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1.
Scand J Rheumatol ; 49(4): 312-322, 2020 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-32484386

RESUMO

OBJECTIVE: This is the first randomized double-blinded, placebo-controlled pilot trial to investigate the efficacy of pamidronate in reducing radiological and clinical disease activity in chronic non-bacterial osteomyelitis (CNO). METHOD: Patients received pamidronate or placebo at baseline and weeks 12 and 24. Whole-body magnetic resonance imaging was performed at baseline and weeks 12 and 36, and computed tomography of the anterior chest wall (ACW) at baseline and week 36. Radiological disease activity was systematically scored in the ACW and spine. Patient-reported outcomes [visual analogue scale (VAS) pain, VAS global health, Health Assessment Questionnaire (HAQ), EuroQol-5 Dimensions (EQ-5D), and 36-item Short-Form Health Survey (SF-36)] and biomarkers of bone turnover and inflammation were assessed at baseline and weeks 1, 4, 12, 24, and 36. Data are expressed as median [interquartile range]. RESULTS: Fourteen patients were randomized and 12 were analysed. From baseline to week 36, the radiological disease activity score in the ACW decreased from 5 [4-7] to 2.5 [1-3] in the pamidronate group, but did not change in the placebo group (p = 0.04). From baseline to week 36, VAS pain and VAS global health tended to decrease more in the pamidronate than in the placebo group (p = 0.11, p = 0.08). Physical functioning (HAQ) and health-related quality of life (EQ-5D, SF-36) did not change. Biomarkers of bone turnover decreased only in the pamidronate group (p ≤ 0.02). CONCLUSION: Pamidronate may improve radiological and clinical disease activity in CNO. Methods to score radiological disease activity in adult CNO were suggested. Clinical Trials: NCT02594878.


Assuntos
Conservadores da Densidade Óssea/uso terapêutico , Osteomielite/tratamento farmacológico , Pamidronato/uso terapêutico , Coluna Vertebral/efeitos dos fármacos , Parede Torácica/efeitos dos fármacos , Adulto , Biomarcadores/sangue , Conservadores da Densidade Óssea/farmacologia , Remodelação Óssea/efeitos dos fármacos , Método Duplo-Cego , Feminino , Humanos , Imagem por Ressonância Magnética , Masculino , Pessoa de Meia-Idade , Osteomielite/sangue , Osteomielite/diagnóstico por imagem , Pamidronato/farmacologia , Medidas de Resultados Relatados pelo Paciente , Projetos Piloto , Coluna Vertebral/diagnóstico por imagem , Parede Torácica/diagnóstico por imagem , Imagem Corporal Total , Adulto Jovem
2.
Pediatr Rheumatol Online J ; 18(1): 4, 2020 Jan 15.
Artigo em Inglês | MEDLINE | ID: mdl-31941491

RESUMO

BACKGROUND: Studies evaluating treatment responses for chronic nonbacterial osteomyelitis (CNO) are lacking. We aimed to measure and compare response rates of medical treatments, time to response of medical treatments among patients with CNO of the mandible, and describe bacterial contamination rates from biopsy. METHODS: We conducted a retrospective chart review of all patients diagnosed with CNO of mandible between 2003 and 2017 and extracted demographic, clinical, laboratory, imaging and surgical data. Detailed medication use and response to medications were recorded. The primary outcome was response to medical treatments defined as improvement of presenting symptoms, inflammatory markers, and imaging if available. Medical treatments included nonsteroidal anti-inflammatory drugs (NSAIDs), glucocorticoids, disease modifying anti rheumatic drugs (DMARDs), anti-tumor necrosis factor (TNF) therapy, and pamidronate. Descriptive analysis was performed when appropriate. Multivariable logistic regression and Kaplan-Meier curves were applied to compare the responses to medical treatments and time to full response. RESULTS: We identified 22 patients with a median age of 11 and 36% were female. Four patients (18%) had multifocal bone lesions. CT findings (n = 21) showed lytic lesions (62%) and sclerosis (90%). MRI (n = 14) revealed hyperintensity within bone marrow (100%), soft tissue (71%) and bony expansion (71%). Non-antibiotic treatments including NSAIDs (n = 18), glucocorticoids (n = 10), DMARDs (n = 9), anti-TNF therapy (n = 5) and pamidronate (n = 6) were applied. Rates of full responses to anti-TNF therapy (60%) and pamidronate (67%) were higher than that to NSAIDs (11%) (p < 0.05). Patients receiving pamidronate responded more rapidly than those receiving anti-TNF therapy (median two vs 17 months, p = 0.01) when there was a full response. All had bone biopsies. Intraoral biopsy was performed in 12 of 13 operated in our center and the most common contaminants were Neisseria spp and Streptococcus viridians. CONCLUSION: Both anti-TNF and pamidronate appeared superior to NSAIDs alone in treating mandibular CNO. Patients receiving pamidronate responded faster than those receiving anti-TNF therapy.


Assuntos
Doenças Mandibulares/tratamento farmacológico , Osteomielite/tratamento farmacológico , Anti-Inflamatórios não Esteroides/uso terapêutico , Criança , Feminino , Humanos , Imagem por Ressonância Magnética , Masculino , Doenças Mandibulares/diagnóstico , Doenças Mandibulares/diagnóstico por imagem , Doenças Mandibulares/patologia , Osteomielite/diagnóstico , Osteomielite/diagnóstico por imagem , Osteomielite/patologia , Pamidronato/uso terapêutico , Estudos Retrospectivos , Tomografia Computadorizada por Raios X , Resultado do Tratamento , Fator de Necrose Tumoral alfa/antagonistas & inibidores
3.
Indian Pediatr ; 57(1): 75-76, 2020 01 15.
Artigo em Inglês | MEDLINE | ID: mdl-31937708

RESUMO

Juvenile dermatomyositis is a rare systemic autoimmune disease wth calcinosis as its hallmark sequelae. We report three patients with juvenile dermatomyositis with calcinosis, who were treated with pamidronate. There was complete clearance of calcinosis in one child.


Assuntos
Calcinose , Dermatomiosite/complicações , Pamidronato/uso terapêutico , Calcinose/tratamento farmacológico , Calcinose/etiologia , Calcinose/patologia , Criança , Pré-Escolar , Feminino , Dedos/patologia , Humanos , Masculino
4.
BMJ Case Rep ; 12(10)2019 Oct 30.
Artigo em Inglês | MEDLINE | ID: mdl-31666255

RESUMO

We report the case of a 59-year-old man with a history of type 2 diabetes, hypertension and chronic kidney disease who presented with symptomatic severe hypercalcaemia (calcium 15.8 mg/dL) and acute kidney injury. Evaluation revealed that the hypercalcaemia was not mediated by parathyroid hormone (PTH), PTH-related peptide or 1,25-hydroxyvitamin D. Adrenal insufficiency was subsequently diagnosed and was initially thought to be the aetiology of the hypercalcaemia. He was treated with intravenous fluid, pamidronate and started on hydrocortisone with resolution of his hypercalcaemia. Over the next several months, despite adherence to hydrocortisone therapy, the patient continued to have recurrent severe hypercalcaemia requiring hospitalisation. Additional laboratory evaluation showed similar results to the initial evaluation. On further questioning, the patient admitted to routinely ingesting the household cleaning product Comet, which contains a large amount of calcium. Psychiatric assessment confirmed the diagnosis of pica. The patient eventually discontinued ingestion of Comet with resolution of his hypercalcaemia.


Assuntos
Ingestão de Alimentos/psicologia , Produtos Domésticos/efeitos adversos , Hipercalcemia/etiologia , Pica/psicologia , Lesão Renal Aguda , Anti-Inflamatórios/administração & dosagem , Anti-Inflamatórios/uso terapêutico , Conservadores da Densidade Óssea/administração & dosagem , Conservadores da Densidade Óssea/uso terapêutico , Produtos Domésticos/toxicidade , Humanos , Hidrocortisona/administração & dosagem , Hidrocortisona/uso terapêutico , Hipercalcemia/tratamento farmacológico , Masculino , Pessoa de Meia-Idade , Pamidronato/administração & dosagem , Pamidronato/uso terapêutico , Pica/diagnóstico , Recidiva , Índice de Gravidade de Doença , Resultado do Tratamento
5.
BMJ Case Rep ; 12(9)2019 Sep 18.
Artigo em Inglês | MEDLINE | ID: mdl-31537587

RESUMO

We describe an 11-year prospective clinical and radiologic course of a 6-year-old boy with bilateral Legg-Calvé-Perthes disease, who was treated with intravenous pamidronate (IV-PAM). His baseline radiographs showed grade IV avascular necrosis/Catterall stage IV, and at worst he progressed to lateral pillar/Herring stage C bilaterally. His disease initially was extremely functionally limiting with expected poor outcome with eventual joint replacement. Because IV-PAM stops bone breakdown and allows for ongoing bone formation while revascularisation of bone occurs, we hypothesised that IV-PAM could act as an adjunct to traditional treatment to help heal the femoral heads. Our patient received nine once monthly doses of IV-PAM (1 mg/kg/dose) over 13 months, along with Petrie/broomstick casts and physiotherapy. Remarkably, over time, his femoral heads healed. Now, at 11-year follow-up, he has excellent functional and radiologic outcome with congruence between femoral head and acetabulum, no residual osteonecrosis and minimal loss of femoral head sphericity.


Assuntos
Conservadores da Densidade Óssea/administração & dosagem , Necrose da Cabeça do Fêmur/tratamento farmacológico , Doença de Legg-Calve-Perthes/tratamento farmacológico , Pamidronato/uso terapêutico , Conservadores da Densidade Óssea/uso terapêutico , Moldes Cirúrgicos , Criança , Cabeça do Fêmur/efeitos dos fármacos , Articulação do Quadril/diagnóstico por imagem , Articulação do Quadril/patologia , Humanos , Doença de Legg-Calve-Perthes/reabilitação , Doença de Legg-Calve-Perthes/cirurgia , Masculino , Pamidronato/administração & dosagem , Tenotomia/métodos , Resultado do Tratamento
6.
Pediatr Rheumatol Online J ; 17(1): 35, 2019 Jul 04.
Artigo em Inglês | MEDLINE | ID: mdl-31272461

RESUMO

BACKGROUND: To study the response to pamidronate using whole body magnetic resonance imaging (WB-MRI) in children with chronic non-bacterial osteitis (CNO) in a tertiary health centre. METHODS: The medical records of children under the age of sixteen with a diagnosis of chronic non-bacterial osteitis between 2005 and 2018 were reviewed. All those who were treated with pamidronate were included and relevant data was collected. Response to therapy was determined based on the status of lesions on WB- MRI. RESULTS: Forty six patients were included in the study. Pre- and post-treatment WB-MRI was available in forty patients. Cumulative lesions pre-treatment were 150 and reduced to 45 (30%) post-treatment. Seventeen patients (42.5%) had a good response with complete resolution of all lesions and nine patients (22.5%) worsened during or following treatment with pamidronate. Vertebral disease had a good response and 82.3% of the lesions resolved completely. CONCLUSION: Our study describes the experience with pamidronate in a tertiary health centre using WB-MRI as a marker of disease activity. Pamidronate was well tolerated in our cohort and treatment response was fairly good. SIGNIFICANCE AND INNOVATION: 1. Bisphosphonates can be used in the treatment of CNO when response to NSAIDs is suboptimal. 2. In the presence of spinal or mandibular lesions bisphosphonates were used as first line. 3. Treatment was escalated to a TNF blocker when response to bisphosphonates was suboptimal.


Assuntos
Osteíte/tratamento farmacológico , Pamidronato/uso terapêutico , Adolescente , Conservadores da Densidade Óssea/uso terapêutico , Criança , Pré-Escolar , Feminino , Humanos , Lactente , Imagem por Ressonância Magnética/métodos , Masculino , Estudos Retrospectivos , Resultado do Tratamento , Imagem Corporal Total/métodos
7.
PLoS One ; 14(6): e0215739, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31166977

RESUMO

INTRODUCTION: Chronic nonbacterial osteomyelitis (CNO) is a rare autoinflammatory bone disorder primarily affecting children and adolescents. It can lead to chronic pain, bony deformities and fractures. The pathophysiology of CNO is incompletely understood. Scientific evidence suggests dysregulated expression of pro- and anti-inflammatory cytokines to be centrally involved. Currently, treatment is largely based on retrospective observational studies and expert opinion. Treatment usually includes nonsteroidal anti-inflammatory drugs and/or glucocorticoids, followed by a range of drugs in unresponsive cases. While randomised clinical trials are lacking, retrospective and prospective non-controlled studies suggest effectiveness of TNF inhibitors and bisphosphonates. The objective of the Bayesian consensus meeting was to quantify prior expert opinion. METHODS: Twelve international CNO experts were randomly chosen to be invited to a Bayesian prior elicitation meeting. RESULTS: Results showed that a typical new patient treated with pamidronate would have an 84% chance of improvement in their pain score relative to baseline at 26 weeks and an 83% chance on adalimumab. Experts thought there was a 50% chance that a new typical patient would record a pain score of 28mm (pamidronate) to 30mm (adalimumab) or better at 26 weeks. There was a modest trend in prior opinion to indicate an advantage of pamidronate vs adalimumab, with a 68% prior chance that pamidronate is superior to adalimumab by some margin. However, it is clear that there is considerable uncertainty about the precise relative merits of the two treatments. CONCLUSIONS: The rarity of CNO leads to challenges in conducting randomised controlled trials with sufficient power to provide a definitive outcome. We address this using a Bayesian design, and here describe the process and outcome of the elicitation exercise to establish expert prior opinion. This opinion will be tested in the planned prospective CNO study. The process for establishing expert consensus opinion in CNO will be helpful for developing studies in other rare paediatric diseases.


Assuntos
Adalimumab/uso terapêutico , Osteomielite/tratamento farmacológico , Pamidronato/uso terapêutico , Teorema de Bayes , Consenso , Feminino , Humanos , Masculino , Osteomielite/complicações , Manejo da Dor , Ensaios Clínicos Controlados Aleatórios como Assunto , Projetos de Pesquisa
11.
Emerg Med J ; 36(1): 17-32, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30635344

RESUMO

CLINICAL INTRODUCTION: A 77-year-old man presented to the ED with a history of fevers, purulent drainage and right mandibular pain. He had been diagnosed with multiple myeloma 2 years previously and was receiving treatment with pamidronate. On presentation, the lower right lip and chin were anaesthetic, tooth number 31 had grade 2 mobility and a 15 mm long ulceration was present on the lingual aspect of the mandible (figure 1). Antibiotics were administered, and a maxillofacial CT without contrast was performed (figure 2).emermed;36/1/17/F1F1F1Figure 1Clinical examination revealing a 15 mm long ulceration (arrow mark) associated with the lingual aspect of tooth number 31.emermed;36/1/17/F2F2F2Figure 2CT maxillofacial (coronal) demonstrating osseous destruction (arrow mark) of the right mandibular body. QUESTION: What is your diagnosis?Odontogenic abscessBenign fibro-osseous lesionMedication-related osteonecrosis of the jaw (MRONJ)Metastatic malignancy.


Assuntos
Osteonecrose/cirurgia , Pamidronato/efeitos adversos , Abscesso/tratamento farmacológico , Abscesso/cirurgia , Idoso , Antibacterianos/uso terapêutico , Conservadores da Densidade Óssea/efeitos adversos , Conservadores da Densidade Óssea/uso terapêutico , Drenagem/métodos , Febre/etiologia , Humanos , Masculino , Mandíbula/microbiologia , Mandíbula/fisiopatologia , Osteonecrose/tratamento farmacológico , Dor/etiologia , Pamidronato/uso terapêutico , Tomografia Computadorizada por Raios X/métodos
12.
BMJ Case Rep ; 12(1)2019 Jan 22.
Artigo em Inglês | MEDLINE | ID: mdl-30674496

RESUMO

This is a case of a 67-year-old woman, known to have multiple medical problems, mainly papillary thyroid cancer status post-total thyroidectomy and cervical neck dissection in addition to radioactive iodine currently in remission for 1 year, who presented to the hospital with severe weakness and fatigue. The initial workup showed significant hypercalcaemia and suppressed Parathyroid hormone (PTH). The patient was treated with hydration and pamidronate and her hypercalcaemia and symptoms improved. The differential was wide, however, a CT scan of the chest, abdomen and pelvis did show multiple liver and splenic nodular lesions; therefore, malignancy was the highest possible diagnosis. Biopsy of the splenic lesion confirmed the diagnosis of sarcoidosis. Therefore, the patient was diagnosed with primary isolated nodular hepatosplenic sarcoidosis mimicking malignancy and causing significant symptomatic hypercalcaemia.


Assuntos
Hipercalcemia/etiologia , Neoplasias/diagnóstico por imagem , Sarcoidose/complicações , Assistência ao Convalescente , Idoso , Conservadores da Densidade Óssea/uso terapêutico , Diagnóstico Diferencial , Feminino , Humanos , Hipercalcemia/diagnóstico , Hipercalcemia/tratamento farmacológico , Hepatopatias/complicações , Hepatopatias/diagnóstico por imagem , Hepatopatias/patologia , Pamidronato/administração & dosagem , Pamidronato/uso terapêutico , Sarcoidose/diagnóstico por imagem , Sarcoidose/patologia , Sarcoidose/terapia , Esplenopatias/complicações , Esplenopatias/diagnóstico por imagem , Esplenopatias/patologia , Tomografia Computadorizada por Raios X , Resultado do Tratamento
13.
J Natl Compr Canc Netw ; 17(1): 22-28, 2019 01.
Artigo em Inglês | MEDLINE | ID: mdl-30659126

RESUMO

Background: Bisphosphonates reduce skeletal-related events (SREs) in patients with multiple myeloma (MM) and, in some studies, improved survival. Since 2011, bisphosphonate use has been recommended by NCCN for all patients with newly diagnosed MM receiving antineoplastic therapy independent of the presence of bone disease. This study investigated their use after these guidelines were established. Methods: We identified patients aged ≥65 years in the SEER-Medicare database with newly diagnosed MM between January 1, 2012, and December 31, 2013, who received antineoplastic therapy, had ≥6 months of follow-up, and did not receive prior bisphosphonates. Presence of SREs at diagnosis was identified, including pathologic fracture, spinal cord compression, radiation to bone, or surgery to bone. Use of bisphosphonates was defined as having ≥1 claim for an intravenous or oral bisphosphonate within 6 months after the start of antineoplastic therapy. We used multivariable modeling to compare users with nonusers, controlling for demographic and clinical covariates. We compared overall survival between users and nonusers using proportional hazards analysis. Results: Of 1,309 patients identified, 720 (55%) used a bisphosphonate. Factors associated with use included SRE at diagnosis (adjusted odds ratio [AOR], 2.60; 95% CI, 1.98-3.40), hypercalcemia (AOR, 1.74; 95% CI, 1.26-2.41), and use of proteasome inhibitor + immunomodulatory imide therapy (AOR, 1.70; 95% CI, 1.21-2.39). Chronic kidney disease (AOR, 0.48; 95% CI, 0.35-0.66) was associated with decreased use. Bisphosphonate use was associated with reduced mortality (hazard ratio, 0.70; 95% CI, 0.56-0.88). Conclusions: Although bisphosphonate use is recommended for all patients with newly diagnosed MM receiving antineoplastic therapy, 45% of patients in the United States did not receive this guideline-recommended care.


Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/efeitos adversos , Conservadores da Densidade Óssea/uso terapêutico , Doenças Ósseas/prevenção & controle , Mieloma Múltiplo/complicações , Idoso , Idoso de 80 Anos ou mais , Conservadores da Densidade Óssea/normas , Doenças Ósseas/epidemiologia , Doenças Ósseas/etiologia , Feminino , Seguimentos , Humanos , Estimativa de Kaplan-Meier , Masculino , Medicare/estatística & dados numéricos , Mieloma Múltiplo/tratamento farmacológico , Mieloma Múltiplo/mortalidade , Pamidronato/uso terapêutico , Guias de Prática Clínica como Assunto , Estudos Retrospectivos , Programa de SEER/estatística & dados numéricos , Resultado do Tratamento , Estados Unidos/epidemiologia , Ácido Zoledrônico/uso terapêutico
14.
J Bone Miner Metab ; 37(3): 545-553, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30187275

RESUMO

The purpose of this study was to clarify the prevalence of scoliosis and determine risk factors for the development of scoliosis in young children with osteogenesis imperfecta (OI) who underwent intravenous pamidronate (PAM) therapy. Thirty-four young children with OI who had no scoliosis at the first PAM administration underwent cyclic PAM therapy alone. The medical records and radiographs of these patients were retrospectively reviewed. We examined the relationship between scoliosis (Cobb angle ≥ 10) and type of OI (Sillence classification: types I, III, and IV), physical mobility, Z-scores of bone mineral density in L2-4 of the lumbar spine (L2-4 BMD Z-scores), age of patients at first treatment with PAM, pelvic frontal tilt and leg-length discrepancy. The prevalence of scoliosis was 23.5% in 34 young children with OI who underwent PAM therapy for a mean of 4.2 years. Lower L2-4 BMD Z-scores, the presence of coronal and sagittal vertebral deformities and higher percentage of corrective osteotomy in the lower extremities were significant risk factors for the development of scoliosis. In patients with type III or IV OI, L2-4 BMD Z-scores were significantly lower (p = 0.02) and the percentage of patients who started PAM therapy in early childhood was significantly lower in scoliosis group than in the non-scoliosis group (p = 0.01). Development of scoliosis depends on the severity of OI and has a strong relationship with bone fragility even under PAM therapy. Starting intravenous PAM therapy in infancy or early childhood has a potential to prevent the occurrence and progression of scoliosis associated with bone fragility in young children with severe type III or IV OI.


Assuntos
Difosfonatos/administração & dosagem , Difosfonatos/uso terapêutico , Osteogênese Imperfeita/complicações , Osteogênese Imperfeita/tratamento farmacológico , Escoliose/complicações , Administração Intravenosa , Adolescente , Criança , Pré-Escolar , Feminino , Humanos , Masculino , Osteogênese Imperfeita/diagnóstico por imagem , Osteogênese Imperfeita/epidemiologia , Pamidronato/farmacologia , Pamidronato/uso terapêutico , Estudos Retrospectivos , Fatores de Risco , Escoliose/diagnóstico por imagem , Fatores de Tempo
15.
J Clin Endocrinol Metab ; 104(3): 721-729, 2019 03 01.
Artigo em Inglês | MEDLINE | ID: mdl-30247615

RESUMO

Context: Primary hyperparathyroidism (PHPT) in pregnancy has historically been associated with substantial maternofetal morbidity and mortality rates. The optimal treatment and timing of surgical intervention in pregnancy remain contested. Objective: To compare maternofetal outcomes of medically and surgically treated patients with PHPT in pregnancy. Design: Retrospective chart review. Setting: Quaternary referral hospital. Patients: Women with PHPT in pregnancy treated between 1 January 2000 and 31 December 2015. Interventions: Medical therapy or parathyroid surgery. Main Outcomes Measured: Timing of diagnosis; maternal corrected serum calcium concentrations; gestation, indication and mode of delivery; complications attributable to PHPT; birth weight; and admission to the neonatal intensive care unit (NICU). Results: Twenty-two pregnancies were managed medically, and six patients underwent parathyroidectomy in pregnancy (five in trimester 2, and one at 32 weeks gestation). Most patients treated medically either had a corrected serum calcium concentration <2.85 mmol/L in early pregnancy or had PHPT diagnosed in trimester 3. Of viable medically managed pregnancies, 30% were complicated by preeclampsia, and preterm delivery occurred in 66% of this group. All preterm neonates required admission to the NICU for complications related to prematurity. All surgically treated patients delivered their babies at term, and there were no complications of parathyroid surgery. Conclusion: Maternofetal outcomes have improved relative to that reported in early medical literature in patients treated medically and surgically, but the rates of preeclampsia and preterm delivery were higher in medically treated patients. The study was limited by its retrospective design and small sample sizes.


Assuntos
Hiperparatireoidismo Primário/terapia , Doenças do Prematuro/epidemiologia , Pré-Eclâmpsia/epidemiologia , Nascimento Prematuro/epidemiologia , Adulto , Anti-Hipertensivos/uso terapêutico , Cálcio/sangue , Feminino , Humanos , Hiperparatireoidismo Primário/sangue , Hiperparatireoidismo Primário/complicações , Recém-Nascido , Doenças do Prematuro/etiologia , Doenças do Prematuro/terapia , Infusões Intravenosas , Unidades de Terapia Intensiva Neonatal/estatística & dados numéricos , Pamidronato/uso terapêutico , Paratireoidectomia/estatística & dados numéricos , Pré-Eclâmpsia/etiologia , Gravidez , Nascimento Prematuro/etiologia , Encaminhamento e Consulta/estatística & dados numéricos , Soluções para Reidratação/administração & dosagem , Estudos Retrospectivos , Fatores de Tempo , Adulto Jovem
16.
Rheumatol Int ; 39(1): 89-96, 2019 01.
Artigo em Inglês | MEDLINE | ID: mdl-30171342

RESUMO

Chronic non-bacterial osteomyelitis (CNO) is a chronic inflammatory bone disease which usually manifests in children and adolescents. There are a few data about pathogenesis and treatment. The aim of the study to compare the efficacy of different treatment approaches in pediatric CNO cohort patient. Fifty two children (25 boys and 27 girls) with CNO with average age at the onset of the disease 8.4 years (5.4; 11.0), number of foci - 3.0 (2.0; 6.0, incl. multifocal cases in 80.8%). Non-steroid anti-inflammatory drugs (NSAID) was the first-line treatment for non-vertebral cases, as well as pamidronate (PAM) for vertebral involvement. Second-line treatment includes sulfasalazine (SSZ), methotrexate (MTX), PAM and tumor necrosis factor-α inhibitors (TNFα-inh). We evaluated the dynamics of pain, patient's and physician's (MDVAS) assessment with visual-analog scale (VAS) and ability to each medication to achieve remission of CNO activity. According to the NSAID, MTX, SSZ, PAM and TNFα-inh groups the following data were registered: patient's VAS: - 14.2% (p = 0.05), - 50.0% (p = 0.04), - 23.1 (p = 0.89), - 83.3% (p = 0.0001), - 73.6% (p = 0.0007); painVAS: - 21.9% (p = 0.01), - 18.6% (p = 0.13), + 36.4 (p = 0.89), - 79.7% (p = 0.00016), - 74.1%, (p = 0.0015); MDVAS: - 13.8% (p = 0.13); - 56.4% (p = 0.09), + 30.8% (p = 0.89), - 74.7%, (p = 0.0001), - 82.1 (p = 0.0015) respectively. The ability of each treatment strategy to achieve the CNO remission was 52.6%, 44.4%, 57,1%, 88.8% and 73.3%, respectively (log-rank test, p = 0.001). The efficacy of treatment approaches for CNO depended on the severity of the disease. NSAID, methotrexate, and sulfasalazine were effective in forms without spine involvement, but pamidronate and TNF-a inhibitors were useful in vertebral forms of CNO. Pamidronate and TNF-a inhibitors more extensively suppressed CNO activity. The randomized controlled trials for assessment of the efficacy and safety of these medications is mandatory to confirm these results.


Assuntos
Anti-Inflamatórios não Esteroides/uso terapêutico , Conservadores da Densidade Óssea/uso terapêutico , Imunossupressores/uso terapêutico , Osteomielite/tratamento farmacológico , Padrões de Prática Médica , Adolescente , Criança , Pré-Escolar , Feminino , Humanos , Masculino , Metotrexato/uso terapêutico , Pamidronato/uso terapêutico , Sulfassalazina/uso terapêutico , Resultado do Tratamento
17.
Int Wound J ; 16(1): 250-255, 2019 Feb.
Artigo em Inglês | MEDLINE | ID: mdl-30393969

RESUMO

Calciphylaxis is a rare and potentially fatal disease that affects the subcutaneous layer of the skin. It is a calcific vasculopathy induced by a systemic process that causes occlusion of small blood vessels. The mortality rate for individuals diagnosed with calciphylaxis is estimated between 52% and 81% with sepsis being the leading cause of death. Uraemic calciphylaxis and its known effective treatments are well documented in the literature. Unfortunately, there is no known effective treatment for non-uraemic calciphylaxis. Most of the current treatments for non-uraemic calciphylaxis are derived from uraemic calciphylaxis treatment protocols. We report a case of a 75-year-old female with calciphylaxis on the right lower extremity who was successfully treated with four pamidronate infusions in addition to local wound care. This case represents a non-uraemic calciphylaxis wound successfully treated with pamidronate infusions and standard wound care, and suggests that IV pamidronate can be an effective treatment option.


Assuntos
Conservadores da Densidade Óssea/uso terapêutico , Calciofilaxia/diagnóstico , Calciofilaxia/tratamento farmacológico , Pamidronato/administração & dosagem , Pamidronato/uso terapêutico , Administração Intravenosa , Idoso , Feminino , Humanos , Resultado do Tratamento
18.
Eur J Health Econ ; 20(4): 487-499, 2019 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-30382484

RESUMO

BACKGROUND: Bone metastases are highly prevalent in breast, prostate, lung and colon cancers. Their symptoms negatively affect quality of life and functionality and optimal management can mitigate these problems. There are two different targeted agents to treat them: bisphosphonates (pamidronate and zoledronic acid) and the monoclonal antibody denosumab. Estimates of cost-effectiveness are still mixed. OBJECTIVE: To conduct a systematic review of economic studies that compares these two options. METHOD: Literature search comprised eight databases and keywords for bone metastases, bisphosphonates, denosumab, and economic studies were used. Data were extracted regarding their methodologic characteristics and cost-effectiveness analyses. All studies were evaluated regarding to its methodological quality. RESULTS: A total of 263 unique studies were retrieved and six met inclusion criteria. All studies were based on clinical trials and other existing literature data, and they had high methodological quality. Most found unfavorable cost-effectiveness for denosumab compared with zoledronic acid, with adjusted ICERS that ranged from $4638-87,354 per SRE avoided and from US$57,274-4.81 M. per QALY gained, which varied widely according to type of tumor, time horizon, among others. Results were sensitive to drug costs, time to first skeletal-related event (SRE), time horizon, and utility. CONCLUSIONS: Denosumab had unfavorable cost-effectiveness compared with zoledronic acid in most of the included studies. New economic studies based on real-world data and longer time horizons comparing these therapeutic options are needed.


Assuntos
Conservadores da Densidade Óssea/uso terapêutico , Neoplasias Ósseas/secundário , Denosumab/uso terapêutico , Difosfonatos/uso terapêutico , Conservadores da Densidade Óssea/economia , Neoplasias Ósseas/tratamento farmacológico , Neoplasias Ósseas/economia , Análise Custo-Benefício , Denosumab/economia , Difosfonatos/economia , Custos de Cuidados de Saúde , Humanos , Pamidronato/economia , Pamidronato/uso terapêutico , Ácido Zoledrônico/economia , Ácido Zoledrônico/uso terapêutico
19.
J Clin Res Pediatr Endocrinol ; 11(2): 140-148, 2019 05 28.
Artigo em Inglês | MEDLINE | ID: mdl-30396880

RESUMO

Objective: No large study has been conducted to date to compare the effectiveness of prednisolone, alendronate and pamidronate as first-line treatment in children with hypercalcemia due to vitamin D intoxication. The aim was to perform a multicenter, retrospective study assessing clinical characteristics and treatment results. Methods: A standard questionnaire was uploaded to an online national database system to collect data on children with hypercalcemia (serum calcium level >10.5 mg/dL) due to vitamin D intoxication [serum 25-hydroxyvitamin D (25(OH)D) level >150 ng/mL] who were treated in pediatric endocrinology clinics. Results: Seventy-four children [median (range) age 1.06 (0.65-1.60) years, 45 males (61%) from 11 centers] were included. High-dose vitamin D intake was evident in 77% of the cases. At diagnosis, serum calcium, phosphorus, alkaline phosphatase, 25(OH)D and parathyroid hormone concentrations were 15±3.2 mg/dL, 5.2±1.2 mg/dL, 268±132 IU/L, 322 (236-454) ng/mL, and 5.5 (3-10.5) pg/mL, respectively. Calcium levels showed moderate correlation with 25(OH)D levels (rs=0.402, p<0.001). Patients were designated into five groups according to the initial specific treatment regimens (hydration-only, prednisolone, alendronate, pamidronate, and combination). Need for another type of specific drug treatment was higher in children who initially received prednisolone (p<0.001). Recurrence rate of hypercalcemia was significantly lower in children who were treated with pamidronate (p=0.02). Conclusion: Prednisolone is less effective in the treatment of children with severe hypercalcaemia secondary to vitamin D intoxication and timely implementation of other treatment regimens should be considered.


Assuntos
Conservadores da Densidade Óssea/uso terapêutico , Hipercalcemia/tratamento farmacológico , Pamidronato/uso terapêutico , Vitamina D/efeitos adversos , Vitaminas/efeitos adversos , Feminino , Seguimentos , Humanos , Hipercalcemia/sangue , Hipercalcemia/induzido quimicamente , Hipercalcemia/patologia , Lactente , Masculino , Prognóstico , Estudos Retrospectivos , Vitamina D/sangue , Vitaminas/sangue
20.
Bone ; 120: 482-486, 2019 03.
Artigo em Inglês | MEDLINE | ID: mdl-30572144

RESUMO

Denosumab is an anti-RANKL antibody that is commonly used for the treatment of osteoporosis; in oncology, bisphosphonates and denosumab have become the standard therapies for the treatment and prevention of skeletal complications in patients with myeloma and solid tumors. In recent years, excessive bone remodeling following the discontinuation of denosumab has raised concerns. Several cases of hypercalcemia have been reported after the discontinuation of high-dose denosumab (120 mg every 4 weeks), mainly in children. In this study, we report a new case of severe refractory hypercalcemia in a 54-year-old woman who received high-dose denosumab for 5 years as an adjuvant therapy for breast cancer. She is currently in remission and undergoing treatment with anastrazole, an aromatase inhibitor. The peculiarities of this case are the presence of associated bone pain with subperiosteal bone resorption on hand X-rays, and diffuse, long bone diaphyseal uptake on a bone scan. Hyperparathyroidism has been ruled out, and existing evidence suggests that this high-level of bone remodeling could be due to a rebound hyperactivation of the RANKL pathway. In addition to rehydration, repeated use of i.v. bisphosphonates was required to control recurrent hypercalcemia. As hypercalcemia is a serious metabolic complication, a gradual dose reduction should be considered when interruption of high dose denosumab therapy is planned.


Assuntos
Reabsorção Óssea/complicações , Hipercalcemia/complicações , Ligante RANK/metabolismo , Doença Aguda , Reabsorção Óssea/diagnóstico por imagem , Reabsorção Óssea/tratamento farmacológico , Osso e Ossos/diagnóstico por imagem , Osso e Ossos/patologia , Feminino , Humanos , Hipercalcemia/diagnóstico por imagem , Hipercalcemia/tratamento farmacológico , Pessoa de Meia-Idade , Pamidronato/uso terapêutico
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