ABSTRACT
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.
Subject(s)
Fluorodeoxyglucose F18 , Immunotherapy, Adoptive , Positron Emission Tomography Computed Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Humans , Female , Child , Positron Emission Tomography Computed Tomography/methods , Immunotherapy, Adoptive/adverse effects , Immunotherapy, Adoptive/methods , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapy , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Neoplasm Recurrence, Local/diagnostic imaging , Neoplasm Recurrence, Local/pathology , Breast Neoplasms/diagnostic imaging , Breast Neoplasms/therapy , Breast Neoplasms/pathology , RadiopharmaceuticalsABSTRACT
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.
Subject(s)
Fluorodeoxyglucose F18 , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Humans , Child , Positron Emission Tomography Computed Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Retrospective Studies , Diagnostic ImagingABSTRACT
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.
Subject(s)
Positron Emission Tomography Computed Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Male , Humans , Child , Bone Marrow/diagnostic imaging , Bone Marrow/pathology , Positron-Emission Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Biopsy, Fine-NeedleABSTRACT
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.
Subject(s)
Burkitt Lymphoma , Leukemia , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Humans , Child , Female , Fluorodeoxyglucose F18 , Bone Marrow/diagnostic imaging , Neoplasm, Residual , Positron-Emission Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapySubject(s)
Orbital Neoplasms , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Child, Preschool , Female , Humans , Orbital Neoplasms/diagnostic imaging , Orbital Neoplasms/metabolism , Orbital Neoplasms/pathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/metabolism , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/pathologySubject(s)
Bone Marrow Neoplasms , Core Binding Factor Alpha 2 Subunit , Oncogene Proteins, Fusion , Pelvic Bones , Positron-Emission Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Bone Marrow Neoplasms/diagnostic imaging , Bone Marrow Neoplasms/genetics , Bone Marrow Neoplasms/metabolism , Bone Marrow Neoplasms/therapy , Child , Core Binding Factor Alpha 2 Subunit/genetics , Core Binding Factor Alpha 2 Subunit/metabolism , Female , Humans , Oncogene Proteins, Fusion/genetics , Oncogene Proteins, Fusion/metabolism , Pelvic Bones/diagnostic imaging , Pelvic Bones/metabolism , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/genetics , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/metabolism , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapySubject(s)
Leukemic Infiltration/diagnostic imaging , Optic Nerve/pathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Recovery of Function , Vision Disorders/diagnosis , Aged , Humans , Leukemic Infiltration/physiopathology , Leukemic Infiltration/radiotherapy , Magnetic Resonance Imaging , Male , Neoplasm Recurrence, Local , Papilledema/physiopathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/physiopathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/radiotherapy , Pupil Disorders/physiopathology , Vision Disorders/physiopathologyABSTRACT
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.
Subject(s)
Immunotherapy, Adoptive/adverse effects , Nervous System Diseases , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Adolescent , Child , Child, Preschool , Comorbidity , Humans , Male , Nervous System Diseases/diagnostic imaging , Nervous System Diseases/etiology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapyABSTRACT
Broken catheter embolism is a rare but often fatal complication of implantable venous access devices. Prompt removal is key to avoiding an adverse outcome.
Subject(s)
Device Removal , Embolization, Therapeutic/adverse effects , Equipment Failure , Foreign-Body Migration , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Tomography, X-Ray Computed , Child , Foreign-Body Migration/diagnostic imaging , Foreign-Body Migration/therapy , Humans , Male , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapy , Pulmonary ArteryABSTRACT
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.
Subject(s)
Hemophilia B/complications , Positron Emission Tomography Computed Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Child, Preschool , Fluorodeoxyglucose F18 , Humans , Male , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/complications , RadiopharmaceuticalsSubject(s)
Heart Failure , Heart Neoplasms , Magnetic Resonance Imaging , Peripheral Blood Stem Cell Transplantation , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Fatal Outcome , Female , Heart Failure/diagnostic imaging , Heart Failure/etiology , Heart Neoplasms/diagnostic imaging , Heart Neoplasms/therapy , Humans , Middle Aged , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapySubject(s)
Adrenal Cortex Hormones/administration & dosage , Antibodies, Monoclonal, Humanized/administration & dosage , Blood Coagulation Disorders , Immunotherapy, Adoptive/adverse effects , Neurotoxicity Syndromes , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Blood Coagulation Disorders/diagnostic imaging , Blood Coagulation Disorders/drug therapy , Blood Coagulation Disorders/etiology , Child , Humans , Male , Neurotoxicity Syndromes/diagnostic imaging , Neurotoxicity Syndromes/drug therapy , Neurotoxicity Syndromes/etiology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/therapyABSTRACT
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.
Subject(s)
Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/surgery , Spinal Neoplasms/surgery , Spine/surgery , Antimetabolites, Antineoplastic/therapeutic use , Combined Modality Therapy , Humans , Magnetic Resonance Imaging , Male , Methotrexate/therapeutic use , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/drug therapy , Spinal Neoplasms/diagnostic imaging , Spinal Neoplasms/drug therapy , Spine/diagnostic imaging , Young AdultSubject(s)
Core Binding Factor Alpha 2 Subunit/genetics , Gene Amplification , Neoplasm Proteins/genetics , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma , Skull Base Neoplasms , Adult , Humans , Male , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/genetics , Skull Base Neoplasms/diagnostic imaging , Skull Base Neoplasms/geneticsABSTRACT
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.
Subject(s)
Bone Neoplasms/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Child, Preschool , Female , Fluorodeoxyglucose F18 , Humans , Magnetic Resonance Imaging , Multimodal Imaging , Positron Emission Tomography Computed Tomography , RadiopharmaceuticalsABSTRACT
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.
Subject(s)
Antineoplastic Combined Chemotherapy Protocols/administration & dosage , Eye Neoplasms/diagnostic imaging , Eye Neoplasms/drug therapy , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/drug therapy , Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Child , Eye Neoplasms/pathology , Female , Humans , Lymph Nodes/pathology , Magnetic Resonance Imaging/methods , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/pathology , Tomography, X-Ray Computed/methods , Treatment OutcomeSubject(s)
Bone Marrow/diagnostic imaging , Lumbar Vertebrae/diagnostic imaging , Osteoporotic Fractures/etiology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Sacrum/diagnostic imaging , Spinal Fractures/etiology , Adult , Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Bone Marrow/pathology , Bone Marrow Examination , Fluorodeoxyglucose F18 , Humans , Low Back Pain/etiology , Low Back Pain/prevention & control , Lumbar Vertebrae/injuries , Lumbar Vertebrae/surgery , Magnetic Resonance Imaging , Male , Multimodal Imaging , Osteoporotic Fractures/physiopathology , Osteoporotic Fractures/surgery , Positron-Emission Tomography , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/drug therapy , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/pathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/physiopathology , Radiopharmaceuticals , Remission Induction , Sacrum/injuries , Sacrum/pathology , Spinal Fractures/physiopathology , Spinal Fractures/surgery , Spinal Fusion , Whole Body ImagingABSTRACT
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.
Subject(s)
Lymphoma, Follicular/diagnostic imaging , Pleural Effusion, Malignant/diagnostic imaging , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/diagnostic imaging , Antigens, CD/metabolism , Ascitic Fluid/pathology , Humans , Lymphoma, Follicular/metabolism , Lymphoma, Follicular/pathology , Male , Middle Aged , Pleural Effusion, Malignant/metabolism , Pleural Effusion, Malignant/pathology , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/metabolism , Precursor B-Cell Lymphoblastic Leukemia-Lymphoma/pathology , RadiographyABSTRACT
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.