RESUMO
Life-threatening disseminated tuberculosis developed in a 17-year-old girl who was treated with the TNF-α blocker adalimumab for refractory SAPHO syndrome. The patient presented to the emergency department with dyspnea and somnolence and within 2 h developed the clinical picture of a septic shock. In addition to this unusual presentation, she showed a complicated course with increasing cerebral granuloma formation in spite of adequate antimycobacterial treatment. Immune reconstitution after discontinuation of TNF blockade may contribute to this "paradoxical reaction." Possible implications for screening, diagnosis, and treatment of tuberculosis in children and adolescents receiving anti-TNF treatment are discussed.
Assuntos
Síndrome de Hiperostose Adquirida/tratamento farmacológico , Anticorpos Monoclonais Humanizados/efeitos adversos , Hospedeiro Imunocomprometido , Imunossupressores/efeitos adversos , Tuberculose Miliar/diagnóstico , Tuberculose Miliar/imunologia , Fator de Necrose Tumoral alfa/antagonistas & inibidores , Síndrome de Hiperostose Adquirida/imunologia , Adalimumab , Adolescente , Anti-Inflamatórios/efeitos adversos , Antituberculosos/uso terapêutico , Compostos Aza/uso terapêutico , Quimioterapia Combinada , Dispneia/microbiologia , Etambutol/uso terapêutico , Feminino , Fluoroquinolonas , Humanos , Moxifloxacina , Quinolinas/uso terapêutico , Índice de Gravidade de Doença , Choque Séptico/microbiologia , Resultado do Tratamento , Tuberculose Miliar/complicações , Tuberculose Miliar/tratamento farmacológicoRESUMO
OBJECTIVE: To describe the successful treatment of an unusual case of severe neonatal Bartter's syndrome refractory to treatment with indomethacin. DESIGN: Case report, clinical. SETTING: Tertiary care intensive care unit. PATIENTS: A patient with neonatal hyperprostaglandin-E syndrome and excessive requirements of intravenous (via central venous catheter) water and salt supplementation, failure to thrive, vomiting, and massive growth retardation, despite adequate treatment with indomethacin. MAIN RESULT: Four weeks after induction of the new cyclooxygenase-2 inhibitor rofecoxib, the patient was well, on full enteral feeds, thriving, and had gained 600 g in weight. A lower supplementary potassium, magnesium, and sodium intake was required. Reinstitution of indomethacin therapy resulted in severe deterioration, despite high indomethacin doses; symptoms improved again after rofecoxib administration. No side effects have been seen thus far. CONCLUSION: This report shows that in selected patients with a severe form of neonatal Bartter's syndrome, the new cyclooxygenase-2 inhibitor rofecoxib may control the clinical symptoms of hyperprostaglandin-E syndrome after ineffective indomethacin therapy.
