Home About us Editorial board Ahead of print Current issue Archives Submit article Instructions Subscribe Search Contacts Login 
  • Users Online: 869
  • Home
  • Print this page
  • Email this page


 
 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 32  |  Issue : 2  |  Page : 196-198

Oral verruciform xanthoma: The great imitator


1 Scandent Imaging Pvt. Ltd., Matunga, Navi Mumbai, Maharashtra, India
2 Department of Oral Medicine and Radiology, YMT Dental College and Research Center, Khargar, Navi Mumbai, Maharashtra, India
3 Department Oral Oncology Unit, SRJ CBCC Cancer Hospital and Research Center, Indore, Madhya Pradesh, India
4 Department of Oral Medicine and Radiology, Modern Dental College and Research Centre, Indore, Madhya Pradesh, India

Date of Submission15-Apr-2020
Date of Decision07-May-2020
Date of Acceptance12-May-2020
Date of Web Publication27-Jun-2020

Correspondence Address:
Dr. Sweety Lalawat
Scandent Imaging Pvt. Ltd., Mumbai, Maharashtra; Address: 102, Gumasta Nagar, Near Vyankatesh Dwar, Indore - 452009, (M.P)
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaomr.jiaomr_61_20

Rights and Permissions
   Abstract 


Oral Verruciform xanthoma (OVX) is an uncommon benign muco-cutaneous lesion having various concepts regarding its etiopathogenesis. Intraorally, this usually occurs on the gingiva and alveolar mucosa. Often asymptomatic, this lesion presents with a flat to verruco-papillary surface morphology. The clinical features of OVX can be misleading and resembles diverse hyperplastic, non-malignant, potentially malignant, and malignant lesions of the oral cavity. Histopathological examination is a key for the diagnosis of this lesion with xanthoma cells being the hallmark. Surgical excision remains the treatment modality and recurrence is rare. This paper presents a case of oral verruciform xanthoma on the buccal mucosa, mimicking a potentially malignant lesion, and a review of its major clinical, histological features and differential diagnosis.

Keywords: Foam cells, non- homogeneous leukoplakia, oral verruciform xanthoma, verruciform appearance, xanthoma cells


How to cite this article:
Lalawat S, Tomar N, Chaudhary A, Reddy V. Oral verruciform xanthoma: The great imitator. J Indian Acad Oral Med Radiol 2020;32:196-8

How to cite this URL:
Lalawat S, Tomar N, Chaudhary A, Reddy V. Oral verruciform xanthoma: The great imitator. J Indian Acad Oral Med Radiol [serial online] 2020 [cited 2020 Jul 15];32:196-8. Available from: http://www.jiaomr.in/text.asp?2020/32/2/196/288141




   Introduction Top


Verruciform xanthoma (VX) is a relatively rare, benign, hyperplastic condition of epithelium. It primarily affects the oral mucosa, the gingiva being the preferred site. Extraoral occurrence is uncommon and it mainly involves the anogenital mucosa and skin.[1],[2],[3]

Clinically, it usually appears as a well-demarcated solitary lesion with either a sessile or pedunculated base. The surface morphology ranges from flat to raised plaque or papule with verruco-papillary surface projections. Depending upon the degree of surface keratinization, the color of the lesion may appear as white, yellow-white, pink or red.[4]

This case report highlights the fact that this varied clinical appearance of VX is non-diagnostic and resembles other verruco-papillary lesions of the oral cavity. The diagnosis is almost always made on histologic examination which has distinct accumulation of foam cells.[1],[4]


   Case Report Top


A 50-year-old male patient reported to the department of oral medicine and radiology with a complaint of white lesion in his oral cavity. He noticed the lesion one year back and since then, there was no history of increase/decrease in the size of the lesion and/or any associated symptoms.

Patient was under regular medication for diabetes mellitus since 2 years. He had a habit of tobacco chewing mixed with lime since 40 years and used to place the quid in right buccal vestibule.

Extra-oral examination did not reveal any abnormality. Intra-oral examination revealed a single, well-demarcated, sessile, grayish white hyperkeratotic lesion with inter-mixed red tissue elements present in right buccal mucosa and vestibule, approx. 2 × 2 cms in dimensions. Overlying surface appeared to be raised, irregular and granular. Anterior and posterior aspect of lesion showed predominantly white, flat and smooth lesion. Lesion was non-tender, firm in consistency, non-indurated and non-scrapable [Figure 1].
Figure 1: Clinical appearance at initial visit

Click here to view


Owing to positive habit history and appearance of the lesion, a provisional diagnosis of non-homogenous leukoplakia on right buccal vestibule was made. Nodular candidiasis and focal verruco-papillary lesions were considered under differential diagnosis. The patient was advised to quit tobacco smoking habit completely and was prescribed application of topical anti-fungal agent (Clotrimazole) for 14 days. On follow-up after 14 days, the keratin component of the lesion showed evidence of reduction and the lesion appeared to have a granular surface with yellowish- pink color and keratotic areas [Figure 2].
Figure 2: Clinical appearance after topical anti-fungal treatment

Click here to view


An excisional biopsy was planned. The excised specimen was sent for histopathological examination which revealed, verrucous hyperparakeratotic stratified squamous epithelium with extremely elongated uniform retepegs. Between the connective tissue papillae there was abundance of large foam cells. This picture was characteristic for verruciform xanthoma, and therefore a final diagnosis of the same was made [Figure 3]. Post-excision healing was uneventful. However, after 3 months, there were signs of recurrence of the lesion.
Figure 3: Histopathology picture

Click here to view



   Discussion Top


Verruciform xanthoma is an uncommon lesion with an incidence rate of 0.025-0.05%. Various concepts exist within the literature regarding its etiopathogenesis, the most recent being it is an immune reaction to local trauma or inflammation. According to Zegarelli et al., inflammation due to local irritant or trauma causes an increase in epithelial cell turn-over rate and degeneration. Lipid is released as degeneration by-product, which is scavenged by macrophages, resulting in formation of foam cells and hence the OVX lesion. Triggering/promoting factors include: tobacco and alcohol, wet microenvironment, periodontal pathogens, allergy to dental materials, and candida infection.[3] In the present case, tobacco appears to be the triggering factor.

An association with lipid metabolism abnormalities, Diabetes mellitus and viral infection are speculated, but it has not yet been established.[5]

OVX occurs in in the 5th to 6th decade of life with a male to female ratio of 1.1:1.[6] Almost half of the intra-oral lesions occur on gingiva (57.4%), followed by the tongue (10.3%), hard palate (7.1%), buccal or vestibular mucosa (6.7%), floor of the mouth (4.6%) and soft palate (3.2%) and lip.[1],[4] In the present case, the buccal and vestibular mucosa is involved, which is a relatively uncommon site for its occurrence.

The lesions are often asymptomatic, well demarcated, solitary and are usually not greater than 2 cms in diameter. Clinically, the surface of the lesion can be divided into three morphological variants as verrucous or warty, papillary or cauliflower like, and flat. More common in occurrence is verruciform pattern which shows papillary/pebbly/granular/roughened appearance. Occasionally, crateriform or ulcerated surface is seen. Rarely slightly elevated flat topped nodules are seen. Color ranges from white to red with/without a yellowish tinge. Some authors suggest that the presence of reddish to orange tint is characteristic of OVX.[2],[6],[7]

In our case also the lesion appeared to be of focal verruco-papillary category. But, on initial examination the lesion appeared to have white hyper-keratotic appearance, which was reduced after application of topical anti-fungal medication, denoting the possibility of candidal super-infection.

In a series of cases Neville et al. observed candidal infection in 3 of 217 cases and Mostafa et al. observed them in 5 of 108 cases of OVX without apparent microscopic diagnosis.[7] OVX can also occur in association with lichen planus, leukoplakia, oral submucous fibrosis, graft versus host disease and malignancy.[3],[4]

Histologically, OVX shows three patterns: Verrucous, Papillary and Flat. Distinct hyperparakeratotic stratified squamous epithelium, acanthosis and extremely elongated uniform retepegs are evident. Most important diagnostic feature in all the types is accumulation of Foam cells/Xanthoma cells. These are numerous large macrophages with foamy cytoplasm, which typically are confined to the connective tissue papillae between the elongated rete-ridges.[8]

It is important to note that the clinical features of OVX are non-specific and mimics other commonly encountered lesions of oral cavity. These includes but is not limited to benign lesions like squamous papilloma, verruca vulgaris, condyloma acuminatum, potentially malignant lesions like verrucous/non-homogeneous leukoplakia, verrucous hyperplasia and malignant lesions such as verrucous, and squamous cell carcinoma.[4]

In series of 212 cases by Belknap et al., 8% of lesions resembled leukoplakia.[9] In our case also, OVX initially mimicked non-homogeneous leukoplakia. Candidal infection complicated the interpretation and a positive habit history also favored this provisional diagnosis.

The treatment of OVX consists of surgical resection of the lesion. Recurrence and malignant transformation of this lesion are rare. However, in our case, there were signs of recurrence of the lesion. If chronic irritation is probable cause, as in the present case, it is necessary to remove the same to prevent recurrence.[10]


   Conclusion Top


Verruciform xanthoma is a rare muco-cutaneous lesion which is thought to be a multifactorial chronic reactive process. Irrespective of its extra-oral or intra-oral location, the clinical appearance is non-pathognomonic and candidal infection may further complicate the interpretation. It should be considered in the differential diagnosis of other commonly occurring verruco-papillary lesions in the oral cavity. A histopathological examination revealing xanthoma cells is mandatory for the confirmatory diagnosis of VX.

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.
Neville B. Epithelial pathology. In: Neville B, Damm D, Allen C, Bouquot J, editors. Neville Text Book of Oral and Maxillofacial Pathology. 2nd ed. United States of America: Saunders; 2005. p. 324-6.  Back to cited text no. 1
    
2.
Rajendran R. Benign and malignant tumors of the oral cavity. In: Rajendran R, Shivapathasundharam B, editors. Shafer's Textbook of Oral Pathology. 6th ed. India: Elsevier Publications; 2009. p. 139.  Back to cited text no. 2
    
3.
Hegde U, Doddawad V, Sreeshyla H, Patil R. Verruciform xanthoma: A view on the concepts of its etiopathogenesis. J Oral Maxillofac Pathol 2013;3:392-6.  Back to cited text no. 3
    
4.
Gannepalli A, Appala A, Reddy L, Babu DB. Insight into verruciform xanthoma with oral submucous fibrosis: Case report and review of literature. J Oral Maxillofac Pathol 2019;23:43-8.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Theofilou V, Sklavounou A, Argyris P, Chrysomali E. Oral verruciform xanthoma within lichen planus: A case report and literature review. Case Report Dent 2018;2018:1615086. doi: 10.1155/2018/1615086.  Back to cited text no. 5
    
6.
Raphael V, Das H, Sarma R, Shunyu B. Oral verruciform xanthoma: A case report. Int J Oral Maxillofac Pathol 2012;3:65-7.  Back to cited text no. 6
    
7.
Barrett A, Boyapati R, Bisase B, Norris P, Shelley M, Collyer J, et al. Verruciform xanthoma of the oral mucosa: A series of eight typical and three anomalous cases. Int J Surg Pathol 2019;27:492-8.  Back to cited text no. 7
    
8.
Bhalerao S, Bhat P, Chhabra R, Tamgadge A. Verruciform xanthoma of buccal mucosa: A case report with review of literature. Contemp Clin Dent 2012;3:257-9.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Belknap A, Islam M, Bhattacharyya I, Cohen D, Fitzpatrick S. Oral verruciform xanthoma: A series of 212 cases and review of the literature. Head Neck Pathol 2020. doi: 10.1007/s12105-019-01123-0.  Back to cited text no. 9
    
10.
Hiraishi Y, Tojyo I, Kiga N, Tanimoto K, Fujita S. A Case of verruciform xanthoma arising in the tongue. J Clin Diagn Res 2016;10:7-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
 
 
  Search
 
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)

   Abstract Introduction Case Report Discussion Conclusion Article Figures
  In this article
 References

 Article Access Statistics
    Viewed36    
    Printed0    
    Emailed0    
    PDF Downloaded28    
    Comments [Add]    

Recommend this journal


[TAG2]
[TAG3]
[TAG4]