Prevalence and spectrum of T-cell lymphoproliferative disorders in patients with Hypereosinophilia: A reference laboratory experience

Shi, Min; Rech, Karen L.; Otteson, Gregory E.; Horna, Pedro; Olteanu, Horatiu; Pardanani, Animesh; Chen, Dong; Jevremović, Dragan

doi:10.1016/j.anndiagpath.2019.151412

Cited by 12 publications

(12 citation statements)

References 14 publications

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“…These cases accounted for 9% (7/77) of patients with HES in whom neither a reactive nor neoplastic cause was identified after an extensive workup (Hu et al, 2018). Reported incidences of L‐HES among patients with eosinophilia vary (Fang et al, 2018; Shi et al, 2019), depending on the patient populations screened. Seven of these patients had clinical symptoms and signs related to an eosinophilic infiltrate or eosinophil activation.…”

Section: Discussionmentioning

confidence: 99%

“…Lymphocytic variant hypereosinophilic syndrome (L-HES) is a biologically distinct type of HES where eosinophilia is reactive/secondary due to the expansion of circulating aberrant T-cells, frequently clonal with a Th2 cell immunophenotype CD2 + CD3-CD4 + CD5bright + CD7−/partial+CD8−, and less commonly CD3 + CD4-CD8-. These T-cells, mostly clonal, can produce eosinophilopoietic cytokines such as IL-4, IL-5, and/or IL-13, with IL-5 to be the most potent growth and activation factor inducing eosinophilic overproduction (Campbell et al, 1987;Roufosse, Cogan, & Goldman, 2007;Shomali & Gotlib, 2019). A diagnosis of L-HES used to rely on PCR TCR gene rearrangement study; however, due to the promiscuity of clonal TCR gene rearrangement as well as the lack of detectable TCR gene rearrangement in a small proportion of such case (Lefevre et al, 2014;Shi et al, 2019), identification of aberrant T-cells by flow cytometry is required to establish a diagnosis (Shomali & Gotlib, 2019;Valent et al, 2012).…”

Section: Introductionmentioning

confidence: 99%

“…These T‐cells, mostly clonal, can produce eosinophilopoietic cytokines such as IL‐4, IL‐5, and/or IL‐13, with IL‐5 to be the most potent growth and activation factor inducing eosinophilic overproduction (Campbell et al, 1987; Roufosse, Cogan, & Goldman, 2007; Shomali & Gotlib, 2019). A diagnosis of L‐HES used to rely on PCR TCR gene rearrangement study; however, due to the promiscuity of clonal TCR gene rearrangement as well as the lack of detectable TCR gene rearrangement in a small proportion of such case (Lefevre et al, 2014; Shi et al, 2019), identification of aberrant T‐cells by flow cytometry is required to establish a diagnosis (Shomali & Gotlib, 2019; Valent et al, 2012). It is known that with long‐term follow‐up, about 5–20% patients may develop T‐cell lymphomas (Lefevre et al, 2014; Lefevre et al, 2015; Ravoet et al, 2005; Roufosse, 2015; Vaklavas et al, 2007).…”

Section: Introductionmentioning

confidence: 99%

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Lymphocytic variant of hypereosinophilic syndrome: A report of seven cases from a single institution

Wang

Thakral

et al. 2020

Cytometry Part B Clinical

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Background: Lymphocytic variant of hypereosinophilic syndrome (L-HES) is a subtype of HES driven by cytokines produced by clonal T-cells. Due to the rarity of its occurrence and challenges in diagnosis, this subtype of HES is under recognized. Methods and Results: We report seven patients with L-HES, diagnosed from a group of 136 patients who were referred to our institution for the work-up of hypereosinophilia. The clinical presentation, symptoms and signs were heterogeneous and uncharacteristic; indistinguishable from idiopathic HES. Flow cytometry immunophenotypic analysis revealed aberrant T-cells in all patients, with a Th2 immunophenotype, CD2 + CD3−CD4 + CD5 + CD7dim+/−CD8− in six of seven (86%) cases. CD10 was partially expressed in one of seven (14%) cases, and clonal TCR gene rearrangement was detected by PCR in five of seven (71%) patients. All patients were treated with corticosteroids and two of seven (29%) patients received anti-IL5 antibody therapy. With a median follow-up time of 7.5 years (2.3-14.1 years), one (11%) patient developed peripheral T-cell lymphoma 6.1 years after the initial diagnosis of L-HES and responded well to chemotherapy. All patients were alive at the last follow-up. Conclusion: In conclusion, a combination of flow cytometry immunophenotyping and molecular analysis allows the identification of aberrant T-cells, facilitating a diagnosis of L-HES in patients with eosinophilia. A correct diagnosis is essential for the proper management of these patients.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Lymphocytic variant of hypereosinophilic syndrome: A report of seven cases from a single institution

Wang

Thakral

et al. 2020

Cytometry Part B Clinical

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“…de triptasa, población anormal de células T, incremento de blastos, displasia, alteraciones citogenéticas o moleculares, fibrosis medular, esplenomegalia o linfadenopatías (2,5,8) . En nuestro paciente no se corroboró la clonalidad de la hipereosinofilia en el debut, pero su evolución clínica en el tiempo nos implicó a elucubrarlo.…”

Section: Neoplasia Mieloide/linfoide Con Eosinofilia Y Reordenamiento...unclassified

Neoplasia mieloide/linfoide con eosinofilia y reordenamiento FIP1L1-PDGFRA asociado a linfoma T periférico NOS. ¿Es probable?

Chiang¹,

González²,

Farfán³

2023

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Presentamos el caso de un paciente masculino con neoplasia mieloide/linfoide con eosinofilia (NMLeo) y reordenamiento PDGFRA (gen del receptor alfa del factor de crecimiento derivado de plaquetas), con antecedente de linfoma T periférico sin otras especificaciones (LT-NOS) e hipereosinofilia. Las NML-eo con PDGFRA surgen de una célula pluripotencial progenitora hematopoyética, que genera linajes de diferenciación tanto mieloide como linfoide. El gen de fusión puede expresarse en eosinófilos, neutrófilos, mastocitos, células T, células B y monocitos. ¿Es posible la asociación entre LT-NOS y NML-eo con PDGFRA?

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“…Reactive HES can be attributed to abnormal T-cell activation, lymphoproliferative disorders, or neoplasms, with abnormal T-cell populations detected by flow cytometry in 12%-27% of cases. [30][31][32] The lymphoid variant (L-HES) is a type of reactive HES in which a clonal T-cell population with associated T-cell receptor rearrangements and overproduction of T H 2 cytokines (ie, interleukin 5 [IL-5]) is identified. 1,33 Hypereosinophilia of undetermined significance is reserved for cases of incidentally detected peripheral hypereosinophilia in which there are no clinical signs or symptoms and no evident organ damage, even in the absence of treatment.…”

Section: Types Of Hesmentioning

confidence: 99%

An Approach to Hypereosinophilic Syndrome Presenting With Cutaneous Features

Fourzali¹,

Yosipovitch²

2022

Dermatitis

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Hypereosinophilic syndrome (HES) is a heterogeneous group of disorders characterized by persistent peripheral hypereosinophilia and eosinophilia-mediated tissue damage. Hypereosinophilic syndrome can have life-threatening effects on multiple organ systems but may initially, or in some cases solely, present with skin lesions. The clinical presentation of HES in the skin represents a diagnostic challenge for the dermatologist, because cutaneous manifestations are highly variable and may be mistaken for several dermatologic conditions. Once peripheral and tissue eosinophilia is diagnosed, the differential diagnosis is quite broad, spanning hematoproliferative disorders, infectious diseases, drug reactions, and many others. Workup and management may also present a challenge, because the prospect for organ system involvement in those with apparent skin-limited disease is unclear. This article provides a dermatology-centered approach to HES and provides a reference for the differential diagnosis, workup, and management of this complex disorder.

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Prevalence and spectrum of T-cell lymphoproliferative disorders in patients with Hypereosinophilia: A reference laboratory experience

Cited by 12 publications

References 14 publications

Lymphocytic variant of hypereosinophilic syndrome: A report of seven cases from a single institution

Lymphocytic variant of hypereosinophilic syndrome: A report of seven cases from a single institution

Neoplasia mieloide/linfoide con eosinofilia y reordenamiento FIP1L1-PDGFRA asociado a linfoma T periférico NOS. ¿Es probable?

An Approach to Hypereosinophilic Syndrome Presenting With Cutaneous Features

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