Skip to main content
SpringerLink
Log in

Cutaneous Normolipemic Plane Xanthoma with Supraglottic Involvement in a Patient with Hand-Schüller-Christian Disease

A Case Report

  • Case Reports
  • NPX in a Patient with Hand-Schüller-Christian Disease
  • Published:
American Journal of Clinical Dermatology Aims and scope Submit manuscript

Abstract

Normolipemic plane xanthoma (NPX) is a histiocytic disorder characterized by yellow-orange plaques in the periorbital areas, neck, upper trunk, and flexural folds. Association with systemic disease or paraproteinemia has been reported previously, but rarely with Langerhans cell histiocytosis (LCH). We report a case of Hand-Schüller-Christian disease (a type of LCH) in a patient who developed NPX with supraglottic involvement. NPX developed after several courses of chemotherapy and the supraglottic xanthoma occurred about 2 years later. The coexistence of LCH and non-LCH histiocytic lesions in this patient could be a result of chemotherapy-induced changes or may be just coincidental.

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

Access this article

Log in via an institution

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

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Table I

Similar content being viewed by others

References

  1. Altman J, Winkelmann RK. Diffuse normolipemic plane xanthoma. Arch Dermatol 1962; 85: 633–40

    Article  PubMed  CAS  Google Scholar 

  2. Elder D, editor. Lever’s histopathology of the skin. 9th ed. Philadelphia (PA): Lippincott-Raven Publishers, 2004: 698–9

    Google Scholar 

  3. Broeshart JH, Prens EP, Habets WJ, et al. Normolipemic plane xanthoma associated with adenocarcinoma and severe itch. J Am Acad Dermatol 2003; 49 (1): 119–22

    Article  PubMed  Google Scholar 

  4. Caputo R, Grimalt R, Laterza A, et al. Mucocutaneous expression of Langerhans cell histiocytosis in adults. Eur J Dermatol 1994; 4: 528–31

    Google Scholar 

  5. Altman J, Winkelmann RK. Xanthomatous cutaneous lesions of histiocytosis X. Arch Dermatol 1963; 87: 164–70

    Article  PubMed  CAS  Google Scholar 

  6. Thannhauser SJ. “Normocholesteric xathomatoses” in lipidoses: diseases of the intracellular lipid metabolism. 3rd ed. New York: Grune & Stratton, Inc., 1958: 360

    Google Scholar 

  7. Stickler GB, Pinkel D. Reticuloendotheliosis complicated by xanthomatous biliary cirrhosis; report of a case. Gastroenterology 1959; 36 (5): 702–8

    PubMed  CAS  Google Scholar 

  8. Feiwei M. Xanthomatosis in cryoglobulinemia and other paraproteinemias with a report of a case. Br J Dermatol 1968; 80: 719–29

    Article  Google Scholar 

  9. Haqqani MT, Hunter RD. Normolipemic plane xanthoma and histiocytic lymphoma [letter]. Arch Dermatol 1976; 112 (10): 1470–1

    Article  PubMed  CAS  Google Scholar 

  10. Marcoval J, Moreno A, Bordas X, et al. Diffuse plane xanthoma: clinicopathologic study of 8 cases. J Am Acad Dermatol 1998; 39 (3): 439–42

    Article  PubMed  CAS  Google Scholar 

  11. Akhyani M, Daneshpazhooh M, Seirafi H, et al. Diffuse plane xanthoma in an otherwise healthy woman. Clin Exp Dermatol 2001; 26 (5): 405–7

    Article  PubMed  CAS  Google Scholar 

  12. Wilkinson SM, Atkinson A, Neary RH, et al. Normolipaemic plane xanthomas: an association with increased vascular permeability and serum lipoprotein(a) concentration. Clin Exp Dermatol 1992; 17 (3): 211–3

    Article  PubMed  CAS  Google Scholar 

  13. Kodama H, Nakagawa S, Tanioku K. Plane xanthomatosis with antilipoprotein autoantibody. Arch Dermatol 1972; 105: 722–7

    Article  PubMed  CAS  Google Scholar 

  14. Williford PM, White WL, Jorizzo JL, et al. The spectrum of normolipemic plane xanthoma. Am J Dermatopathol 1993; 15: 572–5

    Article  PubMed  CAS  Google Scholar 

  15. Walker AE, Sneddon IB. Skin xanthoma following erythroderma. Br J Dermatol 1968; 80: 580–7

    Article  PubMed  CAS  Google Scholar 

  16. Fleischmajor R, Hyman AB, Weidman AL. Normolipemic plane xanthoma. Arch Dermatol 1964; 89: 319–23

    Article  Google Scholar 

  17. Perrone G, Zagami M, Casale M, et al. Immunohistochemistry and differential diagnosis of a solitary flat laryngeal xanthoma: a case report. In Vivo 2007; 21 (1): 119–21

    PubMed  Google Scholar 

  18. Szyfter W, Wierzbicka M, Bem G. Rare case of xanthoma disseminatum with severe dyspnoea. Otolaryngologia Polska 2006; 60 (1): 67–70

    Google Scholar 

  19. Seaton ED, Pillai GJ, Chu AC. Treatment of xanthoma disseminatum with cyclophosphamide. Br J Dermatol 2004; 150 (2): 346–9

    Article  PubMed  CAS  Google Scholar 

  20. Cantarella G, Neglia CB, Marzano AV, et al. Bilateral laryngeal pseudoparalysis in xanthoma disseminatum treated by endoscopic laser medial arytenoidectomy. Ann Otol Rhinol Laryngol 2001; 110 (3): 263–7

    PubMed  CAS  Google Scholar 

  21. Matsumoto T, Nobukawa B, Kobayashi K, et al. Solitary polypoid xanthoma in the larynx. Histopathology 1999; 34 (5): 475–7

    Article  PubMed  CAS  Google Scholar 

  22. Tietge UJ, Maschek H, Schneider A, et al. Xanthoma disseminatum with marked mucocutaneous involvement. Dtsch Med Wochenschr 1998; 123 (45): 1337–42

    Article  PubMed  CAS  Google Scholar 

  23. Fleischmajer R, Schaefer EJ, Gal AE, et al. Normolipemic subcutaneous xanthomatosis. Am J Med 1983; 75 (6): 1065–70

    Article  PubMed  CAS  Google Scholar 

  24. Shani-Adir A, Chou P, Morgan E, et al. A child with both Langerhans and non-Langerhans cell histiocytosis. Pediatr Dermatol 2002; 19: 419–22

    Article  PubMed  Google Scholar 

  25. Patrizi A, Neri I, Bianchi F, et al. Langerhans cell histiocytosis and juvenile xanthogranuloma: two case reports. Dermatology 2004; 209 (1): 57–61

    Article  PubMed  Google Scholar 

Download references

Acknowledgments

No sources of funding were used in the preparation of this case report. The authors have no conflicts of interest that are directly relevant to the content of this case report.

Author information

Authors and Affiliations

  1. Department of Dermatology, Chang Gung Memorial Hospital and Chang Gung University College of Medicine, 199 Tung-Hwa North Road, Taipei 105, Taiwan

    Yueh-Tsung Lu,  Wen-Hung Chung & Hong-Shang Hong

  2. Department of Dermatology, Wanfang Medical Center and Taipei Medical University, Taipei, Taiwan

    Ting-Jui Chen

  3. Institute of Pharmacology, National Yang-Ming University, Taipei, Taiwan

    Ting-Jui Chen

  4. Department of Pathology, Chang Gung Memorial Hospital and Chang Gung University College of Medicine, Linkou, Taiwan

    Tseng-Tong Kuo

Authors
  1. Yueh-Tsung Lu
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Ting-Jui Chen
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Wen-Hung Chung
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Tseng-Tong Kuo
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Hong-Shang Hong
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Wen-Hung Chung.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Lu, YT., Chen, TJ., Chung, WH. et al. Cutaneous Normolipemic Plane Xanthoma with Supraglottic Involvement in a Patient with Hand-Schüller-Christian Disease. Am J Clin Dermatol 10, 189–192 (2009). https://doi.org/10.2165/00128071-200910030-00005

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.2165/00128071-200910030-00005

Keywords

Search

Navigation