Cutaneous verruciform xanthoma: A report of five cases investigating the etiology and nature of xanthomatous cells

Syed K. Mohsin, Min W. Lee, Mahul B. Amin, Mark H. Stoler, Eduardo Eyzaguirre, Chan K. Ma, Richard J. Zarbo

Research output: Contribution to journalArticle

51 Citations (Scopus)

Abstract

Verruciform xanthoma is a rare clinicopathologic entity of uncertain etiology that occurs primarily in the oral mucosa. Aggregates of foam cells in the submucosal stroma or papillary dermis in association with verrucous epithelial hyperplasia are the hallmark of this lesion. Extraoral (cutaneous) occurrence of verruciform xanthoma is much rarer and has been reported mostly in the genital skin. Five cases of extraoral cutaneous verruciform xanthoma (three from the scrotum, one from the penis, and one from the nose) and one histologic 'simulant' (from skin of the nose) were studied. The lesions were solitary, raised, or polypoid with cup-shaped craters filled with parakeratotic cells that blended into keratinocytes of an acanthotic and papillomatous epidermis. There was a neutrophilic infiltrate of varying intensity between plump parakeratotic cells and keratinocytes, near the surface of the epidermis. Aggregates of foam cells were present in the papillary dermis, which was highly vascular. A plasma cell predominant infiltrate was seen at the base in a bandlike fashion. Despite the architectural resemblance of verruciform xanthoma to verrucous mucocutaneous lesions related to human papillomavirus infection, it was not detected by either immunohistochemistry, in situ hybridization, polymerase chain reaction, or Southern blot analysis in any case. The foam cells were weakly positive for cytokeratin and for Factor XIIIa but negative for S-100 protein. The KP1 and Mac 387 immunostain showed focal weak staining in foam cells. We postulate that a cascade of events pursue after initial keratinocytic damage attracting neutrophils, with subsequent phagocytosis of necrotic keratinocytic debris by dermal dendrocytes, eventually leading to the ultimate manifestation of the lesion as verruciform xanthoma. The etiologic agent remains elusive, but based on our findings, we conclude that verruciform xanthoma is most likely not a human papillomavirus-associated squamoproliferative lesion and that the foam cells, a histologic hallmark of the lesion, are most likely derived from dermal dendritic cells.

Original languageEnglish (US)
Pages (from-to)479-487
Number of pages9
JournalAmerican Journal of Surgical Pathology
Volume22
Issue number4
DOIs
StatePublished - 1998
Externally publishedYes

Fingerprint

Xanthomatosis
Foam Cells
Skin
Dermis
Keratinocytes
Nose
Epidermis
Factor XIIIa
Scrotum
Papillomavirus Infections
Langerhans Cells
S100 Proteins
Penis
Mouth Mucosa
Keratins
Southern Blotting
Plasma Cells
Phagocytosis
Hyperplasia
In Situ Hybridization

Keywords

  • Human papillomavirus
  • Immunohistochemistry
  • In situ hybridization
  • Pathogenesis
  • Penis
  • Polymerase chain reaction
  • Scrotum
  • Skin
  • Verruciform xanthoma
  • Xanthoma

ASJC Scopus subject areas

  • Anatomy
  • Pathology and Forensic Medicine

Cite this

Cutaneous verruciform xanthoma : A report of five cases investigating the etiology and nature of xanthomatous cells. / Mohsin, Syed K.; Lee, Min W.; Amin, Mahul B.; Stoler, Mark H.; Eyzaguirre, Eduardo; Ma, Chan K.; Zarbo, Richard J.

In: American Journal of Surgical Pathology, Vol. 22, No. 4, 1998, p. 479-487.

Research output: Contribution to journalArticle

Mohsin, Syed K. ; Lee, Min W. ; Amin, Mahul B. ; Stoler, Mark H. ; Eyzaguirre, Eduardo ; Ma, Chan K. ; Zarbo, Richard J. / Cutaneous verruciform xanthoma : A report of five cases investigating the etiology and nature of xanthomatous cells. In: American Journal of Surgical Pathology. 1998 ; Vol. 22, No. 4. pp. 479-487.
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abstract = "Verruciform xanthoma is a rare clinicopathologic entity of uncertain etiology that occurs primarily in the oral mucosa. Aggregates of foam cells in the submucosal stroma or papillary dermis in association with verrucous epithelial hyperplasia are the hallmark of this lesion. Extraoral (cutaneous) occurrence of verruciform xanthoma is much rarer and has been reported mostly in the genital skin. Five cases of extraoral cutaneous verruciform xanthoma (three from the scrotum, one from the penis, and one from the nose) and one histologic 'simulant' (from skin of the nose) were studied. The lesions were solitary, raised, or polypoid with cup-shaped craters filled with parakeratotic cells that blended into keratinocytes of an acanthotic and papillomatous epidermis. There was a neutrophilic infiltrate of varying intensity between plump parakeratotic cells and keratinocytes, near the surface of the epidermis. Aggregates of foam cells were present in the papillary dermis, which was highly vascular. A plasma cell predominant infiltrate was seen at the base in a bandlike fashion. Despite the architectural resemblance of verruciform xanthoma to verrucous mucocutaneous lesions related to human papillomavirus infection, it was not detected by either immunohistochemistry, in situ hybridization, polymerase chain reaction, or Southern blot analysis in any case. The foam cells were weakly positive for cytokeratin and for Factor XIIIa but negative for S-100 protein. The KP1 and Mac 387 immunostain showed focal weak staining in foam cells. We postulate that a cascade of events pursue after initial keratinocytic damage attracting neutrophils, with subsequent phagocytosis of necrotic keratinocytic debris by dermal dendrocytes, eventually leading to the ultimate manifestation of the lesion as verruciform xanthoma. The etiologic agent remains elusive, but based on our findings, we conclude that verruciform xanthoma is most likely not a human papillomavirus-associated squamoproliferative lesion and that the foam cells, a histologic hallmark of the lesion, are most likely derived from dermal dendritic cells.",
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