is a multiflagellated protozoon which parasitizes the gut of termites and cockroaches. Although L. infection is rare, it can affect lung, maxillary sinuses and genitourinary tract. The presentation of bronchopulmonary lophomonas includes nonspecific symptoms such as fever, cough and dyspnea. Diagnosis is based on identification of living protozoan forms in fresh samples from respiratory secretions (bronchoalveolar lavage). We report the case of a 2-year-old male with a history of severe combined immunodeficiency (T-, B-, NK-), post-hematopoietic stem cell transplant and full immune reconstitution 12 months following a successful transplant who thereafter presented lophomonas.
Severe combined immunodeficiency (SCID) represents the most lethal form of primary immunodeficiency, with mortality rates of greater than 90% within the first year of life without treatment. Hematopoietic stem cell transplantation and gene therapy are the only curative treatments available, and the best-known prognostic factors for success are age at diagnosis, age at hematopoietic stem cell transplantation, and the comorbidities that develop in between. There are no evidence-based guidelines for standardized clinical care for patients with SCID during the time between diagnosis and definitive treatment, and we aim to generate a consensus management strategy on the supportive care of patients with SCID. First, we gathered available information about SCID diagnostic and therapeutic guidelines, then we developed a document including diagnostic and therapeutic interventions, and finally we submitted the interventions for expert consensus through a modified Delphi technique. Interventions are grouped in 10 topic domains, including 123 ''agreed'' and 38 ''nonagreed'' statements. This document intends to standardize supportive clinical care of patients with SCID from diagnosis to definitive treatment, reduce disease burden, and ultimately improve prognosis, particularly in countries where newborn screening for SCID is not universally available and delayed diagnosis is the rule. Our work intends to provide a tool not only for immunologists but also for primary care physicians and other specialists involved in the care of patients with SCID. (J Allergy Clin Immunol 2019;144:897-905.)
ANTECEDENTES: Las inmunodeficiencias primarias (IDP) son trastornos genéticos que afectan el sistema inmune. Aproximadamente 85% de los pacientes con IDP requieren terapia con inmunoglobulina. El retraso en el diagnóstico y tratamiento genera complicaciones clínicas y gastos para el sistema de salud.OBJETIVO: Describir la carga económica que representan las IDP tratadas con IG, y comparar el impacto presupuestal antes y después de tratamiento.MATERIAL Y MÉTODOS: Evaluación económica parcial tipo “descripción de costos” y análisis de impacto presupuestal de pacientes con IDP antes y después del tratamiento con IG. El método de recolección y descripción de costos fue mixto: micro-costeo de 37 expedientes seguido de la estimación “descendente” de costos y análisis de impacto presupuestal sobre el cúmulo de 109 casos de pacientes con IDP en seguimiento activo.RESULTADOS: El costo promedio total por infecciones por paciente por año antes de reemplazo con IG fue de $629,299 vs $155,546 tras el inicio de IG, reduciéndose 75%. La neumonía fue la complicación prevenible más costosa y el costo total por infecciones estimado para 109 pacientes antes del diagnóstico fue $ 68.5 millones vs $ 36 millones posterior al inicio de IG. El costo per cápita antes de tratamiento con IG se estimó en $3.96 vs $2.10 posterior al tratamiento.CONCLUSIONES: El diagnóstico y tratamiento oportuno de las IDP con IG disminuye el costo de la atención clínica en $1.87 pesos mexicanos per capita (3.96 vs 2.10 MXN; p < 0.001), representando un impacto presupuestal con ahorro estimado del 47% anual.
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