Disseminated xanthogranulomas associated with adult T‐cell leukaemia/lymphoma: a case report and review the association of haematologic malignancies

Xanthogranuloma (XG) is rarely observed in adults and has been reported to be associated with chronic myelogenous leukaemia (CML) and/or neurofibromatosis type 1 (NF1). A 68‐year‐old woman with adult T‐cell leukaemia/lymphoma (ATLL) gradually developed disseminated XGs over the 3 years since disease onset. Histopathological examination of a skin biopsy revealed the presence of histiocytes in the dermis with a few Touton giant cells admixed with lymphoid cells. The lesions of XGs persisted despite chemotherapy with prednisolone and chlorambucil for her ATLL. This is the first report of disseminated XGs associated with ATLL. The association of disseminated XGs with haematologic malignancies was reviewed and the possible pathogenesis of this association will be discussed.

[1]  A. Nouri,et al.  [Juvenile xanthogranuloma]. , 2008, Presse medicale.

[2]  K. Karube,et al.  Clinicopathological features of cutaneous lesions of adult T‐cell leukaemia/ lymphoma , 2005, The British journal of dermatology.

[3]  A. Patrizi,et al.  Langerhans Cell Histiocytosis and Juvenile Xanthogranuloma , 2004, Dermatology.

[4]  K. Karube,et al.  Expression of FoxP3, a key molecule in CD4+CD25+ regulatory T cells, in adult T‐cell leukaemia/lymphoma cells , 2004, British journal of haematology.

[5]  P. Cruz,et al.  Concurrent development of eruptive xanthogranulomas and hematologic malignancy: two case reports. , 2004, Journal of the American Academy of Dermatology.

[6]  P. Korkolopoulou,et al.  Adult T-Cell Leukemia/Lymphoma (ATLL): Report of Two Fully Documented Hellenic Patients , 2004, Leukemia & lymphoma.

[7]  R. Caputo,et al.  Juvenile Xanthogranuloma Associated with Neurofibromatosis 1: 14 Patients without Evidence of Hematologic Malignancies , 2004, Pediatric dermatology.

[8]  A. Patrizi,et al.  Langerhans cell histiocytosis and juvenile xanthogranuloma. Two case reports. , 2004, Dermatology.

[9]  P. Humbert,et al.  Leucémie myélomonocytaire juvénile, xanthomes et neurofibromatose de type 1 , 2003 .

[10]  F. Aubin,et al.  [Juvenile myelomonocytic leukaemia, xanthoma, and neurofibromatosis type 1]. , 2003, Archives de pediatrie : organe officiel de la Societe francaise de pediatrie.

[11]  B. Mahé,et al.  [Eruptive xanthogranulomatosis in a trisomy 21 patient with acute lymphoblastic leukemia]. , 2000, Annales de dermatologie et de vénéréologie.

[12]  B. Bostrom,et al.  Juvenile xanthogranuloma: forms of systemic disease and their clinical implications. , 1996, The Journal of pediatrics.

[13]  A. Zvulunov.  Juvenile xanthogranuloma, neurofibromatosis, and juvenile chronic myelogenous leukemia. , 1996, Archives of dermatology.

[14]  A. Donfrancesco,et al.  Sporadic acute lymphocytic leukemia arising in a patient with neurofibromatosis and xanthogranulomatosis. , 1996, Cancer investigation.

[15]  A. Zvulunov,et al.  Juvenile xanthogranuloma, neurofibromatosis, and juvenile chronic myelogenous leukemia. World statistical analysis. , 1995, Archives of dermatology.

[16]  T. Saito,et al.  Adult-onset xanthogranuloma appearing symmetrically on the ear lobes. , 1995, Journal of the American Academy of Dermatology.

[17]  P. O'Connell,et al.  Loss of the normal NF1 allele from the bone marrow of children with type 1 neurofibromatosis and malignant myeloid disorders. , 1994, The New England journal of medicine.

[18]  G. D. de Vaan,et al.  Juvenile xanthogranuloma and acute leukemia: a case report. , 1994, Medical and pediatric oncology.

[19]  T. Ono,et al.  Cutaneous manifestations of adult T-cell leukemia/lymphoma , 1992 .

[20]  L. Shih,et al.  Human T‐cell lymphotropic virus type I associated adult T‐cell leukaemia/lymphoma in Taiwan Chinese , 1991, British journal of haematology.

[21]  P. Morier,et al.  Juvenile chronic granulocytic leukemia, juvenile xanthogranulomas, and neurofibromatosis. Case report and review of the literature. , 1990, Journal of the American Academy of Dermatology.

[22]  B. Cohen,et al.  Xanthogranuloma: Report on Clinical and Histologic Findings in 64 Patients , 1989, Pediatric dermatology.

[23]  R. Caputo,et al.  Histiocytic syndromes: a review. , 1985, Journal of the American Academy of Dermatology.

[24]  L. Shih,et al.  SEROLOGICAL SURVEY OF ANTIBODIES TO THE ADULT T‐CELL LEUKEMIA VIRUS‐ASSOCIATED ANTIGEN (HTLV‐A) IN TAIWAN , 1985, International journal of cancer.

[25]  M. Sakai,et al.  JUVENILE XANTHOGRANULOMA WITH DARIER'S SIGN , 1983, The Journal of dermatology.

[26]  H. Balfour,et al.  Juvenile xanthogranuloma associated with cytomegalovirus infection. , 1971, The American journal of medicine.