Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: A rare case report and review of the literature

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Abstract

Background: Oral verruciform xanthoma is an uncommon benign lesion. Although oral verruciform xanthoma occurs in healthy individuals, it has been also reported in association with some inflammatory conditions. The aim of this study is to report a case of oral verruciform xanthoma associated with chronic graft-versus-host disease and to review the literature on this topic. Case presentation: A 47-year-old Caucasian male presented to the Sector of Oral Medicine "V. Margiotta", University Policlinic "P. Giaccone" of Palermo complaining of a mass on the gingiva. He first noticed the painless mass 1 year ago. He reported to have undergone allogenic hematopoietic stem cell transplantation 15 years ago for acute lymphoblastic leukaemia. Intraoral examination revealed a well-circumscribed, sessile yellowish and verrucous nodule upon canine, multiple yellowish and verrucous nodules on the hard palate, yellowish and verrucous nodules on left buccal mucosa. In addiction an area of white striae in a reticular pattern with erythema and ulceration was present on the dorsum of the tongue. This lesion was consistent with a known history of oral chronic graft versus host disease. Moreover, we observed a suspected area of oral erythroplakia yet on the dorsum of the tongue. In biopsy specimen of hard palate histopathological examination revealed a diagnosis of verrucous xanthoma of the oral cavity; in addiction in biopsy specimen of the dorsum of the tongue revealed the presence of erythroplakia with high grade dysplasia. Conclusion: Verruciform xanthoma of the oral cavity associated with chronic graft-versus-host disease is a rare condition with a usually benign clinical course but malignant transformation has been described in association with oral potential malignant disorder (e.g. chronic graft versus host disease, erythroplakia). Very rare cases showed association with oral chronic graft versus-host-disease. To date, only eight cases were published in the world literature. Therefore it could be important follow up patients also for oral verruciform xanthoma onset.
Lingua originaleEnglish
pagine (da-a)-
Numero di pagine0
RivistaBMC Research Notes
Volume10
Stato di pubblicazionePublished - 2017

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Xanthomatosis
Graft vs Host Disease
Grafts
Tongue
Biopsy
Hard Palate
Mouth
Oral Medicine
Stem cells
Hematopoietic Stem Cell Transplantation
Mouth Mucosa
Medicine
Gingiva
Erythema
Precursor Cell Lymphoblastic Leukemia-Lymphoma
Canidae
Interviews

All Science Journal Classification (ASJC) codes

  • Biochemistry, Genetics and Molecular Biology(all)

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title = "Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: A rare case report and review of the literature",
abstract = "Background: Oral verruciform xanthoma is an uncommon benign lesion. Although oral verruciform xanthoma occurs in healthy individuals, it has been also reported in association with some inflammatory conditions. The aim of this study is to report a case of oral verruciform xanthoma associated with chronic graft-versus-host disease and to review the literature on this topic. Case presentation: A 47-year-old Caucasian male presented to the Sector of Oral Medicine {"}V. Margiotta{"}, University Policlinic {"}P. Giaccone{"} of Palermo complaining of a mass on the gingiva. He first noticed the painless mass 1 year ago. He reported to have undergone allogenic hematopoietic stem cell transplantation 15 years ago for acute lymphoblastic leukaemia. Intraoral examination revealed a well-circumscribed, sessile yellowish and verrucous nodule upon canine, multiple yellowish and verrucous nodules on the hard palate, yellowish and verrucous nodules on left buccal mucosa. In addiction an area of white striae in a reticular pattern with erythema and ulceration was present on the dorsum of the tongue. This lesion was consistent with a known history of oral chronic graft versus host disease. Moreover, we observed a suspected area of oral erythroplakia yet on the dorsum of the tongue. In biopsy specimen of hard palate histopathological examination revealed a diagnosis of verrucous xanthoma of the oral cavity; in addiction in biopsy specimen of the dorsum of the tongue revealed the presence of erythroplakia with high grade dysplasia. Conclusion: Verruciform xanthoma of the oral cavity associated with chronic graft-versus-host disease is a rare condition with a usually benign clinical course but malignant transformation has been described in association with oral potential malignant disorder (e.g. chronic graft versus host disease, erythroplakia). Very rare cases showed association with oral chronic graft versus-host-disease. To date, only eight cases were published in the world literature. Therefore it could be important follow up patients also for oral verruciform xanthoma onset.",
author = "Vito Rodolico and Giuseppina Campisi and Vera Panzarella and Rodolfo Mauceri and Giorgia Capocasale and Dorina Lauritano",
year = "2017",
language = "English",
volume = "10",
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journal = "BMC Research Notes",
issn = "1756-0500",
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TY - JOUR

T1 - Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: A rare case report and review of the literature

AU - Rodolico, Vito

AU - Campisi, Giuseppina

AU - Panzarella, Vera

AU - Mauceri, Rodolfo

AU - Capocasale, Giorgia

AU - Lauritano, Dorina

PY - 2017

Y1 - 2017

N2 - Background: Oral verruciform xanthoma is an uncommon benign lesion. Although oral verruciform xanthoma occurs in healthy individuals, it has been also reported in association with some inflammatory conditions. The aim of this study is to report a case of oral verruciform xanthoma associated with chronic graft-versus-host disease and to review the literature on this topic. Case presentation: A 47-year-old Caucasian male presented to the Sector of Oral Medicine "V. Margiotta", University Policlinic "P. Giaccone" of Palermo complaining of a mass on the gingiva. He first noticed the painless mass 1 year ago. He reported to have undergone allogenic hematopoietic stem cell transplantation 15 years ago for acute lymphoblastic leukaemia. Intraoral examination revealed a well-circumscribed, sessile yellowish and verrucous nodule upon canine, multiple yellowish and verrucous nodules on the hard palate, yellowish and verrucous nodules on left buccal mucosa. In addiction an area of white striae in a reticular pattern with erythema and ulceration was present on the dorsum of the tongue. This lesion was consistent with a known history of oral chronic graft versus host disease. Moreover, we observed a suspected area of oral erythroplakia yet on the dorsum of the tongue. In biopsy specimen of hard palate histopathological examination revealed a diagnosis of verrucous xanthoma of the oral cavity; in addiction in biopsy specimen of the dorsum of the tongue revealed the presence of erythroplakia with high grade dysplasia. Conclusion: Verruciform xanthoma of the oral cavity associated with chronic graft-versus-host disease is a rare condition with a usually benign clinical course but malignant transformation has been described in association with oral potential malignant disorder (e.g. chronic graft versus host disease, erythroplakia). Very rare cases showed association with oral chronic graft versus-host-disease. To date, only eight cases were published in the world literature. Therefore it could be important follow up patients also for oral verruciform xanthoma onset.

AB - Background: Oral verruciform xanthoma is an uncommon benign lesion. Although oral verruciform xanthoma occurs in healthy individuals, it has been also reported in association with some inflammatory conditions. The aim of this study is to report a case of oral verruciform xanthoma associated with chronic graft-versus-host disease and to review the literature on this topic. Case presentation: A 47-year-old Caucasian male presented to the Sector of Oral Medicine "V. Margiotta", University Policlinic "P. Giaccone" of Palermo complaining of a mass on the gingiva. He first noticed the painless mass 1 year ago. He reported to have undergone allogenic hematopoietic stem cell transplantation 15 years ago for acute lymphoblastic leukaemia. Intraoral examination revealed a well-circumscribed, sessile yellowish and verrucous nodule upon canine, multiple yellowish and verrucous nodules on the hard palate, yellowish and verrucous nodules on left buccal mucosa. In addiction an area of white striae in a reticular pattern with erythema and ulceration was present on the dorsum of the tongue. This lesion was consistent with a known history of oral chronic graft versus host disease. Moreover, we observed a suspected area of oral erythroplakia yet on the dorsum of the tongue. In biopsy specimen of hard palate histopathological examination revealed a diagnosis of verrucous xanthoma of the oral cavity; in addiction in biopsy specimen of the dorsum of the tongue revealed the presence of erythroplakia with high grade dysplasia. Conclusion: Verruciform xanthoma of the oral cavity associated with chronic graft-versus-host disease is a rare condition with a usually benign clinical course but malignant transformation has been described in association with oral potential malignant disorder (e.g. chronic graft versus host disease, erythroplakia). Very rare cases showed association with oral chronic graft versus-host-disease. To date, only eight cases were published in the world literature. Therefore it could be important follow up patients also for oral verruciform xanthoma onset.

UR - http://hdl.handle.net/10447/249340

UR - http://www.biomedcentral.com/bmcresnotes/

M3 - Article

VL - 10

SP - -

JO - BMC Research Notes

JF - BMC Research Notes

SN - 1756-0500

ER -