EBV-Associated T-Cell Lymphoproliferative Disorders Demonstrated on FDG PET/CT in a Patient With Hemophagocytic Lymphohistiocytosis

Hao, Ruirui; Yang, Xu; Liu, Zhimou; Yang, Jigang

doi:10.1097/rlu.0000000000002649

Cited by 10 publications

(10 citation statements)

References 15 publications

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“…However, some studies found that the extranodal NK/T-cell lymphoma nasal type, aggressive NK cell leukemia, and some types of B-cell lymphoma occasionally lack lesions with focal FDG uptake [ 9 12 19 ]. Multiple hypermetabolic lymph nodes or patchy bone lesions were observed in several cases of EBV-associated HLH [ 12 21 22 23 ]. In the absence of EBV infection, lesions with focal FDG uptake in the bone marrow are a reliable sign of malignancy in secondary HLH.…”

Section: Discussionmentioning

confidence: 99%

Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Yang

Liu

et al. 2022

Korean J Radiol

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Objective 18 F-fluorodeoxyglucose (FDG) PET/CT is often used for detecting malignancy in patients with newly diagnosed hemophagocytic lymphohistiocytosis (HLH), with acceptable sensitivity but relatively low specificity. The aim of this study was to improve the diagnostic ability of 18 F-FDG PET/CT in identifying malignancy in patients with HLH by combining 18 F-FDG PET/CT and clinical parameters. Materials and Methods Ninety-seven patients (age ≥ 14 years) with secondary HLH were retrospectively reviewed and divided into the derivation (n = 71) and validation (n = 26) cohorts according to admission time. In the derivation cohort, 22 patients had malignancy-associated HLH (M-HLH) and 49 patients had non-malignancy-associated HLH (NM-HLH). Data on pretreatment 18 F-FDG PET/CT and laboratory results were collected. The variables were analyzed using the Mann-Whitney U test or Pearson’s chi-square test, and a nomogram for predicting M-HLH was constructed using multivariable binary logistic regression. The predictors were also ranked using decision-tree analysis. The nomogram and decision tree were validated in the validation cohort (10 patients with M-HLH and 16 patients with NM-HLH). Results The ratio of the maximal standardized uptake value (SUVmax) of the lymph nodes to that of the mediastinum, the ratio of the SUVmax of bone lesions or bone marrow to that of the mediastinum, and age were selected for constructing the model. The nomogram showed good performance in predicting M-HLH in the validation cohort, with an area under the receiver operating characteristic curve of 0.875 (95% confidence interval, 0.686–0.971). At an appropriate cutoff value, the sensitivity and specificity for identifying M-HLH were 90% (9/10) and 68.8% (11/16), respectively. The decision tree integrating the same variables showed 70% (7/10) sensitivity and 93.8% (15/16) specificity for identifying M-HLH. In comparison, visual analysis of 18 F-FDG PET/CT images demonstrated 100% (10/10) sensitivity and 12.5% (2/16) specificity. Conclusion 18 F-FDG PET/CT may be a practical technique for identifying M-HLH. The model constructed using 18 F-FDG PET/CT features and age was able to detect malignancy with better accuracy than visual analysis of 18 F-FDG PET/CT images.

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Section: Discussionmentioning

confidence: 99%

Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Yang

Liu

et al. 2022

Korean J Radiol

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“…Radiomics quantified the spatial complexity of them. This study exhibited that the heterogeneity of spleen and liver may reveal overproliferation of immune cells accompanied with inflammatory infiltration triggered by EBV infection (37)(38)(39)(40). On the other hand, the heterogeneous distribution of metabolism or density may also suggest the involvement of tumor cells (41)(42)(43).…”

Section: Timely Diagnosis and Prognosis Are Critical For Hlh Considering That The Early And Proper Administration Of An Efficaciousmentioning

confidence: 87%

Development and Validation of a Nomogram Based on 18F-FDG PET/CT Radiomics to Predict the Overall Survival in Adult Hemophagocytic Lymphohistiocytosis

Yang

Liu

et al. 2021

Front. Med.

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Purpose: Hemophagocytic lymphohistiocytosis (HLH) is a rare and severe disease with a poor prognosis. We aimed to determine if 18F-fluorodeoxyglucose (18F-FDG) PET/CT-derived radiomic features alone or combination with clinical parameters could predict survival in adult HLH.Methods: This study included 70 adults with HLH (training cohort, n = 50; validation cohort, n = 20) who underwent pretherapeutic 18F-FDG PET/CT scans between August 2016 and June 2020. Radiomic features were extracted from the liver and spleen on CT and PET images. For evaluation of 6-month survival, the features exhibiting p < 0.1 in the univariate analysis between non-survivors and survivors were selected. The least absolute shrinkage and selection operator (LASSO) regression analysis was used to develop a radiomics score (Rad-score). A nomogram was built by the multivariate regression analysis to visualize the predictive model for 3-month, 6-month, and 1-year survival, while the performance and usefulness of the model were evaluated by calibration curves, the receiver operating characteristic (ROC) curves, and decision curves.Results: The Rad-score was able to predict 6-month survival in adult HLH, with area under the ROC curves (AUCs) of 0.927 (95% CI: 0.878–0.974) and 0.869 (95% CI: 0.697–1.000) in the training and validation cohorts, respectively. The radiomics nomogram combining the Rad-score with the clinical parameters resulted in better performance for predicting 6-month survival than the clinical model or the Rad-score alone. Moreover, the nomogram displayed superior discrimination, calibration, and clinical usefulness in both the cohorts.Conclusion: The newly developed Rad-score is a powerful predictor for overall survival (OS) in adults with HLH. The nomogram has great potential for predicting 3-month, 6-month, and 1-year survival, which may timely guide personalized treatments for adult HLH.

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“…The difference may result from the high physiological uptake in children's cervical lymph nodes and the large proportion of EBV infection-related lymphoproliferative diseases (EBV-LPD) in children's HLH. As LPD is often associated with multiple enlarged lymph nodes with increased FDG uptake, as well as focal hypermetabolism sometimes, it is challenging to differentiate LPD from lymphoma [ 11 ]. In this study, two patients with intestinal involvement were pathologically diagnosed with Hodgkin's lymphoma and non-Hodgkin's lymphoma.…”

Section: Discussionmentioning

confidence: 99%

¹⁸F‐FDG PET/CT for Identifying the Potential Primary Diseases and Predicting Prognosis of Secondary Hemophagocytic Lymphohistiocytosis in Children

Wei

et al. 2022

Contrast Media & Molecular Imaging

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Hemophagocytic lymphohistiocytosis (HLH) is a rare, potentially fatal illness, which can be divided into primary HLH (pHLH) and secondary HLH (sHLH). pHLH can be driven by genetic defections. Moreover, the sHLH is usually be triggered by malignancy or non-malignancy diseases. Sixty-two newly diagnosed sHLH patients with known etiology and those who underwent 18F-FDG PET/CT examination from July 2018 to December 2020 were retrospectively analyzed. They were divided into malignancy-associated HLH (M-HLH, n = 13) and non-malignancy-associated HLH (NM-HLH, n = 49). The metabolic parameters of the liver (Li), spleen (Sp), bone marrow (BM), lymph nodes (LN), and their ratios to the liver background (LiBG) and mediastinum (M) were compared between two groups. These metabolic parameters were evaluated for correlation with laboratory parameters and prognostic parameters. We found that the SUVmax-LN/Sp/Li and SUVmean-Sp in M-HLH were significantly higher than those in NM-HLH ( P = 0.031 , 0.035 , 0.016 , and 0.032 ). The malignant disease should be considered when SUVmax-LN was higher than 4.41 (sensitivity 61.5%, specificity 81.6%). Hypermetabolic lesions in extranodal organs were more likely to occur in M-HLH than in NM-HLH ( P = 0.011 ). IFN- γ was positively correlated with SUVmax-BM/Li/Sp and SUVmean-BM/Li/Sp ( P < 0.05 ). Ferritin, sCD25, IL-6, and IL-10 were positively correlated with SUVmax-Sp and SUVmean-Sp ( P < 0.05 ). In Epstein–Barr virus-associated HLH (EBV-HLH), the SUV parameters of bone marrow were significantly correlated with a poor 2-week treatment response, overall survival, and event-free survival ( P < 0.05 ). We conclude that some 18F-FDG PET/CT metabolic parameters can help identify the etiology of sHLH in children and provide directions for further inspection. The malignant disease should be considered when the SUVmax-LN is higher than 4.41 and hypermetabolic lesions occur in extranodal organs. In EBV-HLH, a higher SUV of bone marrow is associated with a poorer prognosis.

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EBV-Associated T-Cell Lymphoproliferative Disorders Demonstrated on FDG PET/CT in a Patient With Hemophagocytic Lymphohistiocytosis

Cited by 10 publications

References 15 publications

Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Development and Validation of a Nomogram Based on 18F-FDG PET/CT Radiomics to Predict the Overall Survival in Adult Hemophagocytic Lymphohistiocytosis

¹⁸F‐FDG PET/CT for Identifying the Potential Primary Diseases and Predicting Prognosis of Secondary Hemophagocytic Lymphohistiocytosis in Children

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EBV-Associated T-Cell Lymphoproliferative Disorders Demonstrated on FDG PET/CT in a Patient With Hemophagocytic Lymphohistiocytosis

Cited by 10 publications

References 15 publications

Development and Validation of 18F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Development and Validation of 18F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Development and Validation of a Nomogram Based on 18F-FDG PET/CT Radiomics to Predict the Overall Survival in Adult Hemophagocytic Lymphohistiocytosis

18F‐FDG PET/CT for Identifying the Potential Primary Diseases and Predicting Prognosis of Secondary Hemophagocytic Lymphohistiocytosis in Children

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Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

Development and Validation of ¹⁸F-FDG PET/CT-Based Multivariable Clinical Prediction Models for the Identification of Malignancy-Associated Hemophagocytic Lymphohistiocytosis

¹⁸F‐FDG PET/CT for Identifying the Potential Primary Diseases and Predicting Prognosis of Secondary Hemophagocytic Lymphohistiocytosis in Children