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Case Reports
. 2019 Feb 27;7(1):59.
doi: 10.1186/s40425-019-0534-z.

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

Teppei Sugano  1 , Masahiro Seike  2 , Yoko Funasaka  3 , Mai Yoshida  3 , Ryoko Takayama  3 , Ken Okamura  1 , Asuka Nakanishi  1 , Toru Tanaka  1 , Susumu Takeuchi  1 , Rintaro Noro  1 , Yuji Minegishi  1 , Kaoru Kubota  1 , Hidehisa Saeki  3 , Akihiko Gemma  1
Affiliations
Case Reports

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

Teppei Sugano et al. J Immunother Cancer. .

Abstract

Background: Pembrolizumab, 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 presentation: We 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.

Conclusions: This is the first report of ILH induced by pembrolizumab in a patient with lung adenocarcinoma.

Keywords: Intralymphatic histiocytosis; Lung adenocarcinoma; Pembrolizumab.

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Conflict of interest statement

Ethics approval and consent to participate

Written informed consent was obtained from the patient for the analysis of the samples and the tissues.

Consent for publication

Consent for publication was obtained from the patient. A copy of the written consent is available for review.

Competing interests

Masahiro Seike, Kaoru Kubota, and Akihiko Gemma have received honorarium from Merck Sharp & Dohme. The authors report no other conflicts of interest in this work.

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Figures

Fig. 1
Fig. 1
Chest computed tomography analysis determines the baseline before pembrolizumab therapy. A tumor approximately 43 mm in diameter in the right upper lobe, right axial and mediastinal lymph node metastases, and pleural effusion were observed (a, b). After palliative radiotherapy, the size of the right axial lymph node metastasis was decreased (c, d)
Fig. 2
Fig. 2
Clinical appearance. A reddish-brown plaque with edema was present on the right side of the trunk of his body (a, b). Histopathological findings of the patient’s skin biopsy specimens (magnification, 200×). Inflammatory cells, including lymphocytes, plasma cells and macrophages, were present in the dilated vessels (c). Immunostaining revealed aggregates of CD68 positive histiocytes (d) and endothelial cells lining the vessels were positive for D2–40 (e)
Fig. 3
Fig. 3
Clinical course of the case study. The TNF-α level was increased during pembrolizumab therapy. After starting cisplatin and pemetrexed treatment, the TNF-α level was decreased
Fig. 4
Fig. 4
After 4 cycles of pembrolizumab administration, the size of the right axial lymph node metastasis was increased (a, b)
See this image and copyright information in PMC

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