Skip to main content

Advertisement

Log in

Indeterminate Cell Histiocytosis in a Pediatric Patient: Successful Treatment with Thalidomide

  • Case Report
  • Published:
Pathology & Oncology Research

Abstract

The 15-year-old male patient presented several 2–6 mm large livid reddish-yellowish, shiny, compact papules on the head, trunk and extremities, which had developed within the last 4 months. Histology showed normal epidermis with dense dermal infiltrate of histiocytes accompanied by few eosinophils, Touton or foamy giant cells. The histiocytes were S100 positive, CD1a negative and did not contain Birbeck granules ultrastructurally. Chest X ray, EEG, skull MRI did not show pathology. Opthalmology, neurology, oto-rhino-laryngology did not reveal alterations. Based upon the clinical symptoms and the histopathology, the diagnosis of indeterminate cell histiocytosis was confirmed. Cryotherapy and cauterization did not stop the progression of the disease, however, under thalidomide treatment no new symptoms developed and the lesions healed with pigmentation.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5

Abbreviations

ICH:

Indetreminate cell histiocytosis

MRI:

Magnetic Resonance Imaging

EEG:

Electroencephalogram

CD1a:

Cluster of Differentiation 1a

CD4:

Cluster of Differentiation 4

CD68:

Cluster of Differentiation 68

References

  1. Gianotti F, Caputo R (1985) Histiocytic syndromes: a review. J Am Acad Dermatol 13:383–404

    Article  PubMed  CAS  Google Scholar 

  2. Bolognia JL, Jorizzo JL, Rapini RP (eds) (2008) Dermatology. Mosby Elsevier, London Edinburgh New York Philadelphia St Louis Sidney Toronto

  3. Ringel E, Moschella S (1985) Primary histiocytic dermatoses. Arch Dermatol 121:1531–1542

    Article  PubMed  CAS  Google Scholar 

  4. Groopman J, Golde D (1981) The histiocytic disorders: a pathophysiologic analysis. Ann Intern Med 94:95–107

    PubMed  CAS  Google Scholar 

  5. Coldiron BM, Santa-Cruz D, Freeman RG et al (1988) Benign non-X histiocytosis: a unique case bridging several of the non-X histiocytic syndromes. J Am Acad Dermatol 18:1282–1289

    Article  PubMed  CAS  Google Scholar 

  6. Manente L, Cotellessa C, Schmitt I et al (1997) Indeterminate cell histiocytosis: a rare histiocytic disorder. Am J Dermatopathol 19:276–283

    Article  PubMed  CAS  Google Scholar 

  7. Rodriguez-Jurado R, Vidaurri-de la Cruz H, Duran-Mckinster C et al (2003) Indeterminate cell histiocytosis. Clinical and pathologic study in a pediatric patient. Arch Pathol Lab Med 127(6):748–751

    PubMed  Google Scholar 

  8. Burgdorf WHC, Plewig G, Wolff HH, Landthaler M (eds) (2009) Braun-Falco’s dermatology. Springer, Heidelberg

    Google Scholar 

  9. Breatnach AS (1963) A new concept of the relation between the Langerhans cell and the melanocyte. J Invest Dermatol 40:279–281

    Google Scholar 

  10. Wood GS, Hu CH, Beckstead JH et al (1985) The indeterminate cell proliferative disorder: report of a case manifesting as an unusual cuteaneous histiocytosis. J Dermatol Surg Oncol 11:1111

    PubMed  CAS  Google Scholar 

  11. Ratzinger G, Burgdorf WH, Metze D et al (2005) Indeterminate cell histiocytosis: fact or fiction? J Cutan Pathol 32(8):552–560

    Article  PubMed  Google Scholar 

  12. Kolde G, Brocker EB (1986) Multiple skin tumors of indeterminate cells in an adult. J Am Acad Dermatol 15:591

    Article  PubMed  CAS  Google Scholar 

  13. Berti E, Gianotti R, Alessi E (1988) Unusual cutaneous histiocytosis expressing an intermediate immunophenotype between Langerhans cells and dermal macrophages. Arch Dermatol 124:1250–1253

    Article  PubMed  CAS  Google Scholar 

  14. Sidoroff A, Zelger B, Steiner H et al (1996) Indeterminate cell histiocytosis—a clinicopathological entity with features of both X- and non-X histiocytosis. Br J Dermatol 134:525

    Article  PubMed  CAS  Google Scholar 

  15. Calatayud M, Güell JL, Gris O et al (2001) Ocular involvement in a case of systemic indeterminate cell hitiocytosis: case report. Cornea 20:769

    Article  PubMed  CAS  Google Scholar 

  16. Zelger B, Burgdorf WHC (2001) The cutaneous histiocytoses. Adv Dermatol 17:77

    PubMed  CAS  Google Scholar 

  17. Segal GH, Mesa MV, Fishleder AJ et al (1992) Precursor Langerhans cell histiocytosis. An unusual histiocytic proliferation in a patient with persistent non-Hodgkin lymphoma and terminal acute monocytic leukemia. Cancer 70:547–553

    Article  PubMed  CAS  Google Scholar 

  18. Vener C, Soligo D, Berti E et al (2007) Indeterminate cell histiocytosis in association with later occurrence of acute myeloblastic leukaemia. Br J Dermatol 156(6):1357–1361

    Article  PubMed  CAS  Google Scholar 

  19. Broekaert SM, Metzler G, Burgdorf W et al (2007) Multisystem Langerhans cell histiocytosis: successful treatment with thalidomide. Am J Clin Dermatol 8(5):311–314

    Article  PubMed  Google Scholar 

  20. Wang CH, Chen GS (2004) Indeterminate cell histiocytosis: a case report. J Med Sci 20:24–30

    Google Scholar 

  21. Malhomme de la Roche H, Lai-Cheong JE, Calonje E et al (2008) Indeterminate cell histiocytosis responding to total skin electron beam therapy. Br J Dermatol 158(4):838–840

    Article  PubMed  CAS  Google Scholar 

  22. Eichholz A, Merchant S, Gaya AM (2010) Anti-angiogenesis therapies: their potential in cancer management. Onco Targets Ther 24(3):69–82

    Google Scholar 

  23. Ventura F, Pereira T, da Luz Duarte M et al (2010) Indeterminate cell histiocytosis in association with acute myeloid leukemia. Dermatol Res Pract 2010:569345. Epub 2010 Jun 21

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Béla Tóth.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Tóth, B., Katona, M., Hársing, J. et al. Indeterminate Cell Histiocytosis in a Pediatric Patient: Successful Treatment with Thalidomide. Pathol. Oncol. Res. 18, 535–538 (2012). https://doi.org/10.1007/s12253-011-9405-8

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s12253-011-9405-8

Keywords

Navigation