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Research Letter |

Lichen Sclerosus Exhibiting Histologic Signs of Lymphedema: An Essential Factor in the Pathogenesis of Verruciform Xanthoma —Reply FREE

Charlotte Fite, MD; Fran çoise Plantier, MD; Micheline Moyal-Barracco, MD
[+] Author Affiliations

Author Affiliations: Departments of Dermatology (Drs Fite and Moyal-Barracco) and Pathology (Dr Plantier), Assistance Publique des H ôpitaux de Paris (APHP), Hospital Cochin, Paris Descartes University, Paris, France.


Arch Dermatol. 2012;148(2):260-262. doi:10.1001/archdermatol.2011.2085.
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In reply

We appreciate the interest of Carlson et al in our article on vulvar VX1 as well as their comment about the possible etiologic roles of lymphostasis and HPV. Our 10 vulvar VX cases were all associated with another vulvar condition, mainly LS but also lichen planus (2 cases), vulvar radiodermatitis (1 case), and Paget disease (1 case). Our findings sustain the hypothesis of Zegarelli et al2 that damage to the epithelium —particularly of the DEJ, in our opinion, could trigger the following cascade: (1) entrapment of epithelial cells in the papillary dermis; (2) subsequent degeneration of these cells and lipid formation; (3) engulfment of released lipids by macrophages; and (4) accumulation of foam cells between the rete ridges.

Carlson et al object that this hypothesis does not explain why macrophages accumulate in the papillary dermis. We think that the superficial location of the xanthomatous cells can be explained by the fact that the papillary dermis is the part of the dermis, which is the closest of the damaged epidermis. The poor lymphatic drainage reported by Carlson et al in 14 genital and 4 trunk LS cases could account for the accumulation of macrophages in the papillary dermis. However, to confirm this hypothesis one should demonstrate the following: (1) that all the other conditions associated with mucosal or cutaneous VX are associated with lymphostasis (eg, Paget disease, lichen planus, graft-vs-host disease, discoid lupus erythematosus, pemphigus vulgaris, recessive dystrophic epidermolysis bullosa, lichen planus, epidermal nevus); (2) that lymphostasis is not just an incidental finding related to inflammation, whatever its cause. In addition, if an increased number and dilation of lymphatic vessels is present in most LS cases, these abnormalities cannot alone explain alone the occurrence of VX with LS. Indeed, VX only exceptionally occurs concomitantly with LS.

The second hypothesis advanced by Carlson et al is that the verrucous epidermal hyperplasia that is a hallmark of VX could be related to an HPV infection. This HPV infection may have been facilitated by the lymphostasis, the source of the disrupted immune-cell trafficking and consequently of localized immunosuppression. This interesting assumption is not corroborated either by the pathologic features of VX or by the available virologic data. Indeed, we found that the verrucous hyperplasia of VX had specific, almost pathognomonic, histologic features that differ from those of HPV infections: wedge-shaped parakeratosis forming deep invaginations into the acanthotic epithelium and exhibiting a characteristic orange hue under hematoxylin-eosin stain; and neutrophilic infiltrate at the junction between the superficial parakeratotic layers and the underlying stratum spinulosum. In addition, neither koilocytes nor atypia were observed.

In our retrospective study, no HPV search was performed. However, the data collected from the literature are mainly negative, even though very sensitive methods were used.3 A few cases with a positive HPV search have been reported,4 but these findings could have been incidental: HPV may be present on normal vulvar or oral mucosa in as many as 23.3% of the cases.5

ARTICLE INFORMATION

Correspondence: Dr Fite, APHP, Department of Dermatology, 27 rue du Faubourg Saint-Jacques, Paris, 75014 France (Charlotte.fite@noos.fr).

Fite C, Plantier F, Dupin N, Avril MF, Moyal-Barracco M. Vulvar verruciform xanthoma: ten cases associated with lichen sclerosus, lichen planus, or other conditions.  Arch Dermatol. 2011;147(9):1087-1092
PubMed   |  Link to Article
Zegarelli DJ, Zegarelli-Schmidt EC, Zegarelli EV. Verruciform xanthoma: further light and electron microscopic studies, with the addition of a third case.  Oral Surg Oral Med Oral Pathol. 1975;40(2):246-256
PubMed   |  Link to Article
Er şahin C, Szpaderska AM, Foreman K, Yong S. Verruciform xanthoma of the penis not associated with human papillomavirus infection.  Arch Pathol Lab Med. 2005;129(3):e62-e64
PubMed
Khaskhely NM, Uezato H, Kamiyama T,  et al.  Association of human papillomavirus type 6 with a verruciform xanthoma.  Am J Dermatopathol. 2000;22(5):447-452
PubMed   |  Link to Article
Llamas-Mart ínez S, Esparza-G ómez G, Campo-Trapero J,  et al.  Genotypic determination by PCR-RFLP of human papillomavirus in normal oral mucosa, oral leukoplakia and oral squamous cell carcinoma samples in Madrid (Spain).  Anticancer Res. 2008;28(6A):3733-3741
PubMed

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References

Fite C, Plantier F, Dupin N, Avril MF, Moyal-Barracco M. Vulvar verruciform xanthoma: ten cases associated with lichen sclerosus, lichen planus, or other conditions.  Arch Dermatol. 2011;147(9):1087-1092
PubMed   |  Link to Article
Zegarelli DJ, Zegarelli-Schmidt EC, Zegarelli EV. Verruciform xanthoma: further light and electron microscopic studies, with the addition of a third case.  Oral Surg Oral Med Oral Pathol. 1975;40(2):246-256
PubMed   |  Link to Article
Er şahin C, Szpaderska AM, Foreman K, Yong S. Verruciform xanthoma of the penis not associated with human papillomavirus infection.  Arch Pathol Lab Med. 2005;129(3):e62-e64
PubMed
Khaskhely NM, Uezato H, Kamiyama T,  et al.  Association of human papillomavirus type 6 with a verruciform xanthoma.  Am J Dermatopathol. 2000;22(5):447-452
PubMed   |  Link to Article
Llamas-Mart ínez S, Esparza-G ómez G, Campo-Trapero J,  et al.  Genotypic determination by PCR-RFLP of human papillomavirus in normal oral mucosa, oral leukoplakia and oral squamous cell carcinoma samples in Madrid (Spain).  Anticancer Res. 2008;28(6A):3733-3741
PubMed

Correspondence

February 1, 2012
J. Andrew Carlson, MD, FRCPC; Grant D. Dias Carlson; Michael Murphy, MD; Angela Rohwedder, PhD
Arch Dermatol. 2012;148(2):260-262. doi:10.1001/archdermatol.2011.1536.
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