A rare case of recessive dystrophic epidermolysis bullosa and verruciform xanthoma

Orpin, S D; Scott, Ian C.; Rajaratnam, Ratna; Colloby, P.; Heagerty, Adrian

doi:10.1111/j.1365-2230.2008.02888.x

Cited by 20 publications

(29 citation statements)

References 9 publications

(9 reference statements)

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“…It is likely that damage to the basal cell and apoptosis seen in lichen planus and oral chronic GVHD release lipids that are phagocytosed by histiocytes or dermal dendrocytes. Other associated conditions that suggest a role for epithelial damage include pemphigus vulgaris and epidermolysis bullosa [9,21].…”

Section: Discussionmentioning

confidence: 99%

“…Although most of the cases of VX occur in otherwise healthy individuals, it has been reported in association with inflammatory conditions such as pemphigus vulgaris [9], lichen planus [10][11][12][13][14], discoid lupus erythematosus [15,16], warty dyskeratoma [17], psoriasis [18], epidermal nevus/CHILD nevus [5,19,20], dystrophic epidermolysis bullosa [21], and seborrheic keratosis [22]. It has also been associated with carcinoma in situ [23], and squamous cell carcinoma [24].…”

Section: Introductionmentioning

confidence: 99%

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Oral Verruciform Xanthoma Associated with Chronic Graft-Versus- Host Disease: A Report of Five Cases and a Review of the Literature

Farahani

Treister

Khan

et al. 2011

Head and Neck Pathol

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Verruciform xanthoma (VX) is an uncommon benign inflammatory mucocutaneous condition that chiefly occurs in the oral cavity. 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 of a series of five cases with VX who concurrently had chronic oral graft-versus-host disease following hematopoietic stem cell transplantation.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Oral Verruciform Xanthoma Associated with Chronic Graft-Versus- Host Disease: A Report of Five Cases and a Review of the Literature

Farahani

Treister

Khan

et al. 2011

Head and Neck Pathol

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“…8 Occasionally, cutaneous VX has been described in association with lymphedema, repeated trauma, pemphigus vulgaris, chronic graft versus host syndrome, congenital hemidysplasia with icthyosifom erythroderma and limb defects syndrome, epidermal nevi, dystrophic recessive epidermolysis bullosa, [9][10][11][12] discoid lupus erythematosus, sundamaged facial skin, and solar keratosis. VX is characterized by the presence of acanthosis, hyperkeratosis with columnar parakeratosis, and papillomatosis without architectural atypia and by the presence of a dense accumulation of xanthomatous foamy cells in the papillary dermis that are positive for the macrophage marker CD68.…”

Section: Discussionmentioning

confidence: 99%

“…However, serial studies have failed to find conclusive evidence of HPV infection within VX cells. [9][10][11][12] The observed lesions corresponded to erythematous, verrucous plaques of variable size (from 8 to 12 cm) developing over the sacrum, leg, shoulder, and inguinal region, respectively. The degenerated epithelium might lead to the formation of lipid droplets that would be trapped by dermal cells.…”

Section: Discussionmentioning

confidence: 99%

Verruciform Xanthoma Developing in Recessive Dystrophic Epidermolysis Bullosa

Curto‐Barredo

Segura

Barranco³

et al. 2014

The American Journal of Dermatopathology

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A 23-year-old male affected of recessive dystrophic epidermolysis bullosa presented with a 2-month history of a growing verrucous plaque on the right flank. The clinical features and evolution suggested the diagnosis of cutaneous squamous cell carcinoma. Histopathological examination showed hyperkeratosis, parakeratossis, and verrucous acanthosis and numerous large xanthoma cells in the papillary dermis. Reflectance confocal microscopy disclosed the absence of epidermal atypia and the presence of aggregates of reflecting cells at dermal-epidermal junction and upper dermis. These cells were ultrastructurally characterized and corresponded to foamy histiocytes. Verruciform xanthoma is a benign reactive lesion that has occasionally been reported to develop in chronically eroded areas in patients with recessive dystrophic epidermolysis bullosa. In this group of patients, verruciform xanthoma may clinically mimic cutaneous squamous cell carcinoma and a correct diagnosis is crucial to avoid inappropriate aggressive therapeutic approaches. In vivo noninvasive image technologies such as reflectance confocal microscopy may be helpful diagnostic tools in this clinical setting.

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“…[2021] The changes may histologically resemble true VX and are considered to be a form of dystrophic xanthomatosis that possibly arises due to repeated epidermal or dermal damage, with accumulation of lipids within macrophages.…”

Section: Discussionmentioning

confidence: 99%

Verruciform xanthoma: Report of five cases

Joshi

Ovhal

2012

Indian J Dermatol

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We describe five cases of verruciform xanthoma (VX). The patients, all males, presented with single warty verrucous lesions of 0.5–2 cm size that had been diagnosed clinically as viral warts (four cases) and leukoplakia (one case). Two patients had the lesion in the oral cavity, two on the genital mucosa, and one on the scrotal skin. Histopathology was diagnostic, with verrucous and papillomatous epidermal hyperplasia with the silhouette of a viral wart but with numerous foamy histiocytes packed in the elongated dermal papillae. Columns of deep parakeratosis and neutrophils in the upper spinous layers, with a dermal plasma cell infiltrate were the other histopathologic findings. Excision of the lesions was curative, without recurrences, in the two patients who had lesions in the oral cavity; follow-up was not available in the cases with genital lesions. VX is an uncommon but distinctive clinicopathologic entity affecting the oral and genital mucosa that may be mistaken for benign, premalignant, and malignant conditions. VX can be diagnosed with certainty only on histopathologic examination.

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A rare case of recessive dystrophic epidermolysis bullosa and verruciform xanthoma

Cited by 20 publications

References 9 publications

Oral Verruciform Xanthoma Associated with Chronic Graft-Versus- Host Disease: A Report of Five Cases and a Review of the Literature

Oral Verruciform Xanthoma Associated with Chronic Graft-Versus- Host Disease: A Report of Five Cases and a Review of the Literature

Verruciform Xanthoma Developing in Recessive Dystrophic Epidermolysis Bullosa

Verruciform xanthoma: Report of five cases

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