Abstract
Verruciform Xanthoma (VX) is an rare benign mucocutaneous condition that occurs predominantly in the oral cavity which occasionally affects skin and genitals. It appears as a papule or single plaque showing a verrucous or papillomatous structure, with variable color from reddish pink to gray. In majority of oral cases, it affects gingiva and alveolar mucosa being a solitary lesion. It is often associated with pre-existing epithelial and/or inflammatory disorder and is characterized histopathologically by papillary epithelial hyperplasia and the presence of foamy macrophages in connective tissue papillae. We report a rare case of widespread verrucous xanthoma of gingiva and alveolar mucosa which was managed by wide local excision and defect was reconstructed using nasolabial flap.
Highlights
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A benign white lesion of oral cavity to be considered in differential diagnosis.
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Lesions may vary in clinical presentation misguiding the clinicians in diagnosis.
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Verrucous Xanthoma of oral cavity is rare and moreover such widespread involvement is very unlikely.
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Surgical excision being the treatment of choice for verrucous xanthoma.
1
Introduction
Verruciform Xanthoma was first described by Shafer in 1971 [ ]. It occurs mainly in the oral mucosa. Extra oral involvements have been reported on the vulva, scrotum, penis, anal region and extremities. In 1981, Kraemer et al. reported the first verruciform xanthoma case arising on the penis [ ]. Verruciform xanthomas of the oral cavity are usually encountered in the middle aged persons. There is no sex predilection [ ]. The color of intra oral lesions of VX can vary between white, pink, and yellow, depending on the thickness of the overlying epidermis. VX contains foam cells, which are lipid-containing histiocytes found within the submucosal substrates. Various oral lesion like leukoplakia, papilloma, verruca vulgaris, condyloma acuminata, and certain malignant lesions mimic similar features and it is thus necessary to differentiate VX from these lesions with a tissue biopsy. The development of VX is thought to be related to a nonspecific reaction to local epithelial trauma or an immune reaction. However, the etiology and pathogenesis of VX are not yet fully understood. The treatment of the VX lesion involves local surgical excision which is almost always curative without recurrence and the disorder usually has a good prognosis [ , ].
2
Case report
A 55 yrs old male patient reported to the Dept of Oral and Maxillofacial Surgery with a chief complaint of whitish growth over his lower gums for past 02 yrs. There has been no history of trauma or habit of tobacco usage. The growth was insidious in onset without pain which gradually increased to involve gingiva of multiple teeth and the lower left alveolar mucosa. There was no h/o bleeding from the growth. The lesion was approximately 06 cm × 03 cm, sessile, pebbled whitish patch over the left lower anterior gingival and alveolar mucosa extending from 33 to 44 region ( Fig. 1 ) with no surface ulceration and tenderness was evident. There was severe generalized attrition of teeth and gingival recession was present in relation to teeth no 42,43. There was spacing with no signs mobility of teeth in the anterior region. Incisional biopsy was performed and the histopathological examination revealed stratified squamous epithelium, parakeratosis, hyperplasia with uniform crest length, hydropic degeneration and some areas of basal layer duplication and exocytosis. In the lamina propria, chronic inflammatory infiltration was observed and in subepithelial and papillary areas, numerous macrophages presented foam cytoplasm and granules in their interior suggestive of verrucous xanthoma. No relevant alterations in laboratory investigations were found (hemogram, total cholesterol, HDL, LDL, VLDL, Blood glucose level). As the lesion was large and surgical management being curative, he was taken up for wide local excision and reconstruction of the defect using nasolabial flap from left side under GA. Intra-operatively, patient was laid supine and scrubbed and drapped aseptically. The entire lesion was excised using cautery( Figs. 2 and 3 ). Marking for nasolabial flap was done on left side along the skin crease 01 cm lateral to corner of mouth being its pedicle. Nasolabial flap was reflected retaining the pedicle at corner of mouth and was tunneled into the oral cavity to cover the defect. The flap was secured over the defect using resorbable sutures. The donor site was closed primarily after undermining ( Figs. 4–8 ). He was put on antibiotic and analgesic for 05 days. Post operative recovery was uneventful. He was reviewed every 02 weeks for 03 months as there was hair growth over the flap which has to be trimmed at regular interval. Laser ablation was done after 03 months to prevent hair growth. He is under follow up for 02 yrs with no recurrence( Figs. 9 and 10 ).
3
Discussion
VX is a relatively rare benign lesion that was first studied and defined by Shafer in 1971. Although there have been no definite studies on the incidence of this disease, a prevalence of approximately 0.025%–0.094% has been reported with an unknown etiopathology [ ]. Damage to the squamous cells due to various factors like trauma, irritation, or infection, which can cause increased epithelial turnover leads to the disease. The epithelial breakdown leads to an inflammatory response and a subsequent release of lipid material from the degenerated cells [ ]. Verrucous xanthoma occurs in otherwise healthy individuals. Due to its clinical and histopathological resemblance to human papilloma virus-induced lesions, verruciform xanthoma was believed to be caused by HPV. However, most investigators have not found any evidence for the presence of HPV in these lesions [ ]. However a few cases have been reported which were associated with inflammatory conditions such as pemphigus vulgaris, lichen planus, discoid lupus erythematosus, warty dyskeratoma, epidermal nevus/CHILD nevus, dystrophic epidermolysis bullosa, and seborrheic keratosis [ ]. A few cases have also been reported to be associated with disorders of lipid metabolism [ , ]. Changes in the lipid metabolism and viruses do not seem to induce verruciform xanthoma [ ]. On serological examination, our case had a normal serum lipid profile. Oliveira et al. [ ] suggest the development of an immune response, similar to that associated with lichen planus. Meanwhile, Ide et al. [ ] speculated that verruciform xanthoma has a multifactorial etiology, including periodontal pathogens, mechanical stimulation, tobacco, alcohol, and dietary allergens or sensitizing agents, among others.
In the 15 cases reported by Yu et al. [ ], the mean length of the verruciform xanthomas was 0.8cm. Only two lesions had a length greater than 1 cm, and the biggest lesion measured 2.0 × 1.0cm. The four lesions reported by Oliveira et al. [ ] varied from 0.4 to 2.0cm where as the lesion in our case is extremely large measuring 06 × 03 cm. Skin verruciform xanthomas can be much larger: a case affecting more than 15cm of skin has already been reported [ ]. The lesion is benign, asymptomatic, and slow growing and rarely exceeds 2 cm in size which is in contrast to our case. It is sessile or pedunculated and can resemble leukoplakia or squamous papilloma. It occurs most commonly in 4th–6th decade of life with equal distribution between both sexes. However, it has been reported that there is a slight male predilection as it correlates with our case [ , , ]. Literature review on verrucous xanthoma of oral cavity has been summarized in Table 1 .
| S No | Study/report | No of Lesion Reported | Sex | Site | Size in cms | Age in yrs | Other associated lesions if any | Habits | Treatment Provided | Prognosis |
|---|---|---|---|---|---|---|---|---|---|---|
| 1 | Present report | 01 | M | Left lower alveolar mucosa extending to gingiva | 06x 03 (Largest) | 55 | No other lesion | Nil | Excision and Reconstruction of defect with Nasolabial flap | No recurrence |
| 2 | Austin N. Belknap et al., 2020 [ ] | 212 | 1.06:1 F:M ratio | Gingiva, Palate Tongue, Buccal mucosa |
Not mentioned | Mean age 61 | Not mentioned | Not mentioned | Excision | 03 recurrence |
| 3 | Ganapalli A et al., 2019 [ ] | 01 | M | Attached gingiva | 0.8 × 2 | 52 | Oral submuous fibrosis | Smoking and Tobacco chewing | Excision under LA | No recurrence |
| 4 | A. W. Barrett et al., 2019 [ ] | 11 | 06 M and 05 F | Ladial mucosa, tongue | 2.0 cm max | 40–69 | No other lesion | Nil | Excision under LA | One required re- excision |
| 5 | P Tamiolakis et al., 2018 [ ] | 13 | 08 m and 05 F | Mostly in gingival and hard palate, tongue | 0.3–2.5 cm | Mean age 48 | No other lesion | Nil | Surgical Excision | No recurrence |
| 6 | Rodrigues JT et al., 2017 [ ] | 01 | F | Marginal and attached gingival | 1.2 × 0.6 cm | 66 | No other lesion | Nil | Excision under LA | No recurrence |
| 7 | Cebeci F et al., 2017 [ ] | 01 | M | Vermilion border lower lip | 0.5 × 0.7 | 61 | No other lesion | Nil | Excision under LA | No recurrence |
| 8 | Byakodi S et al., 2017 [ ] | 01 | F | Tip of tongue | 2.5 cm | 50 | No other lesion | Nil | Excision under GA | No recurrence |
| 9 | Treville Pereira et al., 2016 [ ] | 01 | M | Lower labial mucosa | 0.5 × 1.0cm | 59 | No other lesion | Smoker | Excision under LA | No recurrence |
| 10 | Alexandre Simões Garcia et al., 2016 [ ] | 01 | M | Hard plate | 0.5 | 43 | No other lesion | Nil | Excision under LA | No recurrence |
| 11 | Akshay Shetty et al., 2013 [ ] | 01 | M | Left Buccal mucosa | 1.1 | 60 | Nil | Smoking | Excision under LA | No recurrence |
| 12 | Shyam Prasad Reddy Dorankula et al., 2013 [ ] | 01 | M | Left buccal mucosa | 2 × 2 | 42 | No other lesion | Smoking | Excision under LA | No recurrence |
| 13 | Joshi R et al., 2012 [ ] | 05 | All Male | Tongue and buccal mucosa | 2.5 cmmax | 40–50 | 03 lesion in genital region | Nil | Excision under LA | No recurrence |
| 14 | Ozgur mete et al., 2009 [ ] | 01 | F | Ventral Surface of tongue | 1 | 31 | No other lesion | Nil | Excision under LA | No recurrence |
| 15 | Sah K, Kale AD et al., 2008 [ ] | 02 | Both male | Right Buccal mucosa Lowe labial mucosa (two lesion) |
1 × 2 0.5 × 0.5 and 1 × 1 |
50 40 |
No other lesion No other lesion |
Tobacco chewing Nil |
Excision under LA Excision under LA |
No recurrence No recurrence |
| 16 | Chuan-Hang Yu et al., 2007 [ ] | 15 | 07 M and 06 F | Gingiva, tongue, buccal mucosa | 0.3–2.0 cm | 18–79 | No other lesion | Nil | Surgical Excision | No recurrence |
| 17 | W. J. Hume et al., 1980 [ ] | 03 | 02 F and 01 M | Palate, tongue and left cheek | 1.5 × 1.5 max | 45–60 | No other lesion | Nil | Excision under LA | No recurrence |
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