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1.
Br J Haematol ; 196(4): 984-994, 2022 02.
Artículo en Inglés | MEDLINE | ID: mdl-34799853

RESUMEN

Erdheim-Chester disease (ECD) is a rare histiocytic neoplasm with frequent multiorgan involvement. An accurate diagnosis of ECD requires the correlation of clinical features, histopathologic and radiologic findings. We describe a case series of patients with a referral diagnosis of ECD, whereby the diagnosis was changed to non-histiocytic diseases after comprehensive review at a tertiary care center. This accurate revision of the referral diagnosis of ECD enabled initiation of proper disease-directed therapy in a timely manner for these patients and avoided unnecessary exposure to systemic cytotoxic chemotherapy or targeted agents. Our study highlights the value of a multidisciplinary team of histiocytosis experts in confirming the diagnosis of ECD and also brings attention to other conditions to consider that can mimic ECD, including osteopoikilosis, tenosynovial giant cell tumour, IgG4-related disease, fibrous dysplasia and chronic recurrent multifocal osteomyelitis.


Asunto(s)
Enfermedad de Erdheim-Chester/diagnóstico , Adulto , Anciano , Enfermedad de Erdheim-Chester/patología , Femenino , Humanos , Masculino , Persona de Mediana Edad
2.
Am J Hematol ; 94(3): 291-298, 2019 03.
Artículo en Inglés | MEDLINE | ID: mdl-30516868

RESUMEN

Vitreoretinal lymphoma (VRL) management remains a challenge. We present 72 patients with VRL, diagnosed at Mayo Clinic between 1990-2018. Three nondiffuse large B-cell lymphoma (DLBCL) histology cases were excluded. Among 69 DLBCL, 33 patients had primary VRL (PVRL), 18 concurrent intraocular and central nervous system (CNS) or systemic disease and 18 secondary VRL. Patients received intraocular chemotherapy (intraocular injections of rituximab or metothrexate or steroids or in combination), radiotherapy, systemic or combined systemic plus intraocular treatment in 9, 10, 35, and 15 cases, respectively. Among primary and concurrent VRL, median failure free survival (FFS), CNS relapse-free survival (CNS-RFS) and overall survival (OS) were: 1.8, 4.9, and 4.1 years, respectively; among PVRL, median FFS, CNS-RFS, and OS were: 2.6 year, Not Reached and 9.3 year, respectively. No CNS relapse occurred beyond 4 years in PVRL. Median OS for patients diagnosed between 1990 and 1999 vs between 2000 and 2018 was 1.5 vs 9.4 years, respectively (P = .0002). OS was significantly higher in PVRL, as compared with concurrent VRL (P = .04). Previous immunosuppression and poor performance status were predictive of worse outcome. In PVRL, a combined systemic and intraocular therapy showed higher FFS (P = .002) and CNS-RFS (P = .003), but no differences in OS. Among 18 secondary VRL, at a median follow-up of 1.1 year after vitreoretinal relapse, median FFS and OS were 0.3 and 1.3 years. An improvement in survival of VRL has been observed over the decades. PVRL should undergo combined systemic and intraocular chemotherapy to prevent CNS progression.


Asunto(s)
Antineoplásicos Inmunológicos/uso terapéutico , Terapia Combinada/métodos , Linfoma/terapia , Metotrexato/uso terapéutico , Neoplasias de la Retina/terapia , Rituximab/uso terapéutico , Adulto , Anciano , Anciano de 80 o más Años , Femenino , Rayos gamma/uso terapéutico , Humanos , Inyecciones Intravenosas , Inyecciones Intravítreas , Linfoma/mortalidad , Linfoma/patología , Masculino , Persona de Mediana Edad , Recurrencia , Retina/efectos de los fármacos , Retina/patología , Retina/efectos de la radiación , Neoplasias de la Retina/mortalidad , Neoplasias de la Retina/patología , Estudios Retrospectivos , Análisis de Supervivencia , Resultado del Tratamiento , Cuerpo Vítreo/efectos de los fármacos , Cuerpo Vítreo/patología , Cuerpo Vítreo/efectos de la radiación
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