title: Cytology of primary cutaneous Langerhans cell histiocytosis with a malignant phenotype. A case report.
abstract: BACKGROUND
  Langerhans cell histiocytosis (LCH) is a proliferative disorder of Langerhans cells, but the nature of LCH, whether reactive, benign, or malignant and neoplastic, is controversial. We encountered a case of LCH showing a malignant phenotype initially localized in the skin of an elderly woman. Since there is no other report on the cytologic appearance of primary cutaneous LCH or on LCH with a malignant phenotype, we compared the cytologic features of this case with those of benign cases at other sites reported in the literature.
  
  
  METHODS
  A 74-year-old woman presented with a gradually enlarging and partially ulcerated skin lesion expanding both sides of her right hand. On histologic and ultrastructural analyses of surgically resected tissue, we diagnosed the lesion as Langerhans cell histiocytosis originating in the skin. Although the patient had no recurrence or metastases for six months after surgical resection of the primary skin lesion and radiation therapy, the tumor extended multisystemically, and the patient died of multiple organ failure 14 months after the initial diagnosis.
  
  
  CONCLUSIONS
  Imprint and scrape cytology of multiple skin lesions six months after surgery was useful in immediately diagnosing the recurrent LCH. The tumor cells had indented, twisted or grooved nuclei, and some had intranuclear inclusions. Immunocytochemically the cells were positive for CD1a and S-100 protein. Numerous eosinophils were seen in the background.
authors.affiliation: Second Department of Pathology, Department of Dermatology, Miyazaki Medical College, Pathology Division and Laboratory for Clinical Investigation, Miyazaki Medical College Hospital, Miyazaki, Japan. hitoh@post.miyazaki-med.ac.jp
authors.forename: Hiroshi
authors.initials: H
authors.lastname: Itoh
authors.forename: Yuji
authors.initials: Y
authors.lastname: Hinoura
authors.forename: Yatsuki
authors.initials: Y
authors.lastname: Aratake
authors.forename: Shinya
authors.initials: S
authors.lastname: Sato
authors.forename: Akinobu
authors.initials: A
authors.lastname: Ohno
authors.forename: Hitoshi
authors.initials: H
authors.lastname: Miyaguni
authors.forename: Hiroaki
authors.initials: H
authors.lastname: Kataoka
authors.forename: Masashi
authors.initials: M
authors.lastname: Koono
mesh_terms: D000368:Aged; D018949:Antigens, CD1 / Q000032:analysis* / Q000276:immunology; D001707:Biopsy, Needle; D002467:Cell Nucleus / Q000648:ultrastructure; D005260:Female; D015614:Histiocytosis / Q000145:classification / Q000473:pathology*; D006801:Humans; D007150:Immunohistochemistry; D007801:Langerhans Cells / Q000473:pathology*; D010641:Phenotype; D012008:Recurrence; D009418:S100 Proteins / Q000032:analysis* / Q000276:immunology; D012878:Skin Neoplasms / Q000473:pathology*
chemical_list: D018949:Antigens, CD1; C419393:CD1a antigen; D009418:S100 Proteins