Pagetoid reticulosis tumor cells with double expression of <scp>TCRγδ</scp> and <scp>TCRαβ</scp>: an off‐target phenomenon or genuine expression?

Tomasini, Dario; Niccoli, Annalisa; Crivelli, Filippo

doi:10.1111/cup.12486

Cited by 10 publications

(7 citation statements)

References 19 publications

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“…However, rare expression of γ/ɗ-T-cell receptor chains may also be observed in CD4+ or CD8+ cutaneous lymphomas, such as MF, anaplastic large-cell lymphoma or pagetoid reticulosis, and these lymphomas run a rather indolent or stage-dependent course [ 13 ]. In less than 1% of cases, lineage infidelity with, e.g., aberrant expression of CD20 or concomitant dual expression of both γ/ɗ-chains and αβ-chains of the T-cell receptor [ 14 ] may be observed, as was present in one case of cutaneous PTCL-NOS in our study.…”

Section: Discussionmentioning

confidence: 65%

Cutaneous CD8+ Cytotoxic T-Cell Lymphoma Infiltrates: Clinicopathological Correlation and Outcome of 35 Cases

et al. 2016

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IntroductionCytotoxic CD8+ T-cell lymphomas are only rarely encountered and thus remain only poorly characterized. Our aim was to collect and correlate clinical and histological data of CD8+ skin lymphoma infiltrates to obtain a proper subtype assignment of CD8+ skin lymphoma infiltrates and to derive putative prognostic markers thereof.MethodsFormalin-fixed and paraffin-embedded (FFPE) tissue of 35 patients with CD8+ cytotoxic cutaneous T-cell lymphoma infiltrates was retrieved from the archives of the Institute of Pathology and the Department of Dermatology, University Hospital Wuerzburg, dating back from 1998 until 2015. Cytological, histological, immunohistochemical and molecular genetic features were assessed and correlated with respective clinical data.ResultsThe identified cases of CD8+ cytotoxic atypical lymphoproliferative infiltrates of the skin (n = 35) comprised 13 cases of mycosis fungoides (MF)/Sézary syndrome (SS), 4 cases of subcutaneous panniculitis-like T-cell lymphoma (SPTCL), 5 cases of primary cutaneous acral CD8+ lymphoma [formerly indolent CD8+ lymphoid proliferation (ILP)] and 1 case of aggressive epidermotropic primary cutaneous T-cell lymphoma (AECTCL). Moreover, nine cases were classified as primary cutaneous peripheral T-cell lymphoma, not otherwise specified (PTCL-NOS) and three cases as systemic PTCL-NOS. Multiple skin lesions, a high proliferative index and especially a final subtype attribution to AECTCL or systemic PTCL-NOS were associated with a worse survival. Coexpression of CD68 by tumor cells was exclusively observed in indolent acral CD8+ T-cell lymphoma and thus indicated an invariably benign clinical course. No further distinctive markers could be derived from our analysis.ConclusionCutaneous infiltrates of CD8+ cytotoxic T-cell lymphoma comprise clinically and histologically heterogeneous entities of either primary cutaneous T-cell lymphomas or secondary infiltrates of otherwise systemic peripheral T-cell lymphomas. A thorough clinicopathological correlation with respective staging examinations remains the mainstay for correct subtype assignment and proper prognostication as long as no better markers have been defined.

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Section: Discussionmentioning

confidence: 65%

Cutaneous CD8+ Cytotoxic T-Cell Lymphoma Infiltrates: Clinicopathological Correlation and Outcome of 35 Cases

et al. 2016

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“…Histopathological images captured by our team are provided in Figure 4. Furthermore, other high‐power view images captured can be found in the literature 7–9 . Only 84 cases (58.7%) reported immunohistochemical findings.…”

Section: Resultsmentioning

confidence: 99%

Woringer‐Koloppdisease (localized pagetoid reticulosis): a systematic review

et al. 2022

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Objective Woringer-Kolopp disease (WKD), also known as localized pagetoid reticulosis, is a rare variant of mycosis fungoides as described by the World Health Organization-European Organization for Research and Treatment of Cancer (WHO-EORTC) classification system for cutaneous lymphomas. Our objective was to conduct a comprehensive review that describes and evaluates patient demographics, clinical presentation, immunohistochemical findings, management, and outcomes of WKD. Methods The databases PubMed, Embase, and Cochrane Library were searched for relevant literature. Patient demographics, imaging, treatments, and other clinical characteristics were obtained. The literature search was conducted on December 7, 2020.Studies were included if they contained primary data related to WKD. Non-pertinent studies, non-English studies, non-human studies, review articles, or studies with insufficient case information were excluded. The quality of the included studies and the risk of bias were evaluated using the Grades of Recommendation, Assessment, Development, and Evaluation Group (GRADE) criteria ( 2016), Oxford Centre for Evidence-Based Medicine: Levels of Evidence (OCEBM) (March 2009), and the Methodological Index for Non-Randomized Studies (MINORS) criteria (2003).Results A total of 84 studies with 143 patients met the inclusion criteria for this study. The most common chief complaint was an asymptomatic, slow-growing, and erythematous plaque. The average time from initial skin eruption to presentation was 97.6 months.Histologically, 128 cases of WKD displayed epidermotropism (97.7%). Various therapies, including radiotherapy, surgery, and local chemotherapy, were used. In total, 92 (78.6%) cases had complete remission and 11 (9.4%) cases recurred. Conclusions WKD is a rare cutaneous T-cell lymphoma that often presents as a single plaque on the extremities with an indolent course and a favorable prognosis. However, it is often misdiagnosed, leading to delays in treatment. The diagnosis of WKD involves clinical appraisal, a biopsy of suspicious lesions, and immunohistochemistry. Monotherapy appears to be preferred compared to combination therapy for WKD with radiotherapy demonstrating among the highest cure rates and lowest recurrence. Future retrospective and prospective studies are needed to accurately determine the epidemiology, prognosis, and effective treatments for WKD. Limitations include a possibility of missed studies, heterogeneity in reporting methods, publication, and selection bias.What is already known about this topic?• Woringer-Kolopp disease (WKD) is a rare variant of mycosis fungoides as described by the World Health Organization-European Organization for Research and Treatment of Cancer (WHO-EORTC) classification system for cutaneous lymphomas.• Qualitative systematic analysis can generate new insights compared to individual findings.What does this study add?• Demonstrates the need for accurate diagnosis of Cutaneous T-Cell Lymphomas.

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“…16 In their landmark paper, Rodriguez-Pinilla et al 17 noted that there is no apparent detriment in survival in patients with gamma/delta-positive mycosis fungoides. 13,[17][18][19][20][21][22][23][24][25] Our report is one of few studies to make use of rich case report literature in dermatology and dermatopathology as a means to collect a wide range of cases, particularly when collation of slide materials for such a broad array of patients over time and space would not be possible. Two major limitations of this study are the impact of publication bias and lack of central review.…”

Section: Discussionmentioning

confidence: 99%

The significance of epidermal involvement in primary cutaneous gamma/delta (γδ) T‐cell lymphoma: A systematic review and meta‐analysis

2021

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Background: Primary cutaneous gamma/delta (γδ) T-cell lymphoma (PCGDTCL) is a rare, aggressive peripheral T-cell lymphoma. There is evidence that patients with epidermotropic PCGDTCL may have an improved prognosis compared with those with only dermal and/or subcutaneous involvement.Methods: Systematic review of the literature and application of inclusion criteria yielded 48 manuscripts detailing the cases of 104 patients.Results: Of the 104 patients, 57 were male (51.4%) and 47 were female (48.5%) Based on provided histopathologic descriptions, 57 cases (54.8%) had no epidermotropism, 47 cases (45.2%) patients demonstrated any degree of epidermotropism, and 25 cases were predominantly epidermotropic (25/104, 24%).Five-year overall survivals for patients with no epidermotropism, any epidermotropism, and predominantly epidermotropic presentation were 32.8%, 28.9%, and 40.0%, respectively (p = 0.40). The most commonly performed immunohistochemical markers were CD3, CD4, CD8, CD5, CD7, CD30, CD56, TCR beta, TCR γ, and TCR δ. There was no statistically significant difference in immunophenotype between groups. Lesion morphology was described in the majority of cases (85/104, 80.9%); most cases presented as a combination of nodules, plaques, and tumors (77.4%). Several cases had more atypical presentations, including "mycosisfungoides-like" and ulcerated. Conclusion:In PCGDTCL, neither epidermotropism nor predominantly epidermotropic phenotype predict a better prognosis. In addition, the case report literature in dermatology and dermatopathology is rich and highly valuable. K E Y W O R D S case report, case series, cutaneous T-cell lymphoma, primary cutaneous gamma/delta T-cell lymphoma, review article 1 | INTRODUCTION Primary cutaneous gamma/delta (γδ) T-cell lymphoma (PCGDTCL) is a rare, aggressive peripheral T-cell lymphoma characterized as a subcutaneous lymphoma composed of neoplastic gamma/delta T-cells. PCGDTCL typically presents with ulcerated nodules or tumors on the extremities. Mortality from this lymphoma is relatively high, approaching 89% at 5 years. 1 Patients are at high risk of complications including hemophagocytic lymphohistiocytosis (HLH, also known as hemophagocytic syndrome, or HPS) and sepsis. At this time, IRB statement: This study was IRB exempt.

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Pagetoid reticulosis tumor cells with double expression of TCRγδ and TCRαβ: an off‐target phenomenon or genuine expression?

Cited by 10 publications

References 19 publications

Cutaneous CD8+ Cytotoxic T-Cell Lymphoma Infiltrates: Clinicopathological Correlation and Outcome of 35 Cases

Cutaneous CD8+ Cytotoxic T-Cell Lymphoma Infiltrates: Clinicopathological Correlation and Outcome of 35 Cases

Woringer‐Koloppdisease (localized pagetoid reticulosis): a systematic review

The significance of epidermal involvement in primary cutaneous gamma/delta (γδ) T‐cell lymphoma: A systematic review and meta‐analysis

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