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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 1  |  Page : 27-29

Multiple oral verruciform xanthomas: A rare entity


Department of Dermatology, Seth G. S. Medical College and KEM Hospital, Mumbai, Maharashtra, India

Date of Submission06-Mar-2020
Date of Acceptance15-Apr-2020
Date of Web Publication02-Jun-2020

Correspondence Address:
Sunanda Arun Mahajan
Department of Dermatology, Seth G. S. Medical College and KEM Hospital, Mumbai, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdpdd.ijdpdd_31_20

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  Abstract 


Verruciform xanthoma (VX) is an uncommon benign lesion that can affect oral, genital mucosa, and rarely skin. The characteristic foam cells seen in papillary dermis are a hallmark for diagnosing this entity. A 42-year-old male, tobacco-chewer presented with asymptomatic white-circular plaques on the bilateral buccal mucosa and yellow papules on retromolar area which on histopathological evaluation showed parakeratotic epithelium with surface and deep invagination of columns of parakeratosis throughout the hyperplastic epidermis, few neutrophils in upper spinous layers, and uniform elongation of rete ridges. The rete ridges showed numerous lipid-laden CD 68-positive foam cells. Punch excision coupled with cryotherapy was done, which resulted in the complete resolution of lesions. We publish this uncommon benign entity for increased awareness among physicians to consider it as a differential of verrucous lesion in the mucosa. Our patient had two different morphology of lesions, both suggestive of VX with characteristic foam cells on histopathology.

Keywords: Cryotherapy, oral, verruciform xanthoma


How to cite this article:
Dave JS, Mahajan SA, Dongre AM, Rane M. Multiple oral verruciform xanthomas: A rare entity. Indian J Dermatopathol Diagn Dermatol 2020;7:27-9

How to cite this URL:
Dave JS, Mahajan SA, Dongre AM, Rane M. Multiple oral verruciform xanthomas: A rare entity. Indian J Dermatopathol Diagn Dermatol [serial online] 2020 [cited 2020 Aug 11];7:27-9. Available from: http://www.ijdpdd.com/text.asp?2020/7/1/27/285797




  Introduction Top


Verruciform xanthoma (VX) is an uncommon benign lesion that can affect oral, genital mucosa, and rarely skin. Shafer first diagnosed it in the oral cavity in 1971.[1] It usually presents in the middle-aged individuals as an isolated verrucous plaque which is commonly mistaken as either viral wart, leukoplakia, or verrucous carcinoma.[2] Multiple lesions have been associated with immunocompromised states such as postbone marrow transplant, hepatitis C carriers, snuff dipper's keratosis, oral pemphigus vulgaris, carcinoma in situ, lichen sclerosus, solar keratoses, epithelial nevus, and CHILD syndrome.[3]


  Case Report Top


A 42-year-old male patient has been reported to the outpatient department with a complaint of asymptomatic lesions on the bilateral buccal mucosa for 4 months [Figure 1]. There was a history of tobacco chewing thrice a day for the past 20 years. There was no history of burning or intolerance on eating spicy food. There was no history of any systemic illness. On examination, there were discrete annular and circular white-colored plaques on bilateral buccal mucosa opposite first and second premolars. On further retracting the buccal mucosa with a tongue depressor, multiple yellow-colored discrete and confluent papules were seen on bilateral posterior buccal mucosa opposite second and third molars [Figure 2]. The appearance of the lesions closely resembled either leukoplakia or leukokeratosis and Fordyce's spot; hence, biopsy was done to confirm the diagnosis.
Figure 1: Discrete annular white-colored plaques on the buccal mucosa measuring 0.5 cm in diameter

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Figure 2: Multiple yellow-colored discrete and confluent papules on posterior buccal mucosa and retromolar trigone

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On histopathological examination of white plaques and yellow papules, the hematoxylin and eosin-stained sections showed parakeratotic epithelium with surface and deep invagination of columns of parakeratosis throughout the hyperplastic epidermis, few neutrophils in upper spinous layers, and uniform elongation of rete ridges [Figure 3]. The rete ridges showed numerous lipid-laden foam cells [Figure 4]. The foam cells in the papillary dermis stained positive for CD 68 [Figure 5]. There was infiltrate of lymphohistiocytes at the dermoepidermal junction.
Figure 3: Biopsy from both the lesions showed parakeratotic epithelium with surface and deep invagination of columns of parakeratosis throughout the hyperplastic epidermis, few neutrophils in upper spinous layers, and uniform elongation of rete ridges (H and E, ×40)

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{Figure 4}
Figure 5: The foam cells in the papillary dermis-stained positive for CD 68. (×40)

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The patient underwent punch excision of remaining lesions under local anesthesia. One month after the excision, new lesions appeared for which cryo dip was done for 4–6 sittings, which resulted in the resolution of lesions [Figure 6].
Figure 6: Posttreatment picture after punch excision and four sittings of cryo dip

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  Discussion Top


The morphological appearance of VX is variable. Philipsen et al. conducted a study of 282 patients and observed that the color of the lesion may be of normal mucosa or appear pink or yellowish brown or whitish when hyperkeratotic.[2] The surface may be granular or verrucous, sessile, or pedunculated. The lesion is raised above the normal surroundings and is well demarcated, although depressed and ulcerated lesions have been reported.[2] The most common reported site intraorally was the gingival margin (49.1%) followed by the hard palate, tongue, buccal mucosa, floor of the mouth, alveolar mucosa, soft palate, and lower lip in the order of frequency of occurrence.[2] Extraoral occurrence though rare, there are reports at sites such as the vulva, penis, scrotum, scalp, breast, ear, nose, thigh, forearm, and toe.[4] Inflammatory conditions, epithelial trauma, infections such as candidiasis, human papillomavirus infection, or conditions causing increased epithelial turnover such as carcinoma in situ have been implicated as possible etiologic agents.[3]

Histopathology of VX is characterized by epidermal hyperplasia with parakeratosis and the presence of neutrophils in the spinous layer.[5] The histological hallmark of VX is the characteristic CD 68 staining numerous foam cells in the elongated dermal papillae. These foam cells also called xanthoma cells are lipid-laden macrophages of various sizes with small-round eccentric nucleus with foamy cytoplasm.[1],[5] The foam cells arise as a result of local irritation through gradual degeneration of epithelial cells and subsequent release of the lipid material which is then picked up by the macrophage.[6] Nowparast et al. established three histomorphology patterns in a study of 54 cases, namely verrucous, papillary, and flat.[6] Lesions of verrucous type have a silhouette-like wart except the thickened granular layer seen in wart. Papillary form has multiple finger-like projections forming crypt-like spaces and have extension above the mucosal surface but minimal epithelial proliferation below the surface.[6] In the flat type, epithelial proliferation is seen below the surface with flat variable elongation of rete ridges.[6]

The treatment of choice for VX is complete surgical excision.[2] Other therapeutic options include CO2-laser ablation, cryotherapy, imiquimod, topical steroids, electrosurgery, and radiation therapy.

The variable morphological presentations of VX often challenge physician with many differentials, including malignancy such as squamous cell carcinoma and infections such as wart. Identifying the characteristic foam cells is vital in clinching the diagnosis of VX. We publish this uncommon benign entity for increased awareness among physicians to consider it as a differential of verrucous lesion in the mucosa. Our patient had two different morphologies of lesions, both suggestive of VX with characteristic foam cells on histopathology.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Rattana-Arpha P, Klanrit P, Suwannarong W. Oral verruciform xanthoma: A case report and a literature review. Bangkok Med J 2017;13:59-62.  Back to cited text no. 1
    
2.
Philipsen HP, Reichart PA, Takata T, Ogawa I. Verruciform xanthoma – Biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan. Oral Oncol 2003;39:325-36.  Back to cited text no. 2
    
3.
Hegde U, Doddawad VG, Sreeshyla H, Patil R. Verruciform xanthoma: A view on the concepts of its etiopathogenesis. J Oral Maxillofac Pathol 2013;17:392-6.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Li Z, Wang Y. Verruciform xanthoma of the thumb. Indian J Dermatol Venereol Leprol 2018;84:67-9.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Joshi R, Ovhal A. Verruciform xanthoma: Report of five cases. Indian J Dermatol 2012;57:479-82.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Nowparast B, Howell FV, Rick GM. Verruciform xanthoma. Oral Surg Oral Med Oral Pathol 1981;51:619-25.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 5], [Figure 6]



 

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