Verruciform xanthoma in recessive dystrophic epidermolysis bullosa Hallopeau‐Siemens

Murat-Sušić, Slobodna; Paštar, Zrinjka; Dobrić, Ivan; Varela, A Quiroga; Hutinec, Zdenka; Husar, Karmela; Kljenak, Antun

doi:10.1111/j.1365-4632.2007.03071.x

Cited by 15 publications

(21 citation statements)

References 51 publications

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“…In most cases, it involves the oral mucosa, but extraoral presentations have been reported, most often anogenitally . Histologically, VX demonstrates papillomatosis, hyperkeratosis with columnar parakeratosis, and uniform elongation of the rete ridges with foamy histiocytic dermal infiltration . The histiocytes may be subtle, and misdiagnosis as a verruca is possible if they are missed; these histiocytes contain periodic acid‐Schiff‐positive cytoplasmic granules …”

Section: Discussionmentioning

confidence: 99%

“…No association has been identified with human papillomavirus despite the papillomatous morphology nor with disorders of lipid metabolism despite the presence of lipid‐laden histiocytes . An etiologic hypothesis is epidermal damage from inflammation or trauma driving psoriasiform hyperplasia, epithelial lipid production, and histiocyte recruitment which become foam cells upon phagocytosis of degenerating keratinocytes and lipid droplets . Ultrastructural studies have demonstrated keratinocyte degeneration …”

Section: Discussionmentioning

confidence: 99%

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Verruciform xanthoma in a patient with recessive dystrophic epidermolysis bullosa: Case report and literature review

Stephens

Rubin

Perman

2019

Pediatric Dermatology

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Verruciform xanthoma (VX) is a rare finding thought to be caused by epidermal damage from trauma or inflammation and has been reported in a limited number of patients with recessive dystrophic epidermolysis bullosa (RDEB). Herein, we describe a 20‐year‐old woman with RDEB who developed a large, verrucous, pink plaque on the posterior thigh that was histologically proven to be a VX. We review cases of VX in patients with RDEB and summarize the clinical features, pathophysiology, and management principles.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Verruciform xanthoma in a patient with recessive dystrophic epidermolysis bullosa: Case report and literature review

Stephens

Rubin

Perman

2019

Pediatric Dermatology

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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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“…In the literature, no conclusive evidence of the malignant transformation of preexisting VX exists (4); although, there are some reports of no oral VX associated with SCC (12,13). Murat-Susić et al (6) presented a case of VX associated with epidermolysis bullosa and highlighted that the concomitant presence of VX in patients with epidermolysis bullosa could pose an increased risk of SCC occurrence. Drummond et al (10) reported one case of VX associated with carcinoma in situ and high epithelium dysplasia adjacent to SCC, located in the buccal floor.…”

Section: Discussionmentioning

confidence: 99%

Verruciform xanthoma located in anterior gingiva

Hatakeyama¹,

Guilhermino²,

Brandão³

et al. 2010

J Clin Exp Dent

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Verruciform xanthoma (VX) is a relatively rare benign lesion and oral predominantly, which occasionally affects skin and genital mucosa. It appears as a papule or single plaque showing a verrucous or papillomatous aspect, with variable color from reddish pink to gray. In majority of oral cases, it affects gingiva and alveolar mucosa such a solitary lesion. Histopathological findings are foamy histiocytes within elongated dermal papillae. Treatment consists of conservative excision surgery and recurrence is rare. A clinical case is reported, located in anterior gingiva, showing good prognosis, without recurrence. There was no concomitant oral lesion associated. Local trauma was the only possibility suggested to be related to etiology. No relevant alterations in laboratory exams (hemogram, total cholesterol, HDL, LDL, VLDL, glycose and glycolized hemoglobin) were found. The origin of the lesion remains unclear and investigation for possible associations with other lesions that could present greater risk of carcinogenesis is required.

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Verruciform xanthoma in recessive dystrophic epidermolysis bullosa Hallopeau‐Siemens

Abstract: 956Case report Verruciform xanthoma in RDEB Murat-Su 1 i 2 et al.

Cited by 15 publications

References 51 publications

Verruciform xanthoma in a patient with recessive dystrophic epidermolysis bullosa: Case report and literature review

Verruciform xanthoma in a patient with recessive dystrophic epidermolysis bullosa: Case report and literature review

Verruciform Xanthoma Developing in Recessive Dystrophic Epidermolysis Bullosa

Verruciform xanthoma located in anterior gingiva

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