Chidamide induces long-term remission in rare subcutaneous panniculitis-like T-cell lymphoma: An unusual case report and literature review

Li, Liangliang; Wu, Chongyang; Chai, Yifeng; Dong, Chen; Zhao, Li

doi:10.1177/20587384211009342

Cited by 7 publications

(5 citation statements)

References 31 publications

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“…Only 1 case has reported that Chidamide performs well in the treatment of refractory SPTCL. [7] This case we report is a refractory/recurrent SPTCL, which achieved continuous remission after the addition of Chidamide on the basis of CHOP plus auto-HSCT, once again confirming the efficacy of Chidamide in SPTCL. However, more particularly, this case was accompanied by a mutation in ARID1A, indicating that the good efficacy of Chidamide in this SPTCL case may be related to the ARID1A mutation.…”

Section: Discussionsupporting

confidence: 72%

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Case report: Successful treatment of Chidamide in a refractory/recurrent SPTCL with ARID1A mutation on the basis of CHOP plus auto-HSCT

Zhang,

et al. 2023

Medicine

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Rationale: Subcutaneous panniculitis like T-cell lymphoma (SPTCL) is a rare primary cutaneous lymphoma that belongs to peripheral T cell lymphomas, of which the overall prognosis is poor. Chidamide, a deacetylase inhibitor, has been approved for the treatment of peripheral T cell lymphomas. However, due to the rare occurrence of SPTCL, it is currently unknown whether Chidamide is effective for all SPTCL patients and whether there are molecular markers that can predict its therapeutic effect on SPTCL. Patient concerns and diagnoses: The patient was a sixteen-year-old male and underwent subcutaneous nodule biopsy which showed SPTCL. Next-generation sequencing revealed AT-rich interaction domain 1A (ARID1A) mutation, and positron emission tomography/computed tomography showed scattered subcutaneous fluorodeoxyglucose metabolic lesions throughout the body. Interventions and outcomes: During the first 3 CHOP (cyclophosphamide, doxorubicin, vindesine, and prednisone) treatment, the patient relapsed again after remission, and the successive addition of methotrexate and cyclosporine did not make the patient relapsing again. Then, after adding Chidamide to the last 3 CHOP treatment, the patient was relieved again. The patient underwent autologous hematopoietic stem cell transplantation (auto-HSCT) after completing a total of 8 cycles of chemotherapy, and continued maintenance therapy with Chidamide after auto-HSCT. Currently, the patient has been in continuous remission for 35 months. Lessons subsections: This case is the first report of a refractory/recurrent SPTCL with ARID1A mutation treated with Chidamide. The treatment of Chidamide on the basis of CHOP plus auto-HSCT therapy achieved good results, suggesting that ARID1A may act as a molecular marker to predict the therapeutic effect of Chidamide on SPTCL patients, which helps to improve the precision of SPTCL treatment and the overall prognosis of SPTCL patients.

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Section: Discussionsupporting

confidence: 72%

“…Only 1 case has reported that Chidamide performs well in the treatment of refractory SPTCL. [ 7 ] Nevertheless, it is still currently unknown whether all SPTCL patients can be routinely treated with Chidamide and whether there are molecular markers which can predict the efficacy of Chidamide on SPTCL.…”

Section: Introductionmentioning

confidence: 99%

Case report: Successful treatment of Chidamide in a refractory/recurrent SPTCL with ARID1A mutation on the basis of CHOP plus auto-HSCT

Zhang,

et al. 2023

Medicine

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“…53 Chidamide, a histone deacetylase inhibitor, may play a role in refractory SPTCL case, proposed in a case report in China. 54 Histone deacetylase inhibitors could modulate gene expression via hyperacetylation of histones and change nonhistone proteins via regulation at the epigenetic and posttranslational modification levels. 55 Another kind of histone deacetylase inhibitor, Romidepsin, was also utilized in the treatment of one refractory SPTCL in Chicago by targeting PI3k/AKT/mTOR and the Jak/STAT pathways.…”

Section: Sptcl With Hpsmentioning

confidence: 99%

“…Chidamide, a histone deacetylase inhibitor, may play a role in refractory SPTCL case, proposed in a case report in China 54 . Histone deacetylase inhibitors could modulate gene expression via hyperacetylation of histones and change nonhistone proteins via regulation at the epigenetic and posttranslational modification levels 55 .…”

Section: Case Presentationmentioning

confidence: 99%

The pathophysiology and current treatments for the subcutaneous panniculitis‐like T cell lymphoma: An updated review

Lin

Liao

Fang

et al. 2022

Asia-Pac J Clncl Oncology

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Subcutaneous panniculitis‐like T cell lymphoma (SPTCL) is a rare cutaneous T cell lymphoma, which is indolent in nature but could claim life if not correctly diagnosed and promptly treated. SPTCL is usually presented clinically as painless subcutaneous and erythematous nodules over the trunk or extremities. Active clinical vigilance for these subcutaneous nodules or panniculitis‐like lesions is warranted. A biopsy must be performed in order to make a correct diagnosis. Positron emission tomography scan is utilized for disease staging and treatment follow‐up. Due to the rarity of this lymphoma, a standard treatment protocol is not established yet. However, most cases of SPTCL could be treated well under immunosuppressive or polychemotherapeutic drugs except in cases with hemophagocytic syndrome. Hematopoietic stem cell transplantation may be used in refractory or relapse cases. In this review, we presented a case of SPTCL with long‐term complete remission. Meanwhile, since most clinical evidences and experiences of SPTCL are based mostly on case reports or small case series, and the understanding of the SPTCL pathophysiology is limited, we reviewed and updated the pathophysiology and treatments of SPTCL.

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“…Our case differs from these prior studies in that our patient did not have HLH, failed all initial immunosuppressive therapy, and did not have relapsed SPTCL. Another report demonstrated complete remission of systemic SPTCL with chidamide, a different HDAC inhibitor [15]. However, chidamide was used in combination with chemotherapy to achieve remission, so it is unknown whether the positive clinical response was due solely to the HDAC inhibitor.…”

Section: Case Reports In Oncologymentioning

confidence: 99%

Recalcitrant Subcutaneous Panniculitis-Like T-Cell Lymphoma Responsive to Histone Deacetylase Inhibitor

et al. 2022

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Subcutaneous panniculitis-like T-cell lymphoma (SPTCL) is a rare subtype of cutaneous T-cell lymphoma that usually presents with tender subcutaneous nodules on the trunk and extremities. Immunosuppressive therapy is considered first-line treatment for SPTCL, while multiagent chemotherapy is used for SPTCL complicated by hemophagocytic lymphohistiocytosis (HLH). Here, we report a 42-year-old Hispanic man that presented with a 5-year history of recurrent painful subcutaneous lesions in the absence of constitutional symptoms, lymphadenopathy, and hepatosplenomegaly. A punch biopsy revealed an atypical lymphoid infiltrate in between subcutaneous adipose lobules. Lymphocytes expressed CD3, CD8, and Beta F-1 and did not express CD4 and CD56. Based on clinical and histologic findings, the patient was diagnosed with SPTCL. In addition, laboratory findings did not demonstrate any evidence of HLH. He was initially started on both prednisone and hydroxychloroquine with no improvement. A trial of cyclosporine and methotrexate yielded no clinical improvement. As the lesions failed to resolve after treatment with multiple immunosuppressive agents, romidepsin, an intravenous histone deacetylase (HDAC) inhibitor, was initiated. After two cycles of romidepsin, the patient achieved complete clinical response. He continues to be in remission 12 months later with monthly maintenance therapy. This case illustrates that romidepsin can be useful as monotherapy for refractory SPTCL without HLH.

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Chidamide induces long-term remission in rare subcutaneous panniculitis-like T-cell lymphoma: An unusual case report and literature review

Cited by 7 publications

References 31 publications

Case report: Successful treatment of Chidamide in a refractory/recurrent SPTCL with ARID1A mutation on the basis of CHOP plus auto-HSCT

Case report: Successful treatment of Chidamide in a refractory/recurrent SPTCL with ARID1A mutation on the basis of CHOP plus auto-HSCT

The pathophysiology and current treatments for the subcutaneous panniculitis‐like T cell lymphoma: An updated review

Recalcitrant Subcutaneous Panniculitis-Like T-Cell Lymphoma Responsive to Histone Deacetylase Inhibitor

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