BACKGROUND: Histiocytic sarcoma (HS) is an exceedingly rare tumor and carries a dismal prognosis when patients present with advanced-stage disease. Because of the poor response rates to conventional chemotherapy and the rarity of the disease, no standard of care exists for patients with HS. The authors report the single-agent use of the anti-CD52 antibody alemtuzumab in 2 patients who had advanced-stage HS. METHODS: Two patients with chemotherapy-refractory, metastatic HS with tumors that expressed the CD52 antigen received a prolonged course of treatment with the anti-CD52 monoclonal antibody alemtuzumab. RESULTS: Resected tumor samples from both patients demonstrated CD52 expression. Both patients had marked responses to single-agent alemtuzumab. One patient had a complete response with no evidence of disease for >5 years. The second patient had a major response to alemtuzumab and also remained alive with no evidence of disease for >4 years. CONCLUSIONS: Further studies need to be performed to examine CD52 expression and function in HS as well as the role of alemtuzumab in a larger cohort of patients. However, the clinical impact of single-agent alemtuzumab in the 2 patients described in this report was very encouraging. Given the toxicity and frequent inefficacy of conventional chemotherapy, patients with incompletely resected HS should be strongly considered for alemtuzumab therapy.
BACKGROUND:Histiocytic sarcoma (HS) is an exceedingly rare tumor and carries a dismal prognosis when patients present with advanced-stage disease. Because of the poor response rates to conventional chemotherapy and the rarity of the disease, no standard of care exists for patients with HS. The authors report the single-agent use of the anti-CD52 antibody alemtuzumab in 2 patients who had advanced-stage HS. METHODS: Two patients with chemotherapy-refractory, metastatic HS with tumors that expressed the CD52 antigen received a prolonged course of treatment with the anti-CD52 monoclonal antibody alemtuzumab. RESULTS: Resected tumor samples from both patients demonstrated CD52 expression. Both patients had marked responses to single-agent alemtuzumab. One patient had a complete response with no evidence of disease for >5 years. The second patient had a major response to alemtuzumab and also remained alive with no evidence of disease for >4 years. CONCLUSIONS: Further studies need to be performed to examine CD52 expression and function in HS as well as the role of alemtuzumab in a larger cohort of patients. However, the clinical impact of single-agent alemtuzumab in the 2 patients described in this report was very encouraging. Given the toxicity and frequent inefficacy of conventional chemotherapy, patients with incompletely resected HS should be strongly considered for alemtuzumab therapy.
Authors: Mrinal Gounder; Ved Desai; Deborah Kuk; Narasimhan Agaram; Maria Arcila; Benjamin Durham; Mary L Keohan; Mark A Dickson; Sandra P D'Angelo; Neerav Shukla; Craig Moskowitz; Ariela Noy; Robert G Maki; Diego Adrianzen Herrera; Armando Sanchez; Anita Krishnan; Andrew Pourmoussa; Li-Xuan Qin; William D Tap Journal: Eur J Cancer Date: 2015-08-19 Impact factor: 9.162
Authors: Shree Bose; Joanna Robles; Chad M McCall; Anand S Lagoo; Daniel S Wechsler; Gary R Schooler; David Van Mater Journal: Pediatr Blood Cancer Date: 2018-09-30 Impact factor: 3.167