A Case of Xanthoma Disseminatum with Laryngeal Involvement

후두에 발생한 파종성 황색종 1예

  • Park, Jin Su (Department of Otorhinolaryngology-Head and Neck Surgery, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine) ;
  • Lee, Young Woo (Department of Otorhinolaryngology-Head and Neck Surgery, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine) ;
  • Lee, Sang Hyuk (Department of Otorhinolaryngology-Head and Neck Surgery, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine) ;
  • Jin, Sung Min (Department of Otorhinolaryngology-Head and Neck Surgery, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine)
  • 박진수 (성균관대학교 의과대학 강북삼성병원 이비인후과학교실) ;
  • 이용우 (성균관대학교 의과대학 강북삼성병원 이비인후과학교실) ;
  • 이상혁 (성균관대학교 의과대학 강북삼성병원 이비인후과학교실) ;
  • 진성민 (성균관대학교 의과대학 강북삼성병원 이비인후과학교실)
  • Received : 2015.05.15
  • Accepted : 2015.05.19
  • Published : 2015.06.01

Abstract

Xanthoma disseminatum (XD) is a benign, non-Langerhans cell histiocytosis characterized by disseminated xanthomatous lesions with face, flexures, and mucosa. Most of XD develops in mucocutaneous lesions including skin, oral cavity and pharynx, however laryngeal involvement is uncommon. While the natural course of XD is usually benign and often self-limiting, but XD develop in critical anatomical locations may result in morbidity and mortality. Localized mucous lesions in oropharynx and larynx lead to dysphagia, dyspnea and air way obstruction. The diagnosis of XD was based on clinical, histological and immunohistochemical findings. The treatment is complex and non-consensual. Local treatment with cryotherapy, radiotherapy, surgery, and carbon dioxide lasers have been attempted with various results. Systemic medication with peroxisome proliferator-activated gamma receptors, statins, fenofibrate, chlorodeoxyadenosine, cyclophosphamide, doxycycline, and cyclosporine have also been reported, but none have proven particularly successful. A 59-year-old man presented with respiratory symptoms because of laryngeal involvement of XD. We had to remove the obstructive lesion for relieving the symptoms. We experienced XD in Larynx that was rare in otorhinolaryngology. So we report this case with review of literatures.

Keywords

References

  1. Montgomery H OA. Xanthomathous correlation of clinical, histophathological and chemical studies of cutaneous xanthoma. Arch Derm Syphilol 1938(37):373-402.
  2. Seaton ED, Pillai GJ, Chu AC. Treatment of xanthoma disseminatum with cyclophosphamide. The British Journal of Dermatology 2004;150(2):346-9. https://doi.org/10.1111/j.1365-2133.2004.05772.x
  3. Ansarin H, Berenji Ardestani H, Tabaie SM, Shayanfar N. Xanthoma disseminatum with tumor-like lesion on face. Case Reports in Dermatological Medicine 2014;2014:621798.
  4. Kalz F, Hoffman MM, Lafrance A. Xanthoma disseminatum. Clinical and laboratory observations over a ten year period. Dermatologica 1970;140(3):129-41. https://doi.org/10.1159/000252546
  5. Caputo R, Veraldi S, Grimalt R, Gianotti R, Tosti A, Varotti C, et al. The various clinical patterns of xanthoma disseminatum. Considerations on seven cases and review of the literature. Dermatology 1995;190(1):19-24. https://doi.org/10.1159/000246628
  6. Park M, Boone B, Devos S. Xanthoma disseminatum: case report and mini-review of the literature. Acta Dermatovenerologica Croatica: ADC 2014;22(2):150-4.
  7. Natanzi N, Peng D, Ahdoot E, Ghatan S, Reinstandler A, Ram R. Xanthoma disseminatum in a pair of blind, deaf male twins. ISRN Dermatology 2011;2011:342909.
  8. Kang TW KS. A case of xanthoma disseminatum presenting as pedunculating nodules and plaques. Korean J Dermatol 2007;45:290-3.
  9. Giller RH, Folberg R, Keech RV, Piette WW, Sato Y. Xanthoma disseminatum. An unusual histiocytosis syndrome. The American Journal of Pediatric Hematology/Oncology 1988;10(3):252-7. https://doi.org/10.1097/00043426-198823000-00014
  10. Zak IT, Altinok D, Neilsen SS, Kish KK. Xanthoma disseminatum of the central nervous system and cranium. AJNR American Journal of Neuroradiology 2006;27(4):919-21.
  11. Davies CW, Marren P, Juniper MC, Gray W, Wojnorowska F, Benson MK,et al. Xanthoma disseminatum with respiratory tract involvement and fatal outcome. Thorax 2000;55(2):170-2. https://doi.org/10.1136/thorax.55.2.170
  12. Mahajan VK, Sharma AL, Chauhan PS, Mehta KS, Sharma V, Sharma S, et al. Xanthoma disseminatum: a red herring xanthomatosis. Indian Journal of Dermatology, Venereology and Leprology 2013;79(2):253-4. https://doi.org/10.4103/0378-6323.107655
  13. Khezri F, Gibson LE, Tefferi A. Xanthoma disseminatum: effective therapy with 2-chlorodeoxyadenosine in a case series. Archives of Dermatology 2011;147(4):459-64. https://doi.org/10.1001/archdermatol.2010.378
  14. Kim WJ, Ko HC, Kim BS, Kim MB. Successful treatment of xanthoma disseminatum with combined lipid lowering agents. Annals of Dermatology 2012;24(3):380-2. https://doi.org/10.5021/ad.2012.24.3.380
  15. Caputo R, Veraldi S, Grimalt R, Fianotti R, Tosti A, Varotti C, et al. The various clinical patterns of xanthoma disseminatum: considerations on seven cases and review of the literature. Dermatology 1995;190:19-24. https://doi.org/10.1159/000246628
  16. Hammond RR, Mackenzie IR. Xanthoma disseminatum with massive intracranial involvement. Clinical Neuropathology 1995;14(6): 314-21.
  17. Kuligowski M, Gorkiewicz-Petkow A, Jablonska S. Xanthoma disseminatum. International Journal of Dermatology 1992;31(4):81-3.
  18. Carpo BG, Grevelink SV, Brady S, Gellis S, Grevelink JM. Treatment of cutaneous lesions of xanthoma disseminatum with a CO2 laser. Dermatologic surgery: official publication for American Society for Dermatologic Surgery 1999;25(10):751-4. https://doi.org/10.1046/j.1524-4725.1999.99082.x
  19. Cantarella G, Neglia CB, Marzano AV, Ottaviani A. Bilateral laryngeal pseudoparalysis in xanthoma disseminatum treated by endoscopic laser medial arytenoidectomy. The Annals of Otology, Rhinology, and Laryngology 2001;110(3):263-7. https://doi.org/10.1177/000348940111000311