Oral squamous cell carcinoma in a patient affected by chronic graft versus host disease

A 51-year-old Hispanic man with a history of chronic graft versus host disease (cGVHD) was referred, by his onco-haematologist, to our clinics because of a fast-growing lingual mass, in the absence of local trauma. The patient, a non-smoker, received, 4 years before, allogeneic hematopoietic stem cell transplantation (HSCT) from a human leukocyte antigen-id family donor for a stage IVB Epstein-Barr virus-related angioimmunoblastic T-cell lymphoma. After the HSCT, the patient developed cGVHD, with involvement of the liver, skin and oral mucosa. He was under current therapy with Sirolimus because of the worsening of hepatic values of stasis and necrosis related to the liver cGVHD. He was under follow-up and peritoneal dialysis for a drug-induced chronic renal failure.

Intraoral examination showed, at the left tongue apex, an exophytic, hard, red-whitish mass, surrounded by an erythroplasic lesion with a granular, irregular surface, extended to all the left side of the lingual dorsum (Figure 1). The lesion was painful during palpation. A keratotic white plaque was also detected on the lingual dorsum associated with severe tissue atrophy. Further keratotic white striae and erosions were observed at buccal and palatal mucosae, as signs of cGVHD.

Multiple incisional biopsies of the exophytic lingual mass and of the surrounding red area were performed. The histopathological analyses showed a micro-infiltrating oral squamous cell carcinoma (OSCC) in the context of epithelial dysplasia (Figure 2). After the oncological staging, the patient received surgical intervention and radiation therapy.

At a 2-year follow-up, the patient did not show any sign of recurrences.

Bone marrow transplanted patients are at high risk of developing solid malignancies, which can occur in up to 15% of cases and are responsible for 5%–10% of late deaths [1, 2]. The mechanisms involved in cancer onset may be the results of the combined presence of cGVHD (that can occur in 60%–80% of HSCT survivors and increase the risk of OSCC 35 times), radiation and chemotherapy regimens, chronic inflammation and long-term immunosuppression [3-5]. Oral cGVHD, in particular, is present in 80%–90% of patients affected by systemic cGVHD and it usually occurs as lichenoid lesions, as in this report. Oral cGVHD predisposes patients to OSCC even several years after HSCT, in the range from 1 to 22 years after the transplantation [4, 6]. Consistently, our patient developed oral cancer after 4 years. These patients require lifelong multidisciplinary oral cancer screening, and the role of onco-haematologists is pivotal to guarantee OSCC early diagnosis.

Sem Decani, Elena Maria Varoni and Niccolò Lombardi followed the patient during diagnosis, collected the clinical data, and reviewed the manuscript. Giulia Ghidini collected clinical data and wrote the report. Laura Moneghini performed the histopathological diagnosis. Written informed consent to publication was obtained.

The authors declare no conflict of interest.

This study did not receive any specific grant from any funding agencies in the public, commercial or not-for-profit sectors.

Not applicable. Ethics committee/IRB approval is not required in the case of case reports/clinical images.

The authors obtained a signed patient permission to publish the clinical image related to this manuscript.

The authors have confirmed clinical trial registration is not needed for this submission.