RESUMEN
Tertiary hyperparathyroidism (THPT) is characterized by elevated parathyroid hormone and serum calcium levels after kidney transplantation (KTx). To ascertain whether pre-transplant calcimimetic use and dose information would improve THPT prediction accuracy, this retrospective cohort study evaluated patients who underwent KTx between 2010 and 2022. The primary outcome was the development of clinically relevant THPT. Logistic regression analysis was used to evaluate pre-transplant calcimimetic use as a determinant of THPT development. Participants were categorized into four groups according to calcimimetic dose, developing two THPT prediction models (with or without calcimimetic information). Continuous net reclassification improvement (CNRI) and integrated discrimination improvement (IDI) were calculated to assess ability to reclassify the degree of THPT risk by adding pre-transplant calcimimetic information. Of the 554 patients, 87 (15.7%) developed THPT, whereas 139 (25.1%) received pre-transplant calcimimetic treatment. Multivariate logistic regression analysis revealed that pre-transplant calcimimetic use was significantly associated with THPT development. Pre-transplant calcimimetic information significantly improved the predicted probability accuracy of THPT (CNRI and IDI were 0.91 [p < 0.001], and 0.09 [p < 0.001], respectively). The THPT prediction model including pre-transplant calcimimetic information as a predictive factor can contribute to the prevention and early treatment of THPT in the era of calcimimetics.
Asunto(s)
Calcimiméticos , Calcio , Trasplante de Riñón , Humanos , Trasplante de Riñón/efectos adversos , Estudios Retrospectivos , Masculino , Femenino , Persona de Mediana Edad , Calcimiméticos/uso terapéutico , Calcimiméticos/administración & dosificación , Adulto , Calcio/sangre , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/etiología , Hormona Paratiroidea/sangre , Modelos LogísticosRESUMEN
Colonic pseudo-obstruction, also called Ogilvie's syndrome, occurs due to impaired intestinal propulsion, and may be caused by electrolyte imbalances such as hypokalemia and some endocrine disorders such as hyperparathyroidism. Secretory diarrhea due to intestinal pseudo-obstruction can cause hypokalemia. Diuretics such as amiloride can be used to treat hypokalemia, however in this case, treatment with amiloride induced hypercalcemia and unmasked hyperparathyroidism. A 73-year-old female with a history of hypertension and parathyroid adenoma presented with recurrent colonic pseudo-obstruction and chronic hypokalemia. Her hypokalemia was treated with amiloride, causing hypercalcemia of 14.4 mg/dL, elevated PTH, and altered mental status. Amiloride was subsequently discontinued with improvement in her symptoms, and her hyperparathyroidism was treated with cinacalcet. To our knowledge, this is the first report of amiloride unmasking hyperparathyroidism and inducing hypercalcemia.
Asunto(s)
Seudoobstrucción Colónica , Hipercalcemia , Hiperparatiroidismo , Hipopotasemia , Femenino , Humanos , Anciano , Hipercalcemia/diagnóstico , Hipercalcemia/tratamiento farmacológico , Hipercalcemia/etiología , Hipopotasemia/complicaciones , Hipopotasemia/diagnóstico , Hipopotasemia/tratamiento farmacológico , Amilorida/uso terapéutico , Seudoobstrucción Colónica/complicaciones , Hiperparatiroidismo/complicaciones , Hiperparatiroidismo/diagnóstico , Hiperparatiroidismo/tratamiento farmacológicoRESUMEN
Proton pump inhibitors (PPIs) are associated with increased risk of osteoporotic fracture; however, the mechanism is unclear. PPI users taking calcium supplements were more likely to have hyperparathyroidism compared to non-users (OR 1.56, CI 1.08-2.23, p = 0.018). This highlights the importance of monitoring PPI use, especially in older adults. PURPOSE: Proton pump inhibitors (PPIs) are associated with increased risk of osteoporotic fracture. Hyperparathyroidism may be implicated, but few studies have considered this relationship. This study evaluated the relationship between PPI use and hyperparathyroidism in older adults. METHODS: Participants were from the TUDA study, a large cross-sectional cohort of older Irish adults. Participants with an estimated glomerular filtration rate (eGFR) < 30 ml/min and serum calcium > 2.5 mmol/l were excluded to avoid hyperparathyroidism due to chronic renal disease and primary hyperparathyroidism. Hyperparathyroidism was defined as a parathyroid hormone (PTH) > 65 pg/ml. Multivariate regression models were used to analyse the relationship between PPI use and hyperparathyroidism. RESULTS: A total of 4139 participants met the inclusion criteria, of whom 37.8% (n = 1563) were taking PPI medication. PPI use was identified in 41.4% of calcium supplement users and 35.4% of non-calcium supplement users. Overall, compared to non-users of PPIs, those taking PPIs were older (74.8 vs 72.9 years, p < 0.001) and had a higher prevalence of hyperparathyroidism (17.8 vs 11.0%, p < 0.001). In those taking calcium supplements (but not in non-users), PPI use was significantly associated with hyperparathyroidism (OR 1.56, CI 1.08-2.23, p = 0.018) after adjusting for age, sex, body mass index, serum vitamin D, eGFR, timed-up-and-go, dairy intake, medications, and comorbidities. DISCUSSION: The results are consistent with the hypothesis of PPIs reducing calcium absorption, leading to a rise in PTH which could mediate increased fracture risk. No relationship of PPI use with hyperparathyroidism was observed in non-users of calcium supplements, possibly owing to lower dietary calcium intake. These results highlight the importance of monitoring PPI use, especially in older adults at risk of fracture.
Asunto(s)
Hiperparatiroidismo , Fracturas Osteoporóticas , Humanos , Persona de Mediana Edad , Anciano , Anciano de 80 o más Años , Inhibidores de la Bomba de Protones/efectos adversos , Fracturas Osteoporóticas/inducido químicamente , Fracturas Osteoporóticas/epidemiología , Calcio , Estudios Transversales , Estudios de Cohortes , Hormona Paratiroidea , Hiperparatiroidismo/inducido químicamente , Hiperparatiroidismo/tratamiento farmacológicoRESUMEN
Lithium is an efficient treatment of bipolar disorder. Besides renal insufficiency, many endocrine side effects are described such as the occurrence of thyroid disorders, hypercalcaemia and nephrogenic diabetes insipidus. Lithium inhibits the secretion of thyroid hormones. The prevalence of goiter is 4 times more common in Lithium-treated patients compared as to the general population. Hypothyroidism (8-20%) is more frequent in women and in case of pre-existing thyroid autoimmunity. Grave's disease and other hyperthyroidisms are sometimes reported. Lithium stimulates the proliferation of parathyroid cells by activating the Wnt pathway. An increase in serum calcium and PTH is described in patients treated with Lithium with a 4 to 6-fold higher risk of primary hyperparathyroidism than in the general population. Nevertheless, 24-hour urine calcium is not often increased, and the phenotype can mimic a hypercalcemia-hypocalciuria syndrome that may regress with Lithium discontinuation. Surgery should be cautious since parathyroid hyperplasia is more common than parathyroid adenoma. Nephrogenic diabetes insipidus is frequently reported and may be debilitating, sometimes intricated with severe dehydration, hypernatremia, and acute renal insufficiency. Nephrogenic diabetes insipidus is not generally reversible after Lithium discontinuation, especially in patients who have chronic kidney disease due to interstitial tubule nephritis. In conclusion, clinical assessment (goiter, diuresis) and biological monitoring of serum calcium, sodium creatinine, TSH and lithium are recommended in patients receiving Lithium therapy. The risk of Lithium discontinuation in case of side effects should be weighed against the psychological risk, and must be discussed with the psychiatrist.
Asunto(s)
Diabetes Insípida Nefrogénica , Bocio , Hipercalcemia , Hiperparatiroidismo , Calcio , Diabetes Insípida Nefrogénica/inducido químicamente , Diabetes Insípida Nefrogénica/tratamiento farmacológico , Diabetes Insípida Nefrogénica/epidemiología , Endocrinólogos , Femenino , Bocio/inducido químicamente , Bocio/tratamiento farmacológico , Bocio/epidemiología , Humanos , Hipercalcemia/inducido químicamente , Hipercalcemia/tratamiento farmacológico , Hipercalcemia/epidemiología , Hiperparatiroidismo/tratamiento farmacológico , Litio , Compuestos de Litio/efectos adversosRESUMEN
Hormonal coordination is tightly regulated within the human body and thus regulates human physiology. The parathyroid hormone (PTH), a member of the endocrine system, regulates the calcium and phosphate level within the human body. Under non-physiological conditions, PTH levels get upregulated (hyperparathyroidism) or downregulated (hypoparathyroidism) due to external or internal factors. In case of hyperparathyroidism, elevated PTH stimulates cellular receptors present in the bones, kidneys, and intestines to increase the blood calcium level, leading to calcium deposition. This eventually causes various symptoms, including kidney stones. Currently, there is no known medication that directly targets PTH in order to suppress its function. Therefore, it is of great interest to find novel small molecules or any other means that can modulate PTH function. The molecular signaling of PTH starts by binding its N-terminus to the G-protein coupled PTH1/2 receptor. Therefore, any intervention that affects the N-terminus of PTH could be a lead candidate for treating hyperparathyroidism. As a proof-of-concept, there are various possibilities to inhibit molecular PTH function by (i) a small molecule, (ii) N-terminal PTH phosphorylation, (iii) fibril formation and (iv) residue-specific mutations. These modifications put PTH into an inactive state, which will be discussed in detail in this review article. We anticipate that exploring small molecules or other means that affect the N-terminus of PTH could be lead candidates in combating hyperparathyroidism.
Asunto(s)
Hiperparatiroidismo , Hormona Paratiroidea , Calcio/metabolismo , Descubrimiento de Drogas , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/metabolismo , Riñón/metabolismo , Hormona Paratiroidea/metabolismoRESUMEN
Diarrhea is a major cause of global mortality, and outbreaks of secretory diarrhea such as cholera remain an important problem in the developing world. Current treatment of secretory diarrhea primarily involves supportive measures, such as fluid replacement. The calcium-sensing receptor (CaSR) regulates multiple biological activities in response to changes in extracellular Ca2+. The FDA-approved drug cinacalcet is an allosteric activator of CaSR used for treatment of hyperparathyroidism. Here, we found by short-circuit current measurements in human colonic T84 cells that CaSR activation by cinacalcet reduced forskolin-induced Cl- secretion by greater than 80%. Cinacalcet also reduced Cl- secretion induced by cholera toxin, heat-stable E. coli enterotoxin, and vasoactive intestinal peptide (VIP). The cinacalcet effect primarily involved indirect inhibition of cystic fibrosis transmembrane conductance regulator-mediated (CFTR-mediated) Cl- secretion following activation of CaSR and downstream phospholipase C and phosphodiesterases. In mice, cinacalcet reduced fluid accumulation by more than 60% in intestinal closed loop models of cholera and traveler's diarrhea. The cinacalcet effect involved both inhibition of CFTR-mediated secretion and stimulation of sodium-hydrogen exchanger 3-mediated absorption. These findings support the therapeutic utility of the safe and commonly used drug cinacalcet in CFTR-dependent secretory diarrheas, including cholera, traveler's diarrhea, and VIPoma.
Asunto(s)
Cinacalcet/uso terapéutico , Regulador de Conductancia de Transmembrana de Fibrosis Quística/efectos adversos , Diarrea/tratamiento farmacológico , Reposicionamiento de Medicamentos/métodos , Receptores Sensibles al Calcio/uso terapéutico , Animales , Toxinas Bacterianas , Línea Celular , Toxina del Cólera , Cinacalcet/metabolismo , Colon/metabolismo , Regulador de Conductancia de Transmembrana de Fibrosis Quística/metabolismo , Diarrea/metabolismo , Enterotoxinas , Escherichia coli , Proteínas de Escherichia coli , Femenino , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Mucosa Intestinal/metabolismo , Intestinos/efectos de los fármacos , Masculino , RatonesRESUMEN
BACKGROUND AND AIMS: There are only few data on the influence of cinacalcet on the outcome of parathyroidectomy in patients with renal hyperparathyroidism. Indication and timing of surgery have changed since its introduction, especially with regard to kidney transplantation. Therefore, we retrospectively analyzed patients undergoing parathyroidectomy for renal hyperparathyroidism in our institution. MATERIAL AND METHODS: Between 2008 and 2015, 196 consecutive operations in 191 patients were analyzed. About 80 operations (41%) were performed in patients receiving cinacalcet compared with 116 operations (59%) in patients without cinacalcet. Clinical data, preoperative medication, pre- and postoperative laboratory values, type and details of surgery including complications, as well as cardiovascular complications and kidney transplantation with graft function were recorded. RESULTS: Demographical data were similar in patients with or without cinacalcet treatment. A total of 54% of patients received a kidney graft before or after parathyroidectomy. Pre- and postoperative parathormone levels were similar in both groups (preoperatively 755 vs 742 ng/L, postoperatively 50 vs 46 ng/L, p > 0.10), whereas patients with cinacalcet showed significantly lower calcium levels preoperatively (2.28 vs 2.41 mmol/L, p = 0.0002). There was no difference in recurrence or persistence of hyperparathyroidism, duration of surgery, hospital stay, or complication rate. Creatinine levels in patients with tertiary hyperparathyroidism were similar after 1-year follow-up. CONCLUSION: Cinacalcet did not influence outcome of patients with parathyroidectomy for renal hyperparathyroidism and can be safely offered to patients not responding to medical treatment.
Asunto(s)
Calcimiméticos/uso terapéutico , Cinacalcet/uso terapéutico , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/cirugía , Paratiroidectomía , Biomarcadores/sangre , Enfermedades Cardiovasculares/epidemiología , Femenino , Humanos , Trasplante de Riñón , Masculino , Persona de Mediana Edad , Complicaciones Posoperatorias/epidemiología , Estudios RetrospectivosRESUMEN
Lithium-induced nephrogenic diabetes insipidus (NDI) is a rare and difficult-to-treat condition. We describe the case of an 81-year-old woman with bipolar treated with lithium and no previous history of diabetes insipidus. She was hospitalized due to disturbance of consciousness and was diagnosed with, hypercalcemia, hyperparathyroidism, and NDI. Parathyroidectomy was contraindicated and parathyroid hormone level was improved insufficiently after cinacalcet initiation, percutaneous ethanol injection therapy was performed for the enlarged parathyroid gland. After improvement in hypercalcemia and unsuccessful indapamide treatment, triamterene was administrated to control polyuria. Lithium is one of the indispensable maintenance treatment options for bipolar disorder, but it has the side effect of NDI. Lithium enters the collecting duct's principal cells mainly via the epithelial sodium channel (ENaC) located on their apical membranes, ENaC shows high selectivity for both sodium and lithium, is upregulated by aldosterone, and inhibited by triamterene. To our knowledge, this is the first publication on triamterene use in lithium-induced NDI patients.
Asunto(s)
Diabetes Insípida Nefrogénica/inducido químicamente , Diuréticos/uso terapéutico , Litio/toxicidad , Metales Alcalinos/toxicidad , Triantereno/uso terapéutico , Administración Cutánea , Anciano de 80 o más Años , Trastorno Bipolar/complicaciones , Trastorno Bipolar/tratamiento farmacológico , Depresores del Sistema Nervioso Central/administración & dosificación , Depresores del Sistema Nervioso Central/uso terapéutico , Diabetes Insípida Nefrogénica/tratamiento farmacológico , Diuréticos/administración & dosificación , Etanol/administración & dosificación , Etanol/uso terapéutico , Femenino , Humanos , Hipercalcemia/diagnóstico , Hipercalcemia/etiología , Hiperparatiroidismo/diagnóstico , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/etiología , Litio/efectos adversos , Metales Alcalinos/efectos adversos , Poliuria/tratamiento farmacológico , Poliuria/etiología , Resultado del Tratamiento , Triantereno/administración & dosificaciónRESUMEN
Haemodialysis remains the most widely used treatment for patients with end-stage renal disease. Despite the progress that has occurred in the treatment of end-stage renal disease over the last six decades, there has been a failure to translate this into the desired clinical benefits, with morbidity and mortality rates among patients on haemodialysis remaining unacceptably high. Recently, however, there have been expectations that the significant advances that took place over the last few years may result in improved outcomes. New medications for the treatment of anaemia and secondary hyperparathyroidism, as well as novel trends in the areas of iron therapy, diabetes management and physical exercise are among the most important advances which, taken together, are changing the standards of care for patients on haemodialysis. The latest advances, of relevance not only to specialists in Renal Medicine but also to general practitioners caring for these patients, are reviewed in this collaborative paper.
Asunto(s)
Manejo de la Enfermedad , Fallo Renal Crónico/terapia , Diálisis Renal/efectos adversos , Anemia/tratamiento farmacológico , Anemia/etiología , Diabetes Mellitus/terapia , Ejercicio Físico , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/etiología , Hierro/uso terapéuticoRESUMEN
INTRODUCTION: Primary hyperparathyroidism (PHPT) is rare and usually symptomatic in children. There is no approved medication to lower serum calcium levels in this patient group. Denosumab is used in adult patients with osteoporosis and hyperparathyroidism. To our knowledge, only 1 case of denosumab treatment in a child with severe PHPT has been reported to date. CASE PRESENTATION: A 16-year-old female was referred to our clinic with symptoms including pathologic fractures, nausea, emesis, and progressive weight loss. At admission, her serum total calcium was 4.17 mmol/L (reference range 2.15-2.55), parathyroid hormone 2,151 pg/mL (15-65), and phosphate 1.07 mmol/L (1.45-1.78). Due to potentially life-threatening hypercalcemia, denosumab 60 mg subcutaneously was administered after obtaining informed consent. Serum calcium levels were reduced within 12 h of injection and the patient's condition rapidly improved, which allowed genetic testing to be done prior to surgery. A heterozygous mutation in the CDC73 gene was revealed, and a parathyroidectomy was performed on day 22 after denosumab administration. Morphological examination revealed solitary parathyroid adenoma. After surgery, hypocalcemia developed requiring high doses of alfacalcidol and calcium supplements. CONCLUSION: Our case supports the previous observations in adults that denosumab can be safely and effectively used as a preoperative treatment in patients with PHPT and severe hypercalcemia and shows that it may be used in pediatric patients.
Asunto(s)
Denosumab/administración & dosificación , Hipercalcemia , Hiperparatiroidismo , Adolescente , Niño , Femenino , Humanos , Hipercalcemia/sangre , Hipercalcemia/tratamiento farmacológico , Hiperparatiroidismo/sangre , Hiperparatiroidismo/tratamiento farmacológicoAsunto(s)
Humanos , Hiperparatiroidismo/cirugía , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo Secundario/cirugía , Hiperparatiroidismo Secundario/tratamiento farmacológico , Hormona Paratiroidea , Calcio , Paratiroidectomía , Resultado del Tratamiento , Calcimiméticos/uso terapéutico , Cinacalcet/uso terapéuticoAsunto(s)
Hipercalcemia , Hiperparatiroidismo , Anciano , Femenino , Humanos , Hipercalcemia/inducido químicamente , Hipercalcemia/diagnóstico , Hipercalcemia/tratamiento farmacológico , Hiperparatiroidismo/complicaciones , Hiperparatiroidismo/tratamiento farmacológico , Litio/efectos adversos , Vitamina D/análogos & derivadosAsunto(s)
Hiperparatiroidismo Secundario , Hiperparatiroidismo , Calcimiméticos/uso terapéutico , Calcio , Cinacalcet/uso terapéutico , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/cirugía , Hiperparatiroidismo Secundario/tratamiento farmacológico , Hiperparatiroidismo Secundario/cirugía , Hormona Paratiroidea , Paratiroidectomía , Resultado del TratamientoRESUMEN
OBJECTIVES: To evaluate the practice and attitude of pediatrics nephrologists about cinacalcet use in children. METHODS: An electronic structured questionnaire was answered by pediatric nephrologists practicing in the Kingdom of Saudi Arabia (KSA) and Gulf Council countries (GCC). RESULTS: A total of 42 pediatric nephrologists responded, of them, 42% used cinacalcet for young children ≤5 years of age and 79% used for children. There were wide variations in the method of administration (examples: crushed, divided, whole tablets), monitoring, doses and response definition, and follow-up. No serious complications after starting cinacalcet was observed in 50%, while 40% reported various complications, mainly hypocalcemia (70%). Cinacalcet was stopped without achieving the target parathyroid hormone in more than half (55%) of children because of intractable adverse effects (40%), poor response (30%), non-adherence (25%), or high cost (5%). CONCLUSION: Cinacalcet is used by the majority of pediatric nephrologists in KSA and GCC. A standard clinical guideline is needed to be followed by all users.
Asunto(s)
Enfermedades Óseas Metabólicas/tratamiento farmacológico , Cinacalcet/administración & dosificación , Insuficiencia Renal Crónica/complicaciones , Insuficiencia Renal Crónica/tratamiento farmacológico , Factores de Edad , Enfermedades Óseas Metabólicas/etiología , Niño , Preescolar , Cinacalcet/efectos adversos , Femenino , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/etiología , Lactante , Masculino , Guías de Práctica Clínica como Asunto , Encuestas y Cuestionarios , Resultado del TratamientoRESUMEN
BACKGROUND: Persistent post-transplant hyperparathyroidism (PPTHP) can occur in 20% to 50% of renal transplant recipients. The aim of this study was to analyze safety and efficacy of long-term cinacalcet therapy in a group of renal transplant recipients with PPTHP. METHODS: A single center retrospective cohort study including renal transplant recipients, adults (>18 years old) with PPTHP and hypercalcemia. Inclusion criteria for cinacalcet therapy was increased parathormone levels (PTH > 65 pg/mL) associated with serum calcium >11.5 mg/dL any time after transplant or calcium >10.2 mg/dL within the first year after transplant. The follow-up period was 3 years. Demographic, laboratory data and adverse events were assessed. RESULTS: Forty-six patients were included, mean age of 50 ± 11 years old, majority of white race (60%), male (58%), with a pretransplant length on dialysis of 67 ± 34 months. Cinacalcet therapy was started 37 ± 40 months after transplant, and normal calcium levels were achieved after 6 months of therapy. PTH levels presented a steady reduction over time, reaching levels near normal after 36 months (317 ± 242 vs 145 ± 72 pg/mL, baseline × month 36, P < .05). Renal function remained stable over time (GFR > 60 mL/min/1.73 m2) and no acute rejection episodes were observed. Most common adverse events were mild gastrointestinal symptoms. In 6 patients (12.5%) treatment was interrupted due to adverse events. Only 1 case (2%) was classified as treatment failure. CONCLUSION: Cinacalcet therapy proved to be efficient for PPTHP and safe for graft and patient. Long-term treatment reduced PTH levels to near normal range.
Asunto(s)
Cinacalcet/administración & dosificación , Hipercalcemia/tratamiento farmacológico , Hiperparatiroidismo/tratamiento farmacológico , Trasplante de Riñón/efectos adversos , Complicaciones Posoperatorias/tratamiento farmacológico , Adulto , Calcio/sangre , Esquema de Medicación , Femenino , Humanos , Hipercalcemia/sangre , Hipercalcemia/etiología , Hiperparatiroidismo/sangre , Hiperparatiroidismo/etiología , Masculino , Persona de Mediana Edad , Hormona Paratiroidea/sangre , Complicaciones Posoperatorias/sangre , Complicaciones Posoperatorias/etiología , Estudios Retrospectivos , Factores de Tiempo , Resultado del TratamientoRESUMEN
A 77-year-old man with a history of hypertension, prostate hyperplasia, and urolithiasis was admitted for acute kidney injury caused by hypercalcemia. Neck ultrasonography showed a large cyst adjacent to the right lower thyroid lobe. Although a 99mtechnetium sestamibi scan was negative, an extremely high intracystic intact parathyroid hormone level suggested that the cyst had a parathyroid origin and that a functional parathyroid cyst was present. Immunohistochemical staining for the calcium-sensing receptor (CaSR) after right lower parathyroidectomy revealed CaSR-positive cells lining the cyst, indicating that the functional parathyroid cyst had originated from the hemorrhagic degeneration of a parathyroid adenoma.
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Adenoma/fisiopatología , Cinacalcet/uso terapéutico , Hipercalcemia/complicaciones , Hiperparatiroidismo/tratamiento farmacológico , Glándulas Paratiroides/fisiopatología , Glándulas Paratiroides/cirugía , Neoplasias de las Paratiroides/fisiopatología , Adenoma/etiología , Adenoma/cirugía , Anciano , Calcimiméticos/uso terapéutico , Quistes/fisiopatología , Quistes/cirugía , Humanos , Masculino , Neoplasias de las Paratiroides/etiología , Neoplasias de las Paratiroides/cirugía , Paratiroidectomía , Resultado del TratamientoAsunto(s)
Calcio/sangre , Suplementos Dietéticos/efectos adversos , Trastornos Nutricionales/diagnóstico , Deficiencia de Vitamina D/tratamiento farmacológico , Vitamina D/sangre , Anciano de 80 o más Años , Colecalciferol/administración & dosificación , Colecalciferol/efectos adversos , Colecalciferol/sangre , Colecalciferol/farmacocinética , Cinacalcet/efectos adversos , Relación Dosis-Respuesta a Droga , Ergocalciferoles/administración & dosificación , Ergocalciferoles/efectos adversos , Ergocalciferoles/sangre , Ergocalciferoles/farmacocinética , Femenino , Humanos , Hiperparatiroidismo/tratamiento farmacológico , Trastornos Nutricionales/sangre , Trastornos Nutricionales/inducido químicamente , Hormona Paratiroidea/sangre , Vitamina D/metabolismo , Deficiencia de Vitamina D/sangre , Deficiencia de Vitamina D/inducido químicamenteRESUMEN
Hungry Bone Syndrome (HBS) refers to rapid, profound, and prolonged hypocalcemia associated with hypophosphatemia and hypomagnesemia occurring in patients with increased bone turnover after successful management of the underlying disorder. We describe a male patient with primary hyperparathyroidism (PHPT), in whom HBS was diagnosed 6 months after parathyroidectomy. Histopathologic examination revealed an atypical parathyroid adenoma (APA), while immunohistochemistry showed cell proliferation index Ki-67 10% and overexpression of cyclin D1 (>90%). Preoperative treatment with vitamin D3 had normalized 25OHD and alkaline phosphatase levels, reflected in an improvement in bone turnover prior to surgery. Postoperative treatment for HBS with alfacalcidol, calcium, vitamin D3 and magnesium was administered for a long period. This treatment prevented severe postoperative hypocalcemia and he was discharged two days later. Preoperative cinacalcet treatment did not reduce hypercalcemia implying that the tumor had lack of calciumsensing receptors (CaSR). In conclusion, preoperative restoration of low 25OHD levels is essential for prevention of HBS. Postoperative treatment with active metabolites of vitamin D must be initiated as early as possible, in order to prevent or minimize the development of HBS, and to reduce the duration of hospitalization.
Asunto(s)
Hipocalcemia/etiología , Hipofosfatemia/etiología , Paratiroidectomía/efectos adversos , Complicaciones Posoperatorias/etiología , Adenoma/complicaciones , Adenoma/cirugía , Adulto , Conservadores de la Densidad Ósea/uso terapéutico , Calcio/uso terapéutico , Hormonas y Agentes Reguladores de Calcio/uso terapéutico , Colecalciferol/uso terapéutico , Cinacalcet/uso terapéutico , Humanos , Hidroxicolecalciferoles/uso terapéutico , Hiperparatiroidismo/tratamiento farmacológico , Hiperparatiroidismo/etiología , Hiperparatiroidismo/cirugía , Hipocalcemia/tratamiento farmacológico , Hipofosfatemia/tratamiento farmacológico , Magnesio/uso terapéutico , Masculino , Neoplasias de las Paratiroides/complicaciones , Neoplasias de las Paratiroides/cirugía , Complicaciones Posoperatorias/tratamiento farmacológico , SíndromeRESUMEN
BACKGROUND: X-linked hypophosphatemia (XLH) causes rickets, osteomalacia, skeletal deformities and growth impairment, due to elevated fibroblast growth factor 23 and hypophosphatemia. Conventional therapy requires high doses of phosphate salts combined with active vitamin D analogues. Risks of this regimen include nephrocalcinosis and secondary hyperparathyroidism or progression to tertiary (hypercalcemic) hyperparathyroidism. METHODS: The primary goals were to estimate the prevalence of hyperparathyroidism and to characterize parathyroidectomy outcomes regarding hypercalcemia among XLH patients. XLH patients attending our center from 1/2000 to 12/2017 were included in a retrospective chart review. Prevalence of nephrocalcinosis and eGFRâ¯<â¯60â¯ml/min/1.73m2 was also assessed. RESULTS: Of 104 patients with XLH, 84 had concurrent measurements of calcium and PTH (40 adults and 44 children). Of these, 70/84 (83.3%), had secondary or tertiary hyperparathyroidism at any time point. Secondary hyperparathyroidism was persistent in 62.2% of those with data at multiple timepoints. Tertiary hyperparathyroidism had an overall prevalence of 14/84 (16.7%) patients. Parathyroidectomy was performed in 8/84 (9.5%) of the total population. After parathyroidectomy, persistent or recurrent tertiary hyperparathyroidism was detected in 6/8 (75%) patients at a median of 6â¯years (from 0 to 29â¯years). One patient had chronic post-surgical hypoparathyroidism and one patient remained normocalcemic 4â¯years after surgery. Nephrocalcinosis was more prevalent in patients with tertiary hyperparathyroidism than those without (60.0% vs 18.6%). Chronic kidney disease (eGFRâ¯<â¯60â¯ml/min/1.73m2) was also more prevalent in patients with tertiary hyperparathyroidism than those without (35.7% vs 1.5%). CONCLUSION: The majority of patients with XLH develop secondary hyperparathyroidism during treatment with phosphate and active vitamin D. A significant proportion develops tertiary hyperparathyroidism and most have recurrence or persistence of hypercalcemia after surgery.