Skip to main content

Advertisement

SpringerLink
Log in

Oral verruciform xanthoma in chronic graft-versus-host disease patients

  • Review Article
  • Published:
Supportive Care in Cancer Aims and scope Submit manuscript

Abstract

Background

Verruciform xanthoma (VX) is an uncommon benign epithelial lesion which mainly appears in inflamed oral epithelium. In this study, our aim was to present new cases of oral VX (OVX) in chronic graft-versus-host disease (cGVHD) and review the literature.

Methods

We conducted a retrospective chart review of cGVHD patients (2012–2019) to reveal cases of OVX. The demographics, medical background, clinical presentation, treatment provided, and follow-up were obtained. Additionally, Medline was searched using the terms “graft-versus-host disease,” “verruciform xanthoma,” and “oral.” Of the articles, the above-mentioned demographic and clinical features were retrieved.

Results

The patient pool included 133 oral cGVHD patients. Three cGVHD patients (males, aged 15–49 years, post-hematologic malignancy) were diagnosed with OVX. All patients had oral mucosal lichenoid lesions, but not in close proximity to the VX lesion. Medline searches revealed 9 cases of OVX in cGVHD patients reported in the literature. Eleven of the 12 patients had oral mucosal lichenoid lesions. Four patients had multiple OVX lesions. All lesions were asymptomatic. Six patients had lesions in the masticatory mucosa (hard palate and gingiva) and 4 patients had lesions in the buccal mucosa.

Conclusions

To the best of our knowledge, this is the largest published OVX in cGVHD series, including 12 patients. It appears that despite the higher prevalence in cGVHD patients relative to the general population, OVXs do not necessarily develop in sites with lichenoid lesions. It is advised that clinicians be familiar with the clinical and histological features in order to consider OVX in the differential diagnosis of oral lesions in cGVHD patients.

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

Similar content being viewed by others

References

  1. Shafer WG (1971) Verruciform xanthoma. Oral Surg Oral Med Oral Pathol 31:784–789

    Article  CAS  Google Scholar 

  2. Buchner A, Hansen LS, Merrell PW (1981) Verruciform xanthoma of the oral mucosa. Report of five cases and review of the literature. Arch Dermatol 117:563–565

    Article  CAS  Google Scholar 

  3. Shahrabi Farahani S, Treister NS, Khan Z, Woo SB (2011) Oral verruciform xanthoma associated with chronic graft-versus-host disease: a report of five cases and a review of the literature. Head Neck Pathol 5:193–198

    Article  Google Scholar 

  4. Neville BW, Damm DD, Allen CM, Chi AC (2016) Epithelial pathology. In: Oral and maxillofacial pathology. 4th ed. Elsevier, St. Louis, pp 331–421

    Google Scholar 

  5. Shin HI, Choi KS, Nagatsuka H, Murata M, Nagai N (1997) Verruciform xanthoma of the oral mucosa: an immunohistochemical and ultrastructural study of two cases. Oral Oncol 33:279–283

    Article  CAS  Google Scholar 

  6. Oliveira PT, Jaeger RG, Cabral LA, Carvalho YR, Costa AL, Jaeger MM (2001) Verruciform xanthoma of the oral mucosa. Report of four cases and a review of the literature. Oral Oncol 37:326–331

    Article  CAS  Google Scholar 

  7. Belknap AN, Islam MN, Bhattacharyya I, Cohen DM, Fitzpatrick SG (2020) Oral verruciform xanthoma: a series of 212 cases and review of the literature. Head Neck Pathol 14:742–748. https://doi.org/10.1007/s12105-019-01123-0

    Article  PubMed  Google Scholar 

  8. Val-Bernal JF, Argueta L, Val D, González-Vela MC, Garijo MF (2012) Verruciform xanthoma is another condition associated with pseudoepitheliomatous hyperplasia. Am J Dermatopathol 34:341–342

    Article  Google Scholar 

  9. Whitefield S, Raiser V, Shuster A, Kleinman S, Shlomi B, Kaplan I (2018) The spectrum of oral lesions presenting clinically with papillary-verrucous features. J Oral Maxillofac Surg 76:545–552

    Article  Google Scholar 

  10. Philipsen HP, Reichart PA, Takata T, Ogawa I (2003) Verruciform xanthoma: biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan. Oral Oncol 39:325–336

    Article  CAS  Google Scholar 

  11. Helm KF, Höpfl RM, Kreider JW, Lookingbill DP (1993) Verruciform xanthoma in an immunocompromised patient: a case report and immunohistochemical study. J Cutan Pathol 20:84–86

    Article  CAS  Google Scholar 

  12. Mannes KD, Dekle CL, Requena L, Sangueza OP (1999) Verruciform xanthoma associated with squamous cell carcinoma. Am J Dermatopathol 21:66–69

    Article  CAS  Google Scholar 

  13. Allen CM, Kapoor N (1993) Verruciform xanthoma in a bone marrow transplant recipient. Oral Surg Oral Med Oral Pathol 75:591–594

    Article  CAS  Google Scholar 

  14. Sibaud V, Marit G, Deminière C, Campana F, Taieb A, Fricain JC (2006) Multiple verruciform xanthomas of the oral mucosa associated with graft versus host disease. Ann Dermatol Venereol 133:995–999

    Article  CAS  Google Scholar 

  15. Capocasale G, Panzarella V, Tozzo P, Mauceri R, Rodolico V, Lauritano D, Campisi G (2017) Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: a rare case report and review of the literature. BMC Res Notes 10:631

    Article  Google Scholar 

  16. Jenkyn I, Woodward G, Kelly A, Fowell C (2019) Oral verruciform xanthoma associated with stem cell transplantation and graft versus host disease in a paediatric patient. Bone Marrow Transplant. https://doi.org/10.1038/s41409-019-0772-1

  17. Elad S, Aljitawi O, Zadik Y. Oral graft versus host disease: a pictorial review and a guide for dental practitioners. Int Dent J, in press

  18. Elad S, Zadik Y, Yarom N (2017) Oral complications of nonsurgical cancer therapies. Atlas Oral Maxillofac Surg Clin North Am 25:133–147

    Article  Google Scholar 

  19. Neville BW, Weathers DR (1980) Verruciform xanthoma. Oral Surg Oral Med Oral Pathol 49:429–434

    Article  CAS  Google Scholar 

  20. Stoopler ET, Desai B (2012) A tongue mass in a patient with oral lichen planus. J Can Dent Assoc 78:c60

    PubMed  Google Scholar 

  21. Mainville GN (2019) Non-HPV papillary lesions of the oral mucosa: clinical and histopathologic features of reactive and neoplastic conditions. Head Neck Pathol 13:71–79

    Article  Google Scholar 

  22. Theofilou VI, Sklavounou A, Argyris PP, Chrysomali E (2018) Oral verruciform xanthoma within lichen planus: a case report and literature review. Case Rep Dent 2018:1615086

    PubMed  PubMed Central  Google Scholar 

  23. Arai Y, Kondo T, Shigematsu A, Tanaka J, Ohashi K, Fukuda T, Hidaka M, Kobayashi N, Iwato K, Sakura T, Onizuka M, Ozawa Y, Eto T, Kurokawa M, Kahata K, Uchida N, Atsuta Y, Mizuta S, Kako S (2018) Improved prognosis with additional medium-dose VP16 to CY/TBI in allogeneic transplantation for high risk ALL in adults. Am J Hematol 93:47–57

    Article  CAS  Google Scholar 

  24. Betz SJ (2019) HPV-related papillary lesions of the oral mucosa: a review. Head Neck Pathol 13:80–90

    Article  Google Scholar 

  25. Elad S, Meyerowitz C, Shapira MY, Glick M, Bitan M, Amir G (2008) Oral posttransplantation lymphoproliferative disorder: an uncommon site for an uncommon disorder. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 105:59–64

    Article  Google Scholar 

  26. Demarosi F, Lodi G, Carrassi A, Soligo D, Sardella A (2005) Oral malignancies following HSCT: graft versus host disease and other risk factors. Oral Oncol 41:865–877

    Article  Google Scholar 

  27. Elad S, Zadik Y, Zeevi I, Miyazaki A, de Figueiredo MA, Or R (2010) Oral cancer in patients after hematopoietic stem-cell transplantation: long-term follow-up suggests an increased risk for recurrence. Transplantation 90:1243–1244

    Article  Google Scholar 

  28. Warnakulasuriya S (2020) Oral potentially malignant disorders: a comprehensive review on clinical aspects and management. Oral Oncol, in press 102:104550

    Article  Google Scholar 

  29. Carpenter PA, Kitko CL, Elad S, Flowers MED, Gea-Banacloche JC, Halter JP, Hoodin F, Johnston L, Lawitschka A, McDonald GB, Opipari AW, Savani BN, Schultz KR, Smith SR, Syrjala KL, Treister N, Vogelsang GB, Williams KM, Pavletic SZ, Martin PJ, Lee SJ, Couriel DR (2015) National Institutes of Health Consensus Development Project on criteria for clinical trials in chronic graft-versus-host disease: V. The 2014 Ancillary Therapy and Supportive Care Working Group Report. Biol Blood Marrow Transplant 21:1167–1187

    Article  Google Scholar 

  30. Zadik Y, Elad S, Shapira A, Shapira MY (2017) Treatment of oral mucosal manifestations of chronic graft-versus-host disease: dexamethasone vs. budesonide. Expert Opin Pharmacother 18:235–242

    Article  CAS  Google Scholar 

  31. Zadik Y (2020) Pharmacotherapy of oral mucosal manifestations of chronic graft-versus-host disease: when? What? and How? Expert Opin Pharmacother, In press

Download references

Acknowledgment

The authors thank Dr. Alexander Maly, from the Department of Pathology, Hebrew University-Hadassah Medical Center, Jerusalem, Israel.

Author information

Authors and Affiliations

  1. Department of Oral Medicine, Sedation and Maxillofacial Imaging, Hebrew University–Hadassah School of Dental Medicine, Jerusalem, Israel

    Ori Bar, Aiham Hanut & Yehuda Zadik

  2. Division of Oral Medicine, Eastman Institute for Oral Health, University of Rochester Medical Center, Rochester, NY, USA

    Sharon Elad

  3. Department of Bone Marrow Transplantation and Immunotherapy, Hebrew University-Hadassah Medical Center, Jerusalem, Israel

    Batia Avni

  4. Faculty of Medicine, The Hebrew University of Jerusalem, Jerusalem, Israel

    Batia Avni & Yehuda Zadik

  5. Department of Oral and Maxillofacial Surgery, Hebrew University-Hadassah School of Dental Medicine and Hebrew University-Hadassah Medical Center, Jerusalem, Israel

    Jawad Abu-Tair & Bianca Zaharia

  6. Department of Military Medicine, Hebrew University-Hadassah School of Medicine, P.O. Box 12000, 91120, Jerusalem, Israel

    Yehuda Zadik

Authors
  1. Ori Bar
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Sharon Elad
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Batia Avni
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Jawad Abu-Tair
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Bianca Zaharia
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Aiham Hanut
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Yehuda Zadik
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Yehuda Zadik.

Ethics declarations

Conflict of interest

Dr. Elad is a consultant to Falk Pharma GmbH; Dr. Zadik was a co-investigator in the BUM5 study, sponsored by Falk Pharma, GmbH

Additional information

Publisher’s note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Bar, O., Elad, S., Avni, B. et al. Oral verruciform xanthoma in chronic graft-versus-host disease patients. Support Care Cancer 29, 79–84 (2021). https://doi.org/10.1007/s00520-020-05681-w

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00520-020-05681-w

Keywords

Search

Navigation