Intralymphatic histiocytosis in a patient with lung adenocarcinoma treated with pembrolizumab: a case report

Teppei Sugano,Toru Tanaka,Hidehisa Saeki,Akihiko Gemma,Ken Okamura,Susumu Takeuchi,Yoko Funasaka,Kaoru Kubota,Asuka Nakanishi,Mai Yoshida,Ryoko Takayama,Masahiro Seike,Yuji Minegishi,Rintaro Noro

doi:10.1186/s40425-019-0534-z

Abstract

BackgroundPembrolizumab, an anti-programmed cell death-1 protein monoclonal antibody, is effective for patients with advanced non-small-cell lung cancer. However, immune checkpoint inhibitors such as pembrolizumab induce various immune-related adverse events, involving the lung, liver, gastrointestinal, endocrine system, and skin. Intralymphatic histiocytosis (ILH) is a rare, chronic cutaneous disorder with a reactive inflammatory component, which often occurs in patients with rheumatoid arthritis.Case presentationWe present a 67-year-old man with lung adenocarcinoma who developed ILH associated with pembrolizumab treatment. He was treated with palliative thoracic radiotherapy for superior vena cava syndrome. Subsequently, he received four cycles of pembrolizumab. Approximately 2.5 months after the initiation of pembrolizumab, he developed erythema on the trunk of his body. Based on findings of skin biopsies, he was diagnosed with pembrolizumab-induced ILH. Moreover, the upregulation of tumor necrosis factor-α was observed during pembrolizumab therapy.ConclusionsThis is the first report of ILH induced by pembrolizumab in a patient with lung adenocarcinoma.

Highlights

Pembrolizumab, an anti-programmed cell death-1 protein monoclonal antibody, is effective for patients with advanced non-small-cell lung cancer
This is the first report of Intralymphatic histiocytosis (ILH) induced by pembrolizumab in a patient with lung adenocarcinoma
It was previously reported that tumor necrosis factor α (TNF-α) is associated with the pathogenesis of ILH

Summary

Background

Immune checkpoint inhibitors (ICIs) have shown promising results in clinical trials and are recognized as the standard treatment for advanced non-smallcell lung cancer (NSCLC) [1, 2]. An epidermal growth factor receptor mutation and rearranged anaplastic lymphoma kinase genes were not detected His tumor had invaded the superior vena cava (SVC), leading to the swelling of his right arm and face, suggesting SVC syndrome. 2.5 months after treatment with pembrolizumab, he presented with an asymptomatic, poorly demarcated 1–3 cm erythematous plaque over the right trunk of his body, which gradually developed in size (Fig. 2a and b). He had no symptoms and his blood examination test results showed no remarkable changes. After 4 cycles of pembrolizumab treatment, the size of the tumor in right upper lobe had decreased. After 2 cycles of chemotherapy, he maintained a partial response without any severe adverse events and ILH was gradually resolved with topical steroid therapy

Discussion

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Conclusion