most cases, parathyroidectomy is the treatment of choice.

Resumen El hiperparatiroidismo primario (HPTP) se caracteriza por niveles elevados de calcio y hormona paratiroidea (PTH). Sin embargo, la interpretación de pruebas diag nósticas, como los niveles de calcio sérico y PTH, es compleja en mujeres embarazadas. El objetivo de este re porte es presentar un caso de HPTP en una adolescente embarazada, con especial hincapié en una complicación infrecuente, así como en las estrategias diagnósticas y de tratamiento. Una mujer embarazada de 17 años presentó hiperé mesis gravídica y síntomas neurológicos, lo que llevó al diagnóstico de trombosis venosa cerebral. Posterio res investigaciones revelaron hipercalcemia y niveles persistentemente elevados de PTH, consistentes con HPTP. Tras la realización de estudios de localización, la paciente fue sometida a una paratiroidectomía de emergencia con diagnóstico de adenoma de paratiroi des. Durante el seguimiento, se desarrolló restricción del crecimiento intrauterino y preeclampsia grave, lo que resultó en la necesidad de realizar una cesárea de emergencia. Tanto la madre como el neonato evolucio naron favorablemente. El HPTP es una condición infrecuente en la población embarazada y su diagnóstico puede ser desafiante por la superposición de síntomas con los cambios fisiológicos normales del embarazo. La aparición de complicaciones infrecuentes, como fenómenos trombóticos, resalta la necesidad de un abordaje integral para garantizar la detección y el manejo temprano. En la mayoría de los casos, la paratiroidectomía es el tratamiento de elección.

ABCD syndrome, an uncommon cause of hypercalcemia in pediatrics. / Síndrome ABCD, una causa infrecuente de hipercalcemia en pediatría.

ABCD syndrome (ABnormal Calcium, Calcinosis, and Creatinine in Down syndrome) is characterized by an association of hypercalcemia, hypercalciuria, nephrocalcinosis, and impaired kidney function in patients with Down syndrome. Only 7 cases have been published worldwide, although it is believed to be underdiagnosed. This report describes 2 new patients with ABCD syndrome and compares them with the cases reported to date. Although it is a rare cause of pediatric hypercalcemia, it should be considered in children with Down syndrome once other more common etiologies have been ruled out. Once this diagnosis is confirmed, the recommended treatment is to reduce dietary calcium intake and work with an interdisciplinary team to maintain an adequate calorie and protein intake.

El síndrome ABCD (por sus siglas en inglés, ABnormal Calcium, Calcinosis and Creatinine in Down syndrome) se caracteriza por la asociación de hipercalcemia, hipercalciuria, nefrocalcinosis y alteración de la función renal en pacientes con síndrome de Down. Existen solo 7 casos previamente publicados en el mundo, aunque se cree que está subdiagnosticado. En este reporte, presentamos 2 nuevos pacientes con este síndrome y realizamos una comparación con los casos informados hasta el momento. Si bien es una causa rara de hipercalcemia pediátrica, debe considerarse en niños con síndrome de Down una vez descartadas otras etiologías más frecuentes. Al confirmarse este diagnóstico, el tratamiento recomendado es la reducción de calcio en la dieta, trabajando de manera interdisciplinaria para mantener un aporte calórico proteico adecuado.

Primary hyperparathyroidism in pregnancy: a case report highlighting uncommon complication.

Primary hyperparathyroidism (PHPT) is characterized by elevated levels of calcium and parathyroid hormone (PTH). However, the interpretation of diagnostic tests, such as serum calcium and PTH levels, is complex in pregnant women. The aim of this report is to present a case of PHTP in a pregnant adolescent, with a special emphasis on an uncommon complication, as well as diagnostic and treatment strategies. A 17-year-old pregnant female presented with hyperemesis gravidarum and neurological symptoms, leading to the diagnosis of cerebral venous thrombosis. Further investigations revealed hypercalcemia and persistently elevated PTH levels, consistent with PHPT. After localization studies, the patient underwent an emergency parathyroidectomy with a diagnosis of parathyroid adenoma. During follow-up, intrauterine growth restriction and severe preeclampsia developed, necessitating an emergency cesarean section. Both the mother and neonate had favorable outcomes. PHPT is an infrequent condition in the pregnant population, and its diagnosis can be challenging due to the overlap of symptoms with normal physiological changes during pregnancy. The occurrence of uncommon complications, such as thrombotic phenomena, highlights the need for a comprehensive approach to ensure early detection and management. In most cases, parathyroidectomy is the treatment of choice.

El hiperparatiroidismo primario (HPTP) se caracteriza por niveles elevados de calcio y hormona paratiroidea (PTH). Sin embargo, la interpretación de pruebas diagnósticas, como los niveles de calcio sérico y PTH, es compleja en mujeres embarazadas. El objetivo de este reporte es presentar un caso de HPTP en una adolescente embarazada, con especial hincapié en una complicación infrecuente, así como en las estrategias diagnósticas y de tratamiento. Una mujer embarazada de 17 años presentó hiperémesis gravídica y síntomas neurológicos, lo que llevó al diagnóstico de trombosis venosa cerebral. Posteriores investigaciones revelaron hipercalcemia y niveles persistentemente elevados de PTH, consistentes con HPTP. Tras la realización de estudios de localización, la paciente fue sometida a una paratiroidectomía de emergencia con diagnóstico de adenoma de paratiroides. Durante el seguimiento, se desarrolló restricción del crecimiento intrauterino y preeclampsia grave, lo que resultó en la necesidad de realizar una cesárea de emergencia. Tanto la madre como el neonato evolucionaron favorablemente. El HPTP es una condición infrecuente en la población embarazada y su diagnóstico puede ser desafiante por la superposición de síntomas con los cambios fisiológicos normales del embarazo. La aparición de complicaciones infrecuentes, como fenómenos trombóticos, resalta la necesidad de un abordaje integral para garantizar la detección y el manejo temprano. En la mayoría de los casos, la paratiroidectomía es el tratamiento de elección.

Acute kidney injury and hypercalcemia associated with veterinary supplements applications in adult man / Injúria renal aguda e hipercalcemia associadas a aplicações de suplementos veterinários em homem adulto

The compound "ADE" is an injectable oil for veterinary use which contains large amounts of vitamins A, D and E. The parenteral application in humans leads to a granuloma reaction which triggers hypercalcemia. A 42-year-old man was admitted with lower limb pain, nephrolithiasis and nephrocalcinosis. Laboratory tests revealed creatinine 4.59 mg/dl, calcium 13.3 mg/dl and parathormone 13.8 pg/ml. He underwent an ureterolithotripsy, stent placement, intravenous crystalloid fluids, and corticosteroid. He improved symptoms, kidney function and normalized serum calcium. The "ADE"-induced hypercalcemia diagnosis can be challenging. The early diagnosis may avoid negative outcomes.

O composto "ADE'' é um óleo veterinário injetável que contém grandes quantidades de vitaminas A, D e E. A aplicação parenteral causa reação granulomatosa e hipercalcemia. Um homem de 42 anos foi admitido com dor no membro inferior, nódulos musculares endurecidos, nefrolitíase e nefrocalcinose. O laboratório revelou creatinina 4,59 mg/dl, cálcio 13,3 mg/dl e paratormônio 13,8 pg/ml. Foi tratado com ureterolitotripsia, cateter duplo-J, cristaloide intravenoso e corticoterapia. Ele apresentou melhora dos sintomas, função renal e normalizou cálcio. O diagnóstico da hipercalcemia pelo "ADE'' pode ser desafiador. O diagnóstico precoce pode evitar desfechos negativos.

Silicone-induced granuloma, hypercalcemia and chronic kidney disease in a transgender patient / Granuloma induzido por silicone, hipercalcemia e doença renal crônica em um paciente transgênero

Background: hypercalcemia mediated by a silicone-induced granulomatous reaction is a condition that, despite being rare, must be remembered since there is an increasing demand for aesthetic procedures among the general population. It is necessary to rule out other causes of hypercalcemia, such as hyperparathyroidism, hypercalcemia of malignancy, infectious and non-infectious granulomatous diseases, as well as the use of drugs. Case presentation: We present a patient with a history of large amounts of silicone implants who was admitted to a hospital presenting hypercalcemia and chronic kidney disease. A skin biopsy revealed a granulomatous foreign body reaction associated with exogenous siliconomatous material. Treatment with corticosteroids was initiated, with partial renal function improvement, with no renal replacement therapy needed. Conclusion: Patients with silicone implants should be carefully monitored for renal function and may be informed to avoid nephrotoxic agents as there is sufficient evidence that this type of aesthetic procedure can trigger hypercalcemia and CKD.

Introdução: a hipercalcemia mediada por uma reação granulomatosa induzida por silicone é uma condição que, apesar de rara, deve ser lembrada, pois há uma demanda crescente por procedimentos estéticos na população em geral. É necessário descartar outras causas de hipercalcemia, como hiperparatireoidismo, hipercalcemia da malignidade, doenças granulomatosas infecciosas e não infecciosas, bem como o uso de medicamentos. Relato de caso: apresentamos um paciente com histórico implante de grandes quantidades de silicone, que foi admitido em um hospital apresentando hipercalcemia e doença renal crônica (DRC). A biópsia de pele revelou reação granulomatosa tipo corpo estranho associada a material siliconomatoso exógeno. Foi iniciado tratamento com corticosteroides, com melhora parcial da função renal, sem necessidade de terapia renal substitutiva. Conclusão: os pacientes com implantes de silicone devem ser cuidadosamente monitorados quanto à função renal e devem ser informados para evitar agentes nefrotóxicos, pois há evidências suficientes de que esse tipo de procedimento estético pode desencadear hipercalcemia e DRC.

Mucormycosis-induced hypercalcemia: a case report.

Hypercalcemia is a vital laboratory marker because it can show underlying severe diseases like cancer and infections. Of all the causes of hypercalcemia, primary hyperparathyroidism, and malignancies are the most common, but granulomatous diseases, such as certain fungal infections, can also be the cause. Here we describe the case of a 29-year-old woman, an insulin-dependent diabetic, found unconscious and tachypneic at home. In the emergency room, the medical team diagnosed diabetic ketoacidosis (DKA) and acute kidney injury (AKI). During hospitalization, despite resolving acidemia, persistent hypercalcemia attracted attention. Laboratory tests showed decreased parathyroid hormone (PTH) levels, confirming non-PTH-dependent hypercalcemia. Computed tomography (CT) of the chest and abdomen demonstrated no alterations, but an upper digestive endoscopy revealed an ulcerated and infiltrative lesion in the stomach. A biopsy showed a granulomatous infiltrate due to mucormycosis infection. The patient received liposomal amphotericin B for 30 days and isavuconazonium for two months. Serum calcium levels improved during treatment. Inquiry of the etiology of hypercalcemia should begin with the PTH assay; high levels are consistent with hyperparathyroidism; low levels, with calcium or vitamin D intoxication, malignancies, prolonged immobilization, and granulomatous diseases. In the latter cases, the overproduction of 1-alpha-hydroxylase by the granulomatous tissue increases the conversion of 25(OH)vitamin D into 1-25(OH)vitamin D, which causes the intestinal absorption of calcium. We have described the first hypercalcemia related to mucormycosis infection in a young diabetic patient, although case presentations associate other fungal infections with elevated serum calcium.

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)

Bilateral giant parathyroid adenoma and hungry bone syndrome: a case report.

BACKGROUND: There is some evidence supporting the idea that double parathyroid adenomas represent a different entity from multiglandular hyperplasia; however, the distinction among them is not straightforward. CASE PRESENTATION: We described a case of primary hyperparathyroidism (PHPT) with pronounced clinical manifestations, caused by a bilateral giant parathyroid adenoma. A 34-year-old Hispanic/Latino male was diagnosed with PHPT caused by two giant parathyroid adenomas. The preoperative tests were neck ultrasound and computed tomography scan (CT-scan), showing two masses in the territory of parathyroid glands, bilaterally distributed (right was 31 × 18 × 19 mm and the left was 38 × 15 × 14 mm); sestamibi scan was not available. Parathyroid hormone (PTH) was highly elevated. Multiple complications of PHPT were present, such as bone lytic lesions, renal and pancreatic calcifications, and cardiovascular disease, the latter of which is an overlooked complication of PHPT. Multiple endocrine neoplasia 1 and 2 (MEN 1/2) were ruled out by the absence of clinical, biochemical, and radiological findings in other endocrine glands. The patient underwent subtotal parathyroidectomy with an intraoperative histopathological study; both intraoperative and definitive histopathology results were consistent with parathyroid adenomas; afterward, adequate suppression of PTH was assured, and later on, the patient presented hungry bone syndrome (HBS). CONCLUSIONS: The diagnosis of double parathyroid adenomas is difficult. Regarding the similarities between multiglandular hyperplasia and parathyroid adenomas, this case report contributes to the further distinction between these two clinical entities. This case report also represents, in particular, the challenge of difficult diagnosis in places with limited resources, such as developing countries.

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.

Ectopic parathyroid adenoma causing hyperparathyroidism-induced psychosis: A case report.

Primary hyperparathyroidism is a disease with multisystemic and heterogeneous manifestations, characterized by underlying high parathormone concentrations. Despite neuropsychiatric involvement being one of the manifestations, psychosis is rare. This is the case of a 68-year-old female with a 10-day clinical course of anorexia, mutism, dysphagia, constipation, and weight loss. The patient had disorganized speech associated with paranoid delusions. Prior to this visit, the patient was recently diagnosed with a mixed anxiety-depressive disorder. For this reason, treatment with antidepressants in combination with atypical antipsychotics was administered without a satisfactory response. Neuroimaging, infectious panel, and toxicology screening showed no abnormal findings. Hypercalcemia secondary to a retropharyngeal ectopic parathyroid adenoma was the causative etiology of her primary hyperparathyroidism, and hypercalcemia treatment resolved the psychotic episode. We highlight the importance of recognizing psychosis as a possible initial presentation of hyperparathyroidism and hypercalcemia. Ruling out organic etiologies prior to diagnosing a primary cause of psychosis is crucial, as their treatment can reverse the psychotic symptoms.

Planned parathyroidectomy: the new standard in hypercalcemic crisis.

Objective: The study sought to determine the clinical features of hyperparathyroid-induced hypercalcemic crisis (HIHC) along with treatment options and outcomes. Subjects and methods: This is a retrospective analysis of our historical cohort of patients with primary hyperparathyroidism (PHPT). Patients were divided in groups according to their calcium levels and clinical presentation. HIHC (group 1) was assumed when patients had high calcium levels and needed emergency hospitalization. Group 2 was composed of patients with calcium levels above 16 mg/dL or patients who needed hospitalization for classical PHPT symptoms. Group 3 was composed of clinically stable patients with calcium levels between 14 and 16 mg/dL, who were electively treated. Results: Twenty-nine patients had calcium levels above 14 mg/dL. HIHC group had seven patients, and initial clinical measures had good response in two patients, moderate response in one patient, and poor response in four patients. All poor responders underwent immediate surgery, and one of them died due to HIHC complications. Group 2 had nine patients, and all were successfully treated during hospitalization. Group 3 had 13 patients, and all had a successful elective surgery. Conclusion: HIHC is a life-threatening condition that requires fast clinical intervention. Surgery is the only definitive treatment and should be planned for all patients. Poor response to initial clinical measures should direct treatment toward surgery to avoid disease progression and clinical deterioration.

Planned parathyroidectomy: the new standard in hypercalcemic crisis

ABSTRACT Objective: The study sought to determine the clinical features of hyperparathyroid-induced hypercalcemic crisis (HIHC) along with treatment options and outcomes. Subjects and methods: This is a retrospective analysis of our historical cohort of patients with primary hyperparathyroidism (PHPT). Patients were divided in groups according to their calcium levels and clinical presentation. HIHC (group 1) was assumed when patients had high calcium levels and needed emergency hospitalization. Group 2 was composed of patients with calcium levels above 16 mg/dL or patients who needed hospitalization for classical PHPT symptoms. Group 3 was composed of clinically stable patients with calcium levels between 14 and 16 mg/dL, who were electively treated. Results: Twenty-nine patients had calcium levels above 14 mg/dL. HIHC group had seven patients, and initial clinical measures had good response in two patients, moderate response in one patient, and poor response in four patients. All poor responders underwent immediate surgery, and one of them died due to HIHC complications. Group 2 had nine patients, and all were successfully treated during hospitalization. Group 3 had 13 patients, and all had a successful elective surgery. Conclusion: HIHC is a life-threatening condition that requires fast clinical intervention. Surgery is the only definitive treatment and should be planned for all patients. Poor response to initial clinical measures should direct treatment toward surgery to avoid disease progression and clinical deterioration.

Malignant hypercalcemia as the debut of acute lymphoblastic leukemia in a pediatric patient-a diagnostic and therapeutic approach: Case report.

Background: Hypercalcemia is a rare metabolic disorder in the pediatric population, with several differential diagnoses that resemble hematologic malignancies. In cases of severe hypercalcemia, therapeutic strategies other than hyperhydration, such as the use of bisphosphonates, have been described. Case presentation: We present the case of a previously healthy 12-year-old boy who was admitted to the emergency department due to fatigue, hypo-responsiveness, and progressively worsening poor appetite for the previous 19 days. Initial laboratory tests revealed severe hypercalcemia (total calcium: 19 mg/dl), hyperphosphatemia, elevated creatinine, and hyperuricemia. Management with hyperhydration and xanthine oxidase inhibitor (allopurinol) was provided. The patient was transferred to the pediatric intensive care unit where treatment with furosemide, systemic corticosteroid, and zoledronic acid was started. Metabolic, infectious, renal, and endocrinological causes were excluded. Follow-up paraclinical studies showed a progressive hematologic involvement with heterogeneous hypochromic microcytic anemia, thrombocytopenia, and elevated lactic dehydrogenase. Bone marrow aspiration and biopsy were performed, which confirmed the diagnosis of B-precursor acute lymphoblastic leukemia. Hypercalcemia was resolved 72 h after the application of bisphosphonates. Conclusion: Hypercalcemia as an oncological metabolic emergency in the onset of acute lymphoblastic leukemia is uncommon in children. The use of intravenous bisphosphonates is an effective therapy in the early resolution of the condition. We present the case of a 12-year-old patient with malignant hypercalcemia who responded favorably to the use of a single dose of bisphosphonates.

Malignant hypercalcemia at a national referral center. A retrospective analysis

Malignant hypercalcemia is the most frequent endocrinological emergency in cancer. The factors related to its occurrence have not been evaluated, nor are there any related studies in Colombia. Objective: to determine the factors associated with the onset of malignant hypercalcemia (MH) in patients hospitalized at Instituto Nacional de Cancerología (2014-2019). Design: a retrospective analytical case-control study Population: 230 cases of adults with cancer and calcium corrected for albumin equal to or greater than 10.5 mg/dL and 223 controls of cancer patients with normal corrected calcium (8.5 to 10.4 mg/ dL) were included. Squamous cell carcinoma was used as the pairing variable. Analysis: univariate and bivariate analyses between the variables and the occurrence of MH were conducted to determine association, and raw ORs were calculated. Depending on their statis tical significance (p<0.05), they were included in the logistic regression for multivariate analysis and to rule out bias. Results: the median calcium for cases: 12.98 mg/dL (11.64-14.42) vs. 9.4 mg/dL (9.02-9.74) (p < 0.0001). The two groups were similar in age and sex. There was more metastasis in the cases (60.0% vs. 39.91%) (p< 0.0001). Altogether, 54.34% of the cases developed neurological symp toms. The median hospital stay was 16 days (IQR 9-27) vs. 9 days (IQR 6-17) for the controls (p< 0.0001), and inpatient deaths occurred in 48.70% vs. 16.59% (p < 0.0001). The following showed an association with MH: normal albumin, OR 0.41 (95% CI 0.29-0.55); a Karnofsky Index greater than or equal to 70, OR 0.98 (95% CI 0.97-0.99); and metastasis, OR 1.87 (95% CI 1.23-2.84). (Acta Med Colomb 2022; 47. DOI:https://doi.org/10.36104/amc.2022.2555).

La hipercalcemia maligna es la urgencia endocrinológica en cáncer más frecuente, los factores relacionados a su presentación no han sido evaluados, ni hay estudios relacionados en Colombia. Objetivo: determinar los factores asociados a la presentación de hipercalcemia maligna (HM), en pacientes hospitalizados en el Instituto Nacional de Cancerología (2014-2019). Diseño: estudio analítico retrospectivo de casos y controles. Población: se incluyeron 230 casos de adultos con cáncer y calcio corregido por albúmina mayor e igual a 10.5 mg/dL y 223 controles de pacientes oncológicos con calcio corregido normal (8.5 a 10.4 mg/dL), se usó el carcinoma escamocelular como variable pareadora. Análisis: para establecer asociación se realizó el análisis univariado y bivariado entre las variables y la presentación de HM, se calcularon OR crudos, según significancia estadística (p<0.05) fueron incluidas en regresión logística para el análisis multivariado y excluir sesgos. Resultados: mediana de calcio en los casos: 12.98 mg/dL (11.64-14.42) vs. 9.4 mg/dL (9.02-9.74) (p < 0.0001). Los dos grupos presentaron edad y sexo similares. Hubo mayor proporción de metástasis en los casos (60.0% vs. 39.91%) (p < 0.0001). El 54.34% de los casos desarrolló síntomas neuro lógicos. La mediana de estancia hospitalaria fue de 16 días (RIC 9-27) vs. nueve días (RIC 6-17) para los controles (p < 0.0001) y la muerte intrahospitalaria se presentó en el 48.70% vs. 16.59% (p < 0.0001). Demostraron asociación con HM: albúmina normal OR 0.41 (IC 95% 0.29-0.55), índice de Karnofsky mayor o igual 70 OR 0.98 (IC95% 0.97-0.99) y presencia de metástasis OR 1.87 (IC 95% 1.23-2.84). (Acta Med Colomb 2022; 47. DOI:https://doi.org/10.36104/amc.2022.2555).

Medical management of primary hyperparathyroidism.

Primary hyperparathyroidism (PHPT) is an endocrine disorder resulting from the hyperfunction of one or more parathyroid glands, with hypersecretion of parathyroid hormone (PTH). It can be managed by parathyroidectomy (PTX) or non-surgically. Medical therapy with pharmacological agents is an alternative for those patients with asymptomatic PHPT who meet guidelines for surgery but are unable or unwilling to undergo PTX. In this review, we focus upon these non-surgical aspects of PHPT management. We emphasize the most studied and widely used pharmacological alternatives: bisphosphonates, denosumab, cinacalcet and hormone therapy, in addition to combined therapy. We also address the relevant aspects of perioperative management.

Medical management of primary hyperparathyroidism

ABSTRACT Primary hyperparathyroidism (PHPT) is an endocrine disorder resulting from the hyperfunction of one or more parathyroid glands, with hypersecretion of parathyroid hormone (PTH). It can be managed by parathyroidectomy (PTX) or non-surgically. Medical therapy with pharmacological agents is an alternative for those patients with asymptomatic PHPT who meet guidelines for surgery but are unable or unwilling to undergo PTX. In this review, we focus upon these non-surgical aspects of PHPT management. We emphasize the most studied and widely used pharmacological alternatives: bisphosphonates, denosumab, cinacalcet and hormone therapy, in addition to combined therapy. We also address the relevant aspects of perioperative management.

Manifestaciones craneofaciales en pacientes con osteodistrofia renal / Craneofacial manifestaciones in patients with renal osteodystrophy

Introducción: La osteodistrofia renal es una osteopatía metabólica difusa, relacionada con la insuficiencia renal crónica, que incluye diversas patologías en el sistema musculoesquelético. Se produce en respuesta a trastornos metabólicos generados por cambios electrolíticos, la inflamación crónica y la alteración hormonal. Estas variaciones modifican el proceso de remodelación ósea. Las manifestaciones clínicas incluyen alteración en el parénquima y estroma óseo, y van desde lesiones expansivas, produciendo deformidad, hasta fracturas patológicas del hueso. Objetivo: Relacionar las manifestaciones clínicas, imagenológicas e histológicas en el componente óseo cráneo facial para el diagnóstico de la osteodistrofia renal. Presentación de caso: Se presentan dos casos clínicos de pacientes con enfermedad renal crónica e hiperparatiroidismo secundario de base, con múltiples masas en maxilares que causan asimetría facial y alteración funcional. Las imágenes tomográficas revelan alteración en la morfología ósea cortical y trabecular. Ambos individuos evidenciaron alteraciones en niveles de hormona paratiroidea, fosfatasa alcalina, fósforo y calcio sérico. La histopatología comprobó tejido fibroóseo con hueso neoformado y gran vascularización, con células multinucleadas tipo osteoclastos sin presencia de hemosiderina. Pacientes manejados de forma interdisciplinaria entre medicina interna, endocrinología y cirugía maxilofacial. Conclusiones: Los pacientes con enfermedad renal crónica avanzada presentan alteración de la estructura y del metabolismo óseo y mineral. Tal situación puede comprometer el complejo óseo craneofacial. Los casos graves de osteodistrofia renal se caracterizan por una marcada expansión de los maxilares, que genera asimetría y rasgos de leontiasis. Las imágenes tomográficas asociadas a osteodistrofia renal presentan óseos trabeculares con expansión de cortical, que evidencia el recambio óseo inmaduro presente. La histopatología no es específica y puede ser similar a los casos de displasia ósea craneofacial. Ante lo anteriormente planteado es fundamental relacionar estos hallazgos con la clínica para definir un diagnóstico adecuado(AU)

(AU)Introduction: Renal osteodystrophy is a diffuse metabolic osteopathy, related to chronic renal failure, which includes various pathologies in the musculoskeletal system. It occurs in response to metabolic disorders generated by electrolyte changes, chronic inflammation and hormonal alteration. These variations modify the process of bone remodeling. Clinical manifestations include alteration in the parenchyma and bone stroma, and range from expansive lesions, producing deformity, to pathological fractures of the bone. Objective: Relate the clinical, imaging and histological manifestations in the skull-facial bone component for the diagnosis of renal osteodystrophy. Case Presentation: Two clinical cases of patients with chronic kidney disease and secondary underlying hyperparathyroidism are presented, with multiple masses in the jaws that cause facial asymmetry and functional alteration. Tomographic images reveal alteration in cortical and trabecular bone morphology. Both individuals showed alterations in levels of parathyroid hormone, alkaline phosphatase, phosphorus and serum calcium. Histopathology verified fibro-bone tissue with neoformed bone and great vascularization, with multinucleated osteoclast-like cells without the presence of hemosiderin. Patients were attended in an interdisciplinary way between internal medicine, endocrinology and maxillofacial surgery. Conclusions: Patients with advanced chronic kidney disease present alteration of the structure and bone and mineral metabolism. Such a situation can compromise the craniofacial bone complex. Severe cases of renal osteodystrophy are characterized by a marked expansion of the jaws, which generates asymmetry and traits of leonthiasis. The tomographic images associated with renal osteodystrophy present trabecular bones with cortical expansion, which evidences the immature bone turnover present. Histopathology is not specific and may be similar to cases of craniofacial bone dysplasia. Given the above, it is essential to relate these findings to the clinic to define an adequate diagnosis(AU)

Crisis hipercalcémica como causa de pancreatitis aguda / Hypercalcemia as a cause of acute pancreatitis. Report of one case

Severe hypercalcemia may cause acute pancreatitis. We report a 75-yearold male presenting with abdominal pain and confusion. The initial laboratory showed elevated amylase levels and a serum calcium of 19.0 mg/dl with highly elevated parathormone levels. An abdominal CT scan disclosed pancreatitis. A neck CT scan showed a parathyroid tumor, which was successfully excised. The pathology of the surgical piece showed a parathyroid adenoma.