A Case of Primary Cutaneous Gamma-Delta T-Cell Lymphoma with Pautrier Microabscess

Kato, Kohei; Namiki, Takeshi; Ueno, Makiko; Iikawa, Madoka; Tokoro, Shown; Nishizawa, Aya; Yamamoto, Kouhei; Miura, Keiko; Yokozeki, Hiroo

doi:10.5021/ad.2017.29.2.229

Cited by 6 publications

(8 citation statements)

References 5 publications

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“…Intriguingly, in our case, cytotoxic molecules including TIA‐1, granzyme B and perforin were negative. Two similar cases have been reported with detailed description of clinical and immunological features . One case was previously reported from our department .…”

Section: Discussionmentioning

confidence: 68%

“…In the patient reported by Kato et al. , cyclophosphamide, doxorubicin, vincristine and prednisone therapy was effective and the symptoms had been under control for at least 2 years. Among 53 cases, Guitart et al.…”

Section: Discussionmentioning

confidence: 90%

“…12 The patient had survived for 13 years since the diagnosis with treatment by oral etretinate, narrowband ultraviolet B irradiation, radiation and/or oral etoposide. In the patient reported by Kato et al, 13 cyclophosphamide, doxorubicin, vincristine and prednisone therapy was effective and the symptoms had been under control for at least 2 years. Among 53 cases, Guitart et al 3 found only four CGD-TCL cases negative for TIA-1 and granzyme B.…”

Section: Discussionmentioning

confidence: 91%

“…Two similar cases have been reported with detailed description of clinical and immunological features. 12,13 One case was previously reported from our department. 12 The patient had survived for 13 years since the diagnosis with treatment by oral etretinate, narrowband ultraviolet B irradiation, radiation and/or oral etoposide.…”

Section: Discussionmentioning

confidence: 99%

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Primary cutaneous γδ T‐cell lymphoma with unusual immunophenotype: A case report and review of published work

Kamijo

Miyagaki

Norimatsu³

et al. 2020

The Journal of Dermatology

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Primary cutaneous γδ T‐cell lymphoma (CGD‐TCL) is a rare form of primary cutaneous lymphoma. The histopathological features of CGD‐TCL are still unclear because of its rarity. Here, we report a case of a 77‐year‐old Japanese man who presented with a 9‐month history of erythematous plaques on his left forearm. Skin biopsy specimens revealed the infiltration of atypical medium/large‐sized lymphocytes from the epidermis to the deep dermis. Atypical lymphocytes were positive for CD3, CD5, CD8 and Vδ1, and negative for CD4, CD7, CD56, EBER‐ISH, intracellular antigen‐1, granzyme B and perforin. CD30 was partially expressed. We also reviewed 246 cases of CGD‐TCL from the published work. CD4−CD8− double‐negative cases were 113 of 196 cases (57.6%), followed by CD4−CD8+ cases (52/196, 26.5%). CD5 was expressed in 25.8% of the cases (34/132). At least one cytotoxic molecule marker was expressed in 150 of 160 cases (93.8%). Some cases showed an indolent clinical course, especially in mycosis fungoides‐like CGD‐TCL cases. CD5 positivity and lack of cytotoxic molecule expression could be associated with a better prognosis. In addition, CD30 expression was found in approximately half of CGD‐TCL cases (51/112 cases), suggesting that brentuximab vedotin could be a good treatment option for such patients. Further studies with more cases with detailed clinical and pathological information are necessary to elucidate the etiology and prognostic markers of this entity.

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Section: Discussionmentioning

confidence: 68%

Section: Discussionmentioning

confidence: 90%

Section: Discussionmentioning

confidence: 91%

Section: Discussionmentioning

confidence: 99%

See 2 more Smart Citations

Primary cutaneous γδ T‐cell lymphoma with unusual immunophenotype: A case report and review of published work

Kamijo

Miyagaki

Norimatsu³

et al. 2020

The Journal of Dermatology

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“…PCGDTCL is an extremely rare malignancy amounting to <1% of all skin lymphomas. Accordingly, only a small number of cases have been reported making PCGDTCL a poorly understood lymphoma (3)(4)(5)(6). In those cases reported, the course of PCGDTCL was mostly short and highly aggressive with resistance to chemo-and radiotherapy (7,8).…”

Section: Introductionmentioning

confidence: 99%

Primary Cutaneous Gamma-Delta T-Cell Lymphoma With Long-Term Indolent Clinical Course Initially Mimicking Lupus Erythematosus Profundus

et al. 2020

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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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A Case of Primary Cutaneous Gamma-Delta T-Cell Lymphoma with Pautrier Microabscess

Cited by 6 publications

References 5 publications

Primary cutaneous γδ T‐cell lymphoma with unusual immunophenotype: A case report and review of published work

Primary cutaneous γδ T‐cell lymphoma with unusual immunophenotype: A case report and review of published work

Primary Cutaneous Gamma-Delta T-Cell Lymphoma With Long-Term Indolent Clinical Course Initially Mimicking Lupus Erythematosus Profundus

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

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