title: Gastrointestinal involvement in disseminated Langerhans cell histiocytosis (LCH) with durable complete response to 2-chlorodeoxyadenosine and high-dose cytarabine.
abstract: Successful treatment of infants with gastrointestinal involvement in Langerhans cell histiocytosis (LCH) has been poor, with no specific chemotherapeutic regimen of clear benefit. An 8-month-old male, diagnosed with LCH by skin and gastrointestinal biopsies, was treated with several cycles of 2-chlorodeoxyadenosine, vinblastine and prednisone with only partial response. Ultimately, two cycles of 2-chlorodeoxyadenosine concomitant with high-dose cytarabine led to a durable complete response. Twenty-seven months since the last course of chemotherapy, the patient continues to thrive free of disease. Treatment with 2-chlorodeoxyadenosine and cytarabine should be considered for further study in patients with poor-prognosis LCH.
authors.affiliation: Department of Pediatrics, New York University Medical Center, New York, New York, USA
authors.forename: Sung W
authors.initials: SW
authors.lastname: Choi
authors.forename: Babu S
authors.initials: BS
authors.lastname: Bangaru
authors.forename: C Daniel
authors.initials: CD
authors.lastname: Wu
authors.forename: Jonathan L
authors.initials: JL
authors.lastname: Finlay
mesh_terms: D001706:Biopsy; D017338:Cladribine / Q000627:therapeutic use*; D003561:Cytarabine / Q000627:therapeutic use*; D004359:Drug Therapy, Combination; D005767:Gastrointestinal Diseases / Q000188:drug therapy* / Q000209:etiology; D006646:Histiocytosis, Langerhans-Cell / Q000150:complications / Q000188:drug therapy*; D006801:Humans; D007166:Immunosuppressive Agents / Q000627:therapeutic use*; D007223:Infant; D008297:Male
chemical_list: D007166:Immunosuppressive Agents; D003561:Cytarabine; D017338:Cladribine