RESUMEN
BACKGROUND: B-cell acute lymphoblastic leukemia (B-ALL) is the most common childhood malignancy. Medullary and extramedullary disease relapse is a well-known occurrence in B-ALL pediatric patients treated with standard chemo-immunotherapy and, more recently, with chimeric antigen receptor (CAR)-T cell therapy. 18F-Fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) emerges as a sensitive imaging tool for detecting disease relapse at extra-medullary sites, with only limited literature evidence in the CAR-T therapy setting. CASE REPORT: In a 12-year-old female treated with CAR-T therapy for B-ALL relapse, 18F-FDG PET/CT scan performed for surveillance, after disease remission, detected a solitary and clinically occult relapse in the breast parenchyma that was histologically confirmed. CONCLUSION: At our knowledge, this is the first report about a pediatric B-ALL patient with a solitary and occult breast relapse after CAR-T therapy, early discovered by 18F-FDG PET/CT during disease monitoring.
Asunto(s)
Fluorodesoxiglucosa F18 , Inmunoterapia Adoptiva , Tomografía Computarizada por Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Humanos , Femenino , Niño , Tomografía Computarizada por Tomografía de Emisión de Positrones/métodos , Inmunoterapia Adoptiva/efectos adversos , Inmunoterapia Adoptiva/métodos , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapia , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Recurrencia Local de Neoplasia/diagnóstico por imagen , Recurrencia Local de Neoplasia/patología , Neoplasias de la Mama/diagnóstico por imagen , Neoplasias de la Mama/terapia , Neoplasias de la Mama/patología , RadiofármacosRESUMEN
18 F-fluorodeoxyglucose-positron emission tomography/computed tomography (PET/CT) imaging is currently not used in standard diagnostics for B-cell precursor lymphoblastic lymphoma (BCP-LBL), and it is unknown whether PET/CT imaging would lead to agreement between detection of lesions with the gold standard imaging methods. Therefore, we performed a retrospective cohort study in which we included 32 pediatric BCP-LBL patients and determined localizations by reviewing local imaging reports. There was a disagreement between protocol-based imaging and PET/CT in 59% of the patients, and the discrepancies mostly comprise of additional lesions detected with PET/CT, typically in lymph node and bone or the absence of bone marrow involvement with PET/CT. If PET/CT was leading in determining definite stage of disease, this would lead to a different stage and therapy branch in 31% and 28% of the patients, respectively.
Asunto(s)
Fluorodesoxiglucosa F18 , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Humanos , Niño , Tomografía Computarizada por Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Estudios Retrospectivos , Diagnóstico por ImagenRESUMEN
ABSTRACT: Extensive extramedullary involvement as presentation is uncommon in pediatric B-cell acute lymphoblastic leukemia. A 7-year-old boy was diagnosed with painless parotid gland enlargement. He had pancytopenia and significantly raised serum lactate dehydrogenase. Fine-needle aspiration cytology from the parotid was suggestive of lymphoid malignancy. Flow cytometry and bone marrow biopsy suggested B-cell acute lymphoblastic leukemia. 18 F-FDG PET/CT revealed extensive bone marrow disease and the involvement of the spleen, pancreas, kidneys, and the parotid, submandibular, and lacrimal glands. He had negligible physiological brain uptake.
Asunto(s)
Tomografía Computarizada por Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Masculino , Humanos , Niño , Médula Ósea/diagnóstico por imagen , Médula Ósea/patología , Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Biopsia con Aguja FinaRESUMEN
BACKGROUND: Chimeric antigen receptor (CAR) T cell therapy is an effective salvage therapy for pediatric relapsed B-cell acute lymphoblastic leukemia (B-ALL), yet is challenged by high rates of post-CAR relapse. Literature describing specific relapse patterns and extramedullary (EM) sites of involvement in the post-CAR setting remains limited, and a clinical standard for post-CAR disease surveillance has yet to be established. We highlight the importance of integrating peripheral blood minimal residual disease (MRD) testing and radiologic imaging into surveillance strategies, to effectively characterize and capture post-CAR relapse. MAIN BODY: Here, we describe the case of a child with multiply relapsed B-ALL who relapsed in the post-CAR setting with gross non-contiguous medullary and EM disease. Interestingly, her relapse was identified first from peripheral blood flow cytometry MRD surveillance, in context of a negative bone marrow aspirate (MRD <0.01%). Positron emission tomography with 18F-fluorodeoxyglucose revealed diffuse leukemia with innumerable bone and lymph node lesions, interestingly sparing her sacrum, the site of her bone marrow aspirate sampling. CONCLUSIONS: We highlight this case as both peripheral blood MRD and 18F-fluorodeoxyglucose positron emission tomography imaging were more sensitive than standard bone marrow aspirate testing in detecting this patient's post-CAR relapse. Clinical/Biologic Insight: In the multiply relapsed B-ALL setting, where relapse patterns may include patchy medullary and/or EM disease, peripheral blood MRD and/or whole body imaging, may carry increased sensitivity at detecting relapse in patient subsets, as compared with standard bone marrow sampling.
Asunto(s)
Linfoma de Burkitt , Leucemia , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Humanos , Niño , Femenino , Fluorodesoxiglucosa F18 , Médula Ósea/diagnóstico por imagen , Neoplasia Residual , Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapiaAsunto(s)
Neoplasias Orbitales , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Preescolar , Femenino , Humanos , Neoplasias Orbitales/diagnóstico por imagen , Neoplasias Orbitales/metabolismo , Neoplasias Orbitales/patología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/metabolismo , Leucemia-Linfoma Linfoblástico de Células Precursoras B/patologíaAsunto(s)
Neoplasias de la Médula Ósea , Subunidad alfa 2 del Factor de Unión al Sitio Principal , Proteínas de Fusión Oncogénica , Huesos Pélvicos , Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Neoplasias de la Médula Ósea/diagnóstico por imagen , Neoplasias de la Médula Ósea/genética , Neoplasias de la Médula Ósea/metabolismo , Neoplasias de la Médula Ósea/terapia , Niño , Subunidad alfa 2 del Factor de Unión al Sitio Principal/genética , Subunidad alfa 2 del Factor de Unión al Sitio Principal/metabolismo , Femenino , Humanos , Proteínas de Fusión Oncogénica/genética , Proteínas de Fusión Oncogénica/metabolismo , Huesos Pélvicos/diagnóstico por imagen , Huesos Pélvicos/metabolismo , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/genética , Leucemia-Linfoma Linfoblástico de Células Precursoras B/metabolismo , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapiaAsunto(s)
Infiltración Leucémica/diagnóstico por imagen , Nervio Óptico/patología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Recuperación de la Función , Trastornos de la Visión/diagnóstico , Anciano , Humanos , Infiltración Leucémica/fisiopatología , Infiltración Leucémica/radioterapia , Imagen por Resonancia Magnética , Masculino , Recurrencia Local de Neoplasia , Papiledema/fisiopatología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/fisiopatología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/radioterapia , Trastornos de la Pupila/fisiopatología , Trastornos de la Visión/fisiopatologíaRESUMEN
Chimeric antigen receptor T cells (CAR-T) are an effective and potentially durable treatment for refractory and multiply relapsed B-cell acute lymphoblastic leukemia. Neurotoxicity is frequent after CAR-T cell therapy. Mechanisms driving neurotoxicity are incompletely understood, and symptoms can range from transient and mild to severe and life-threatening. Providers have exercised caution in providing CAR-T to patients with neurological comorbidities or extramedullary disease. Here, we report three patients with prior significant neurologic morbidity who safely tolerated CAR-T cell infusion after bridging therapy with conventional chemotherapy.
Asunto(s)
Inmunoterapia Adoptiva/efectos adversos , Enfermedades del Sistema Nervioso , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Adolescente , Niño , Preescolar , Comorbilidad , Humanos , Masculino , Enfermedades del Sistema Nervioso/diagnóstico por imagen , Enfermedades del Sistema Nervioso/etiología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapiaRESUMEN
Broken catheter embolism is a rare but often fatal complication of implantable venous access devices. Prompt removal is key to avoiding an adverse outcome.
Asunto(s)
Remoción de Dispositivos , Embolización Terapéutica/efectos adversos , Falla de Equipo , Migración de Cuerpo Extraño , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Tomografía Computarizada por Rayos X , Niño , Migración de Cuerpo Extraño/diagnóstico por imagen , Migración de Cuerpo Extraño/terapia , Humanos , Masculino , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapia , Arteria PulmonarRESUMEN
A 4-year-old boy with history of hemophilia B presented with increasingly enlarged scalp masses. Although they were initially thought as hematomas, unresponsiveness to the therapy lead to suspicion of malignancy, which prompted FDG PET/CT. The FDG PET/CT images demonstrated increased FDG uptake in the scalp masses and cervical lymph nodes. The pathology from the left scalp mass and the left cervical lymph nodes revealed precursor B-cell lymphoblastic lymphoma.
Asunto(s)
Hemofilia B/complicaciones , Tomografía Computarizada por Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Preescolar , Fluorodesoxiglucosa F18 , Humanos , Masculino , Leucemia-Linfoma Linfoblástico de Células Precursoras B/complicaciones , RadiofármacosAsunto(s)
Insuficiencia Cardíaca , Neoplasias Cardíacas , Imagen por Resonancia Magnética , Trasplante de Células Madre de Sangre Periférica , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Resultado Fatal , Femenino , Insuficiencia Cardíaca/diagnóstico por imagen , Insuficiencia Cardíaca/etiología , Neoplasias Cardíacas/diagnóstico por imagen , Neoplasias Cardíacas/terapia , Humanos , Persona de Mediana Edad , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapiaAsunto(s)
Corticoesteroides/administración & dosificación , Anticuerpos Monoclonales Humanizados/administración & dosificación , Trastornos de la Coagulación Sanguínea , Inmunoterapia Adoptiva/efectos adversos , Síndromes de Neurotoxicidad , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Trastornos de la Coagulación Sanguínea/diagnóstico por imagen , Trastornos de la Coagulación Sanguínea/tratamiento farmacológico , Trastornos de la Coagulación Sanguínea/etiología , Niño , Humanos , Masculino , Síndromes de Neurotoxicidad/diagnóstico por imagen , Síndromes de Neurotoxicidad/tratamiento farmacológico , Síndromes de Neurotoxicidad/etiología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/terapiaRESUMEN
Acute lymphoblastic lymphoma is a malignant hematologic disease in childhood but rarely initially involves epidural compartment in adults. A 20-year-old male presented with progressive osphyalgia with constipation. Contrasted magnetic resonance imaging showed multiple vertebrae of hypointense T1 signals and an intraspinal epidural lesion. Subtotal resection was achieved. Histopathology suggested malignant B-cell lymphoma with Ki-67 of 90% and positivity of leukocyte common antigen. A bone marrow biopsy was unequivocally diagnostic of B-cell acute lymphoblastic lymphoma followed by chemotherapy (methotrexate) and partial recovery was observed. The marrow biopsy was necessary if without hypercalcemia and abnormal peripheral blood examination.
Asunto(s)
Leucemia-Linfoma Linfoblástico de Células Precursoras B/cirugía , Neoplasias de la Columna Vertebral/cirugía , Columna Vertebral/cirugía , Antimetabolitos Antineoplásicos/uso terapéutico , Terapia Combinada , Humanos , Imagen por Resonancia Magnética , Masculino , Metotrexato/uso terapéutico , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/tratamiento farmacológico , Neoplasias de la Columna Vertebral/diagnóstico por imagen , Neoplasias de la Columna Vertebral/tratamiento farmacológico , Columna Vertebral/diagnóstico por imagen , Adulto JovenAsunto(s)
Subunidad alfa 2 del Factor de Unión al Sitio Principal/genética , Amplificación de Genes , Proteínas de Neoplasias/genética , Leucemia-Linfoma Linfoblástico de Células Precursoras B , Neoplasias de la Base del Cráneo , Adulto , Humanos , Masculino , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/genética , Neoplasias de la Base del Cráneo/diagnóstico por imagen , Neoplasias de la Base del Cráneo/genéticaRESUMEN
A 4-year old girl presenting gait difficulty was referred for spine X-ray and Magnetic Resonance Imaging (MRI). MRI showed several diffuse hypointense signals in sacral and lumbar vertebrae. In order to exclude a possible lymphoproliferative disease a 18-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG-PET/CT) was requested. The PET/CT scan confirmed the MRI findings and demonstrated additional findings in the sternum. Therefore, a bone marrow biopsy was performed and a diagnosis of acute lymphoblastic leukaemia - early B type was made.
Asunto(s)
Neoplasias Óseas/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Preescolar , Femenino , Fluorodesoxiglucosa F18 , Humanos , Imagen por Resonancia Magnética , Imagen Multimodal , Tomografía Computarizada por Tomografía de Emisión de Positrones , RadiofármacosRESUMEN
An 11-year-old girl was admitted for further investigation as to the cause of her bilateral papilloedema and periorbital swelling. She had a 2-week history of headache and unilateral eyelid swelling, and a 2-day history of right-sided groin swelling. CT and MRI scans revealed soft tissue adjacent to the lateral orbital walls within the extraconal lateral aspects of both orbits, more on the right than the left. The scans also revealed extensive lymphadenopathy above and below the diaphragm. The patient underwent bone marrow studies and biopsy of the lymph node in her groin. The results revealed normal bone marrow with no evidence of malignancy. The lymph node histology confirmed malignant lymphoma in keeping with B cell lymphoblastic lymphoma. The patient was started on the UKALL 2011 chemotherapy trial.
Asunto(s)
Protocolos de Quimioterapia Combinada Antineoplásica/administración & dosificación , Neoplasias del Ojo/diagnóstico por imagen , Neoplasias del Ojo/tratamiento farmacológico , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/tratamiento farmacológico , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Niño , Neoplasias del Ojo/patología , Femenino , Humanos , Ganglios Linfáticos/patología , Imagen por Resonancia Magnética/métodos , Leucemia-Linfoma Linfoblástico de Células Precursoras B/patología , Tomografía Computarizada por Rayos X/métodos , Resultado del TratamientoAsunto(s)
Médula Ósea/diagnóstico por imagen , Vértebras Lumbares/diagnóstico por imagen , Fracturas Osteoporóticas/etiología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Sacro/diagnóstico por imagen , Fracturas de la Columna Vertebral/etiología , Adulto , Protocolos de Quimioterapia Combinada Antineoplásica/uso terapéutico , Médula Ósea/patología , Examen de la Médula Ósea , Fluorodesoxiglucosa F18 , Humanos , Dolor de la Región Lumbar/etiología , Dolor de la Región Lumbar/prevención & control , Vértebras Lumbares/lesiones , Vértebras Lumbares/cirugía , Imagen por Resonancia Magnética , Masculino , Imagen Multimodal , Fracturas Osteoporóticas/fisiopatología , Fracturas Osteoporóticas/cirugía , Tomografía de Emisión de Positrones , Leucemia-Linfoma Linfoblástico de Células Precursoras B/tratamiento farmacológico , Leucemia-Linfoma Linfoblástico de Células Precursoras B/patología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/fisiopatología , Radiofármacos , Inducción de Remisión , Sacro/lesiones , Sacro/patología , Fracturas de la Columna Vertebral/fisiopatología , Fracturas de la Columna Vertebral/cirugía , Fusión Vertebral , Imagen de Cuerpo EnteroRESUMEN
Lymphomas showing both MYC/8q24 rearrangement and IGH@BCL2/t(14;18)(q32;q21), also referred to as "double-hit" or "dual-hit" lymphomas (DHL) are rare B-cell malignancies with a germinal center B-cell immunophenotype and heterogeneous cytologic and histologic features. Such lymphomas may arise de novo or through transformation of follicular lymphomas and are classified either as "B-cell lymphoma, unclassifiable, with features intermediate between diffuse large B-cell lymphoma (DLBCL) and Burkitt lymphoma (BL)" (most commonly), DLBCL, or, rarely, as B-lymphoblastic lymphoma. We report a case of B-lymphobastic lymphoma arising through transformation of follicular lymphoma diagnosed on peritoneal fluid cytology, flow cytometry, and cytogenetic studies in a 53-year-old man who presented with abdominal pain, shortness of breath, night sweats, extensive lymphadenopathy, pleural effusion, and ascites. Cytologic examination of the ascitic fluid showed two distinct populations of neoplastic lymphoid cells, a predominant population of larger cells with fine powdery ("blastic") chromatin, visible to prominent nucleoli and occasional small cytoplasmic vacuoles and a less numerous population of smaller cells with centrocytic morphology. Flow cytometry also showed two distinct monotypic B-cell populations, both expressing CD10, and TdT-positivity was demonstrated immunohistochemically. Fluorescence in situ hybridization (FISH) demonstrated both MYC rearrangement and IGH/BCL2 gene fusion and cytogenetic analysis showed a complex karyotype including both t(14;18)(q32;q21) and t(8;22)(q24.1;q11.2). Since DHL pursue an aggressive clinical course, respond poorly to therapy, and have a poor outcome, it is important to suspect the diagnosis when encountering neoplastic lymphoid cells that are difficult to classify in effusion cytology specimens and to order the appropriate immunophenotyping and cytogenetic studies.
Asunto(s)
Linfoma Folicular/diagnóstico por imagen , Derrame Pleural Maligno/diagnóstico por imagen , Leucemia-Linfoma Linfoblástico de Células Precursoras B/diagnóstico por imagen , Antígenos CD/metabolismo , Líquido Ascítico/patología , Humanos , Linfoma Folicular/metabolismo , Linfoma Folicular/patología , Masculino , Persona de Mediana Edad , Derrame Pleural Maligno/metabolismo , Derrame Pleural Maligno/patología , Leucemia-Linfoma Linfoblástico de Células Precursoras B/metabolismo , Leucemia-Linfoma Linfoblástico de Células Precursoras B/patología , RadiografíaRESUMEN
Hypothalamic obesity syndrome can affect brain tumor patients following surgical intervention and irradiation. This syndrome is rare at diagnosis in childhood cancer, but has been reported with relapse of acute lymphoblastic leukemia. Here we present a case of hypothalamic obesity syndrome as the primary presentation of a toddler found to have CNS+ B-cell lymphoblastic lymphoma. Cytogenetic studies on diagnostic cerebrospinal fluid revealed MLL gene rearrangement (11q23). Hyperphagia and obesity dramatically improved following induction and consolidation chemotherapy. We describe a novel presentation of hypothalamic obesity syndrome in CNS B-cell lymphoblastic lymphoma, responsive to chemotherapy.