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Estudio de 9 pacientes" "tieneTextoCompleto" => 0 "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "339" "paginaFinal" => "342" ] ] "autores" => array:2 [ 0 => array:4 [ "autoresLista" => "Javier Grau, Josep-Maria Ribera, José-María Indiano, Javier Vercher, Virgilio Sandoval, Gemma Ramírez, Ana Sastre, Eduardo Flores, Javier García-Conde" "autores" => array:9 [ 0 => array:3 [ "nombre" => "Javier" "apellidos" => "Grau" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:4 [ "nombre" => "Josep-Maria" "apellidos" => "Ribera" "email" => array:1 [ 0 => "jmribera@ns.hugtip.scs.es" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 2 => array:3 [ "nombre" => "José-María" "apellidos" => "Indiano" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "Javier" "apellidos" => "Vercher" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] 4 => array:3 [ "nombre" => "Virgilio" "apellidos" => "Sandoval" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0025" ] ] ] 5 => array:3 [ "nombre" => "Gemma" "apellidos" => "Ramírez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">f</span>" "identificador" => "aff0030" ] ] ] 6 => array:3 [ "nombre" => "Ana" "apellidos" => "Sastre" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">g</span>" "identificador" => "aff0035" ] ] ] 7 => array:3 [ "nombre" => "Eduardo" "apellidos" => "Flores" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">h</span>" "identificador" => "aff0040" ] ] ] 8 => array:3 [ "nombre" => "Javier" "apellidos" => "García-Conde" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:8 [ 0 => array:3 [ "entidad" => "Hospital Universitari Germans Trias i Pujol. Badalona. Barcelona" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Hospital Clínico Universitario.Valencia" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Hospital de Basurto. Bilbao." "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Hospital Can Misses. Ibiza" "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] 4 => array:3 [ "entidad" => "Hospital Virgen Blanca.León" "etiqueta" => "<span class="elsevierStyleSup">e</span>" "identificador" => "aff0025" ] 5 => array:3 [ "entidad" => "Hospital Virgen de la Victoria." "etiqueta" => "<span class="elsevierStyleSup">f</span>" "identificador" => "aff0030" ] 6 => array:3 [ "entidad" => "Hospital La Paz. Madrid." "etiqueta" => "<span class="elsevierStyleSup">g</span>" "identificador" => "aff0035" ] 7 => array:3 [ "entidad" => "Hospital Gregorio Marañón. Madrid" "etiqueta" => "<span class="elsevierStyleSup">h</span>" "identificador" => "aff0040" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "*" "correspondencia" => "Correspondencia: Servicio de Hematología-Hemoterapia. Unitat Hematooncològica. Hospital Universitari Germans Trias i Pujol. Carretera de Canyet, s/n. 08916 Badalona. Barcelona." ] ] ] 1 => array:2 [ "autoresLista" => "Mar Tormo" "autores" => array:1 [ 0 => array:2 [ "nombre" => "Mar" "apellidos" => "Tormo" ] ] ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Results of treatment with 2-chlorodeoxyadenosine in refractory or relapsed.langerhans cell histiocytosis. study of 9 patients." ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2000-12-30" "fechaAceptado" => "2001-02-01" "PalabrasClave" => array:2 [ "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec220870" "palabras" => array:4 [ 0 => "Histiocitosis de células de Langerhans (HCL)" 1 => "Refractaria" 2 => "Recaída" 3 => "2-clorodesoxiadenosina (2CdA)" ] ] ] "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Key words" "identificador" => "xpalclavsec220869" "palabras" => array:4 [ 0 => "Langerhans cell histiocytosis (LCH)" 1 => "Refractory" 2 => "Relapsed" 3 => "2-chlorodeoxyadenosine (2-CdA)" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "es" => array:1 [ "resumen" => "<span class="elsevierStyleSectionTitle">Fundamento</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Analizar los resultados del tratamiento con 2-clorodesoxiadenosina (2CdA) en pacientes diagnosticados de histiocitosis de células de Langerhans (HCL) multisistémica, refractaria o en recaída,atendidos en 8 hospitales españoles entre 1993 y 1999</p> <span class="elsevierStyleSectionTitle">Pacientes y método</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Se recogieron los siguientes datos de los 9 pacientes: edad, sexo, afección orgánica por la HCL, tratamiento inicial y respuesta al mismo, dosis, número de ciclos y forma de administración de la 2CdA, respuesta al tratamiento con 2CdA, toxicidad, supervivencia libre de enfermedad y supervivencia global</p> <span class="elsevierStyleSectionTitle">Resultados</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">La edad mediana era de 25 años (límites, 6-63). Todos ellos presentaban afección multiorgánica por la HCL, con disfunción grave de órganos en 4 casos. La 2CdA se administrócomo tratamientode segunda línea en 7 casos y de tercera línea en dos. La dosis de 2CdA fue de 0,1 mg/kg y día durante 5 días en la mayoría de los pacientes, con un intervalo de 4 semanas entre los ciclos. Hubo respuesta completa (RC) en dos casos y parcial (RP) en 4 (respuesta global del 66%percnt;). La principal toxicidad fue la hematológica, con neutropenia de grado superior a 2 en 5 casos y trombocitopenia mayor de 2 en 5. Cuatro pacientes presentaron infecciones, con evolución fatal en uno de ellos. Tras una mediana de seguimiento de 8 meses (intervalo, 2-17), dos enfermos se hallan en RC (12 mesesen ambos), 4 en RP (intervalo, 2 12meses) y uno con enfermedad activa (17 meses). Los dos restantes fallecieron por progression de la enfermedad y sepsis por Aspergillus spp., respectivamente. Las probabilidades actuariales de supervivencia libre de enfermedad y supervivencia global al año fueron del 58%percnt; (intervalo de confianza [IC] del 95%percnt;, 38-78%percnt;) y del 71%percnt; (IC del 95%percnt;, 54-88%percnt;), respectivamente</p> <span class="elsevierStyleSectionTitle">Conclusiones</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La 2CdA es un fármaco eficaz en pacientes con HCL refractaria o en recaída, y su efecto tóxico principal es la mielodepresión. La utilidad de la 2CdA, aislada o en combinación con otros fármacos, en pacientes con HCL multisistémica, refractaria o en recaída, debe demostrarse en estudios controlados</p>" ] "en" => array:1 [ "resumen" => "<span class="elsevierStyleSectionTitle">Background</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">To analyse the results of the treatment with 2-chlorodeoxyadenosine (2CdA) in patients with refractory or relapsed Langerhans cell histiocytosis (LCH) tracted in 8 Spanish hospitals between 1993 and 1999</p> <span class="elsevierStyleSectionTitle">Patients and method</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">In the 9 patients the following data were recorded: age, sex, organ involvement by LCH, first treatment and response, dose, number of cycles and schedule of administration 2CdA, response to 2CdA treatment, toxicity, disease-free survival (DFS) and overall survival (OS)</p> <span class="elsevierStyleSectionTitle">Results</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">Median age was 25 years (range, 6-63). All patients had multiorganic involvement by LCH, with severe organ dysfunction in 4. 2CdA was administered as second line treatment in 7 cases and as third line in 2. The 2CdA dose was 0.1 mg/kg per day during 5 days in the majority of patients, and interval between cycles was 4 weeks. In 2 cases a complete remission (CR) was achieved and 4 cases attained a partial remission (PR) (overall response rate 66%percnt;). The main toxicity was hematologic, with neutropenia grade > 2 in 5 cases and thrombocytopenia > 2 in 5. Four patients had infections, with fatal evolution in one of them. After a median follow-up of 8 months (range 2-17), 2 patients remained in CR (12 months both), 4 in PR (range 2-12 months) and one had active disease (17 months). The other two died due to progressive disease and Aspergillus spp. sepsis, respectively. The actuarial probabilities of DFS and OS were 58%percnt; (95%percnt; CI, 38-78%percnt;) and 71%percnt; (95%percnt; CI, 54-88%percnt;), respectively</p> <span class="elsevierStyleSectionTitle">Conclusions</span><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">2CdA is an active drug for patients with refractory or relapsed LCH, and its main toxicity is myelosuppression. The usefulness of 2CdA, isolated or in combination with other drugs, in patients with refractory or relapsed LCH must be assessed in controlled studies</p>" ] ] "bibliografia" => array:2 [ "titulo" => "Referencias Bibliográficas" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:28 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1." 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Journal Information
Nota ClíNica
Resultados del tratamiento con 2-clorodesoxiadenosina en la histiocitosis de células de langerhans resistente o en recaída. Estudio de 9 pacientes
Results of treatment with 2-chlorodeoxyadenosine in refractory or relapsed.langerhans cell histiocytosis. study of 9 patients.
Javier Graua, Josep-Maria Riberaa,
, José-María Indianoc, Javier Vercherd, Virgilio Sandovale, Gemma Ramírezf, Ana Sastreg, Eduardo Floresh, Javier García-Condeb
Corresponding author
jmribera@ns.hugtip.scs.es
Correspondencia: Servicio de Hematología-Hemoterapia. Unitat Hematooncològica. Hospital Universitari Germans Trias i Pujol. Carretera de Canyet, s/n. 08916 Badalona. Barcelona.
Correspondencia: Servicio de Hematología-Hemoterapia. Unitat Hematooncològica. Hospital Universitari Germans Trias i Pujol. Carretera de Canyet, s/n. 08916 Badalona. Barcelona.
a Hospital Universitari Germans Trias i Pujol. Badalona. Barcelona
b Hospital Clínico Universitario.Valencia
c Hospital de Basurto. Bilbao.
d Hospital Can Misses. Ibiza
e Hospital Virgen Blanca.León
f Hospital Virgen de la Victoria.
g Hospital La Paz. Madrid.
h Hospital Gregorio Marañón. Madrid
Mar Tormo