Oral Verruciform Xanthoma: A Case Report and Literature Review
Identifieur interne : 003C64 ( Pmc/Corpus ); précédent : 003C63; suivant : 003C65Oral Verruciform Xanthoma: A Case Report and Literature Review
Auteurs : Akshay Shetty ; Kourosh Nakhaei ; Yogesh Lakkashetty ; Maryam Mohseni ; Iman MohebatzadehSource :
- Case Reports in Dentistry [ 2090-6447 ] ; 2013.
Abstract
Verruciform xanthoma is a benign mucocutaneous, uncommon, nonsymptomatic lesion of uncertain etiopathology, which occurs mostly on the oral mucosa of middle-aged individuals. Histopathologically, VX is diagnosed by presence of lipid-laden foam cells in papillary region of connective tissue. A 60-year-old male patient presented with a painless growth on the left buccal mucosa. On clinical examination a yellowish white exophytic lesion, measuring 11 × 7 mm in size, was found, which was cauliflower-shaped on inspection and painless on palpation. Histopathological examination revealed varying degrees of surface parakeratosis and the accumulation of numerous foam cells in the connective tissue papillae among the uniformly elongated epithelial ridges. On immunohistochemical staining, there was a neutrophilic infiltrate of the epidermis with CD68 positive xanthoma cells restricted to the papillary dermis, mixed with other chronic inflammatory cells.
Url:
DOI: 10.1155/2013/528967
PubMed: 24396610
PubMed Central: 3876769
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Oral Verruciform Xanthoma: A Case Report and Literature Review</title><author><name sortKey="Shetty, Akshay" sort="Shetty, Akshay" uniqKey="Shetty A" first="Akshay" last="Shetty">Akshay Shetty</name><affiliation><nlm:aff id="I1">Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Nakhaei, Kourosh" sort="Nakhaei, Kourosh" uniqKey="Nakhaei K" first="Kourosh" last="Nakhaei">Kourosh Nakhaei</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Lakkashetty, Yogesh" sort="Lakkashetty, Yogesh" uniqKey="Lakkashetty Y" first="Yogesh" last="Lakkashetty">Yogesh Lakkashetty</name><affiliation><nlm:aff id="I3">Department of Oral and Maxillofacial Pathology, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Mohseni, Maryam" sort="Mohseni, Maryam" uniqKey="Mohseni M" first="Maryam" last="Mohseni">Maryam Mohseni</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Mohebatzadeh, Iman" sort="Mohebatzadeh, Iman" uniqKey="Mohebatzadeh I" first="Iman" last="Mohebatzadeh">Iman Mohebatzadeh</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">24396610</idno><idno type="pmc">3876769</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3876769</idno><idno type="RBID">PMC:3876769</idno><idno type="doi">10.1155/2013/528967</idno><date when="2013">2013</date><idno type="wicri:Area/Pmc/Corpus">003C64</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">003C64</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Oral Verruciform Xanthoma: A Case Report and Literature Review</title><author><name sortKey="Shetty, Akshay" sort="Shetty, Akshay" uniqKey="Shetty A" first="Akshay" last="Shetty">Akshay Shetty</name><affiliation><nlm:aff id="I1">Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Nakhaei, Kourosh" sort="Nakhaei, Kourosh" uniqKey="Nakhaei K" first="Kourosh" last="Nakhaei">Kourosh Nakhaei</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Lakkashetty, Yogesh" sort="Lakkashetty, Yogesh" uniqKey="Lakkashetty Y" first="Yogesh" last="Lakkashetty">Yogesh Lakkashetty</name><affiliation><nlm:aff id="I3">Department of Oral and Maxillofacial Pathology, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Mohseni, Maryam" sort="Mohseni, Maryam" uniqKey="Mohseni M" first="Maryam" last="Mohseni">Maryam Mohseni</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author><author><name sortKey="Mohebatzadeh, Iman" sort="Mohebatzadeh, Iman" uniqKey="Mohebatzadeh I" first="Iman" last="Mohebatzadeh">Iman Mohebatzadeh</name><affiliation><nlm:aff id="I2">Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</nlm:aff></affiliation></author></analytic><series><title level="j">Case Reports in Dentistry</title><idno type="ISSN">2090-6447</idno><idno type="eISSN">2090-6455</idno><imprint><date when="2013">2013</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Verruciform xanthoma is a benign mucocutaneous, uncommon, nonsymptomatic lesion of uncertain etiopathology, which occurs mostly on the oral mucosa of middle-aged individuals. Histopathologically, VX is diagnosed by presence of lipid-laden foam cells in papillary region of connective tissue. A 60-year-old male patient presented with a painless growth on the left buccal mucosa. On clinical examination a yellowish white exophytic lesion, measuring 11 × 7 mm in size, was found, which was cauliflower-shaped on inspection and painless on palpation. Histopathological examination revealed varying degrees of surface parakeratosis and the accumulation of numerous foam cells in the connective tissue papillae among the uniformly elongated epithelial ridges. On immunohistochemical staining, there was a neutrophilic infiltrate of the epidermis with CD68 positive xanthoma cells restricted to the papillary dermis, mixed with other chronic inflammatory cells.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Mohsin, Sk" uniqKey="Mohsin S">SK Mohsin</name></author><author><name sortKey="Lee, Mw" uniqKey="Lee M">MW Lee</name></author><author><name sortKey="Amin, Mb" uniqKey="Amin M">MB Amin</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Rajendran, R" uniqKey="Rajendran R">R Rajendran</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Buchner, A" uniqKey="Buchner A">A Buchner</name></author><author><name sortKey="Hansen, Ls" uniqKey="Hansen L">LS Hansen</name></author><author><name sortKey="Merrell, Pw" uniqKey="Merrell P">PW Merrell</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Shafer, 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<front><journal-meta><journal-id journal-id-type="nlm-ta">Case Rep Dent</journal-id><journal-id journal-id-type="iso-abbrev">Case Rep Dent</journal-id><journal-id journal-id-type="publisher-id">CRIM.DENTISTRY</journal-id><journal-title-group><journal-title>Case Reports in Dentistry</journal-title></journal-title-group><issn pub-type="ppub">2090-6447</issn><issn pub-type="epub">2090-6455</issn><publisher><publisher-name>Hindawi Publishing Corporation</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">24396610</article-id><article-id pub-id-type="pmc">3876769</article-id><article-id pub-id-type="doi">10.1155/2013/528967</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Oral Verruciform Xanthoma: A Case Report and Literature Review</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Shetty</surname><given-names>Akshay</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><contrib-id contrib-id-type="orcid" authenticated="false">http://orcid.org/0000-0002-7340-882X</contrib-id><name><surname>Nakhaei</surname><given-names>Kourosh</given-names></name><xref ref-type="aff" rid="I2"><sup>2</sup></xref><xref ref-type="corresp" rid="cor1">*</xref></contrib><contrib contrib-type="author"><name><surname>Lakkashetty</surname><given-names>Yogesh</given-names></name><xref ref-type="aff" rid="I3"><sup>3</sup></xref></contrib><contrib contrib-type="author"><name><surname>Mohseni</surname><given-names>Maryam</given-names></name><xref ref-type="aff" rid="I2"><sup>2</sup></xref></contrib><contrib contrib-type="author"><name><surname>Mohebatzadeh</surname><given-names>Iman</given-names></name><xref ref-type="aff" rid="I2"><sup>2</sup></xref></contrib></contrib-group><aff id="I1"><sup>1</sup>Department of Oral and Maxillofacial Surgery, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</aff><aff id="I2"><sup>2</sup>Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</aff><aff id="I3"><sup>3</sup>Department of Oral and Maxillofacial Pathology, Sri Rajiv Gandhi College of Dental Sciences and Hospital, Cholanagar, RT Nagar, Bangalore 560032, India</aff><author-notes><corresp id="cor1">*Kourosh Nakhaei: <email>koorosh_intheflesh@yahoo.com</email></corresp><fn fn-type="other"><p>Academic Editors: A. Ferri and A. Markopoulos</p></fn></author-notes><pub-date pub-type="ppub"><year>2013</year></pub-date><pub-date pub-type="epub"><day>16</day><month>12</month><year>2013</year></pub-date><volume>2013</volume><elocation-id>528967</elocation-id><history><date date-type="received"><day>23</day><month>10</month><year>2013</year></date><date date-type="accepted"><day>19</day><month>11</month><year>2013</year></date></history><permissions><copyright-statement>Copyright © 2013 Akshay Shetty et al.</copyright-statement><copyright-year>2013</copyright-year><license xlink:href="https://creativecommons.org/licenses/by/3.0/"><license-p>This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Verruciform xanthoma is a benign mucocutaneous, uncommon, nonsymptomatic lesion of uncertain etiopathology, which occurs mostly on the oral mucosa of middle-aged individuals. Histopathologically, VX is diagnosed by presence of lipid-laden foam cells in papillary region of connective tissue. A 60-year-old male patient presented with a painless growth on the left buccal mucosa. On clinical examination a yellowish white exophytic lesion, measuring 11 × 7 mm in size, was found, which was cauliflower-shaped on inspection and painless on palpation. Histopathological examination revealed varying degrees of surface parakeratosis and the accumulation of numerous foam cells in the connective tissue papillae among the uniformly elongated epithelial ridges. On immunohistochemical staining, there was a neutrophilic infiltrate of the epidermis with CD68 positive xanthoma cells restricted to the papillary dermis, mixed with other chronic inflammatory cells.</p></abstract></article-meta></front><body><sec id="sec1"><title>1. Introduction</title><p>Verruciform xanthoma (VX) is an uncommon benign mucocutaneous lesion that resembles virus-induced papilloma but has an unknown etiology and uncertain nature. It was first reported by Shafer in 1971. VX usually occurs in the oral mucosa of the middle-aged individuals. It most commonly presents with a verrucous appearance. However in some instances it may appear polypoid, papillomatous, or sessile. It occurs as a small (0.2–2 cm), solitary, asymptomatic, slow growing, white or yellowish red lesion with no sex predilection [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B2" ref-type="bibr">2</xref>].</p><p>Histologically VX is distinguished from other lesions by the presence of large numbers of foam cells in, and essentially limited to, the connective tissue papillae. The foam cells on ultrastructural studies have been concluded to be fat-laden macrophages [<xref rid="B3" ref-type="bibr">3</xref>, <xref rid="B4" ref-type="bibr">4</xref>]. Other cell types, including Langerhans cells, intraepithelial neutrophilic infiltrate, and even fibroblasts, have been reported [<xref rid="B1" ref-type="bibr">1</xref>–<xref rid="B3" ref-type="bibr">3</xref>]. A variable degree of parakeratosis is observed which is present in the crypts between papillae which are of variable length and thickness, often extending close to the surface. The rete pegs are extremely elongated and uniform [<xref rid="B2" ref-type="bibr">2</xref>]. Almost every VX case is diagnosed on histological examination as the clinical appearance is not diagnostic. Differentiation from other lesions with foamy or granular cells is not difficult as the VX is the only lesion to have these cells confined to the papillae [<xref rid="B4" ref-type="bibr">4</xref>, <xref rid="B5" ref-type="bibr">5</xref>].</p><p>Differential diagnosis includes erythroplasia of Queyrat (Bowen disease of the glans penis), seborrheic keratosis, verrucous carcinoma, verruca simplex, and condyloma acuminatum [<xref rid="B6" ref-type="bibr">6</xref>, <xref rid="B7" ref-type="bibr">7</xref>]. The treatment of the VX lesion involves local surgical excision which is almost always curative, and recurrence is rare [<xref rid="B2" ref-type="bibr">2</xref>].</p></sec><sec id="sec2"><title>2. Case Presentation</title><p>A-60-year old male patient presented with the chief complaint of a painless growth on the left buccal mucosa (<xref ref-type="fig" rid="fig1">Figure 1</xref>). He had a habit of smoking for the past 10 years. On clinical examination, a yellowish white exophytic lesion, measuring 11 × 7 mm in size, was found, which had a cauliflower shape on inspection. The lesion was asymptomatic and soft in consistency. Lymph nodes were not palpable. There were no systemic diseases and he was otherwise healthy. A provisional diagnosis of papilloma was made. After clinical examination an excisional biopsy was taken to rule out malignancy.</p><p>On histopathological examination, the hematoxylin and eosin (H&E) stained sections showed parakeratotic epithelium with columns of parakeratin plugging into it. Owing to the uniformly elongated rete pegs, deep connective tissue papillae were seen, some of which extended into overlying surface epithelium (Figures <xref ref-type="fig" rid="fig2">2</xref> and <xref ref-type="fig" rid="fig3">3</xref>). Few neutrophils were also seen in the upper spinous layer. The papillary zone of lamina propria showed numerous lipid-laden foam cells. There was no evidence of dysplasia or malignancy. The fact that the foam cells were confined to the papillary region of connective tissue confirmed the diagnosis as VX.</p><p>The immunohistochemical staining for CD68 was positive for the foamy macrophages. All the foam cells were strongly stained with anti-macrophage antibodies (Figures <xref ref-type="fig" rid="fig4">4</xref> and <xref ref-type="fig" rid="fig5">5</xref>).</p><p>The treatment was performed under local anesthesia. The whole lesion was excised in one piece during excisional biopsy (<xref ref-type="fig" rid="fig6">Figure 6</xref>). Postoperative check-up showed no sign of recurrence.</p></sec><sec id="sec3"><title>3. Discussion</title><p>Verruciform xanthoma is a rare lesion, accounting for 0.025–0.095% of all cases [<xref rid="B3" ref-type="bibr">3</xref>] with an unknown etiopathology. It can be because of the damage to the squamous cells due to trauma, irritation, or infection, which can cause increased epithelial turnover leading to the disease. The epithelial breakdown leads to an inflammatory response and a subsequent release of lipid material from the degenerated cells [<xref rid="B2" ref-type="bibr">2</xref>, <xref rid="B8a" ref-type="bibr">8</xref>–<xref rid="B10" ref-type="bibr">11</xref>]. Most of the cases occur 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 [<xref rid="B3" ref-type="bibr">3</xref>]. 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 [<xref rid="B11" ref-type="bibr">12</xref>]. A few cases have also been reported to be associated with disorders of lipid metabolism [<xref rid="B12" ref-type="bibr">13</xref>]. On serological examination, our case had a normal serum lipid profile. The lesion is benign, nonsymptomatic, and slow growing and rarely exceeds 2 cm in size. 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 [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B2" ref-type="bibr">2</xref>, <xref rid="B13" ref-type="bibr">14</xref>]. The lesion is usually present in the intraoral regions especially on alveolar ridge, gingiva, followed by buccal mucosa, palate, floor of the mouth, and lip. Extra oral sites include vulva, scrotum, penis, and skin of thigh and perineum, which are usually associated with other conditions like lymphedema, epidermal nevi, congenital hemidysplasia, and limb defect syndrome [<xref rid="B6" ref-type="bibr">6</xref>, <xref rid="B7" ref-type="bibr">7</xref>, <xref rid="B14" ref-type="bibr">15</xref>].</p><p>Histologically, VX shows three patterns: verrucous (most common), flat, and papillary (least common) [<xref rid="B15" ref-type="bibr">16</xref>, <xref rid="B16" ref-type="bibr">17</xref>]. There is parakeratosis of the hyperplastic epithelium which is variable in extent and is usually more marked in the verrucous and papillary patterns. The rete pegs are elongated, uniform, and thin, with deep central keratinized clefts and keratin plugging. However, there is no evidence of dysplasia. The characteristic histological feature is the presence of xanthoma cells in the connective tissue [<xref rid="B2" ref-type="bibr">2</xref>, <xref rid="B17" ref-type="bibr">18</xref>]. The lipid-laden foam cells are present in the superficial connective tissue. There is a controversy over the exact origin of these cells. They are said to be a lineage of monocytes/macrophages [<xref rid="B18" ref-type="bibr">19</xref>, <xref rid="B19" ref-type="bibr">20</xref>]. The lipid found in the xanthoma cells is said to be the same as seen in other inflammatory reactions [<xref rid="B11" ref-type="bibr">12</xref>]. In our case, the foam cells showed strong CD68 immunoreactivity. CD68 is a cell marker confirming the possible role of macrophages in the formation of foam cells [<xref rid="B11" ref-type="bibr">12</xref>].</p><p>The treatment of choice is complete surgical excision which is very effective with no recurrence. 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person-group-type="author"><name><surname>Connolly</surname><given-names>SB</given-names></name><name><surname>Lewis</surname><given-names>EJ</given-names></name><name><surname>Lindholm</surname><given-names>JS</given-names></name><name><surname>Zelickson</surname><given-names>BD</given-names></name><name><surname>Zachary</surname><given-names>CB</given-names></name><name><surname>Tope</surname><given-names>WD</given-names></name></person-group><article-title>Management of cutaneous verruciform xanthoma</article-title><source><italic>Journal of the American Academy of Dermatology</italic></source><year>2000</year><volume>42</volume><issue>2</issue><fpage>343</fpage><lpage>347</lpage><pub-id pub-id-type="other">2-s2.0-0033911654</pub-id><pub-id pub-id-type="pmid">10640929</pub-id></element-citation></ref></ref-list></back><floats-group><fig id="fig1" orientation="portrait" position="float"><label>Figure 1</label><caption><p>Clinical photograph showing the exophytic lesion on the left buccal mucosa.</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.001"></graphic></fig><fig id="fig2" orientation="portrait" position="float"><label>Figure 2</label><caption><p>Showing epithelial hyperplasia with parakeratosis and elongated rete pegs. There is an abundance of foam cells in the connective tissue papilla (H&E stain 10x).</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.002"></graphic></fig><fig id="fig3" orientation="portrait" position="float"><label>Figure 3</label><caption><p>Presence of large numbers of lipid-laden foamy histiocytes confined to the connective tissue papillae (H&E stain 40x).</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.003"></graphic></fig><fig id="fig4" orientation="portrait" position="float"><label>Figure 4</label><caption><p>Showing foam cells with strong cytoplasmic CD68 immunostaining. The epithelial cells were negative (IHC stain 10x).</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.004"></graphic></fig><fig id="fig5" orientation="portrait" position="float"><label>Figure 5</label><caption><p>The infiltrated foam cells in the papillary dermis were CD68 positive (IHC stain 40x).</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.005"></graphic></fig><fig id="fig6" orientation="portrait" position="float"><label>Figure 6</label><caption><p>Showing the lesion after excision.</p></caption><graphic xlink:href="CRIM.DENTISTRY2013-528967.006"></graphic></fig></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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