Two patients underwent allogeneic hematopoietic stem cell transplantation (HSCT) for acute myeloid leukemia. Chronic graft-versus-host disease (GVHD) developed, with persistent symptomatic oral lesions. At 2 and 6 years post-HSCT, both patients developed squamous cell carcinoma (SCC) of the tongue in areas previously involved by chronic GVHD. None had any known risk factor for SCC. Histologically, moderate to severe dysplasia was present in noncancerous oral mucosa. Oral SCC is rarely described after HSCT, and a review of the reported cases showed chronic GVHD to be a common risk, suggesting that the chronic inflammation associated with GVHD might be of pathogenetic significance. Am. J. Hematol. 77:200-202, 2004. ª 2004 Key words: squamous cell carcinoma; tongue; GVHD; HSCT The development of secondary malignancies is an important complication after haematopoietic stem cell transplantation (HSCT) and curtails the survival of patients otherwise cured of their original malignancies. The commonest secondary malignancies are therapyrelated myelodysplastic syndrome/acute myeloid leukemia, lymphomas, and solid tumors including squamous cell carcinoma (SCC), melanomas, and sarcomas [1].The occurrence of secondary malignancies has been attributed to cumulative toxicity of previous chemotherapy and radiotherapy. Risk factors include young age at the time of HSCT, and the use of total body irradiation during conditioning [1].We present two cases of carcinoma of the tongue, with chronic oral mucosal graft-versus-host disease (GVHD) as a possible risk factor.
CASE REPORTS Case 1A 45-year-old man, non-smoker and non-drinker, presented with acute myeloid leukemia in 1995, which relapsed in 1996. Allogeneic HSCT was performed, the donor being an HLA-identical sister. The conditioning was busulphan (Bu), cyclophosphamide (Cy), and total body irradiation (TBI), with cyclosporine and methotrexate as GVHD prophylaxis. He developed severe GVHD affecting the liver as well as extensive oral mucosal and skin GVHD (grade III), requiring steroid, thalidomide, and azathioprine for control. Subsequently, chronic GVHD affecting the skin and mouth supervened. Six years later, an ulcerative growth at the dorsum of the tongue was found in a background of lichenoid mucositis, which on biopsy showed welldifferentiated SCC. The tumor was T3N0M0, stage III. Total glossectomy, right functional neck dissection, and left submandibular gland dissection was