Coexistence of segmental vitiligo, scleroderma en coup de sabre and cleft lip on the same hemiface: association with mosaicism?

Ubaldo, Heloísa Del Castanhel; Castro, Caio César Silva de

doi:10.1590/abd1806-4841.20198110

Cited by 6 publications

(8 citation statements)

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“…11 different reports were found which makes it by far the most frequently observed comorbidity [57.1% (12/21 patients) of reported patients with autoimmune comorbidities]. In most cases, the morphea lesions affected the same body area as the SV, although multifocal morphea was also documented (36)(37)(38)(39)(40)(41)(42)(43)(44)(45)(46). Cases with lichen striatus are interesting because of the striking similar pathogenesis (48) (Figure 1).…”

Section: Cases Of Segmental Vitiligo Displaying Additional Skin Disormentioning

confidence: 99%

Autoimmunity in Segmental Vitiligo

et al. 2020

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The autoimmune basis of segmental vitiligo (SV) has only recently been recognized. Systemic autoimmune diseases are less frequently associated compared to non-segmental vitiligo (NSV), but localized skin disordersin particular linear morpheahave been repeatedly observed in patients with SV. The inflammatory response is documented on a clinical level with cases displaying erythematous borders or a hypochromic stage, on a histopathological level with predominantly CD8 lymphocytes migrating toward the basal layer and by flow cytometry demonstrating the antimelanocyte specificity of these cytotoxic T cells. The increased risk for halo naevi and NSV in these patients further underline the immunemediated mechanisms of SV. Nonetheless, the localized and unique distribution pattern points to somatic mosaicism. This places SV in a category of similar diseases such as lichen striatus, blaschkitis, linear lupus erythematosus, and linear scleroderma where an immune reaction against genetically mutated skin cells is believed to be the underlying cause. All these disorders are characterized by a young age of onset, a temporary disease activity with spontaneous resolution, limited response to treatment, and often long-term sequelae. Although challenging, genetic research proving this genetic mosaicism could offer crucial insights into the pathogenesis of both segmental and non-segmental vitiligo.

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Section: Cases Of Segmental Vitiligo Displaying Additional Skin Disormentioning

confidence: 99%

Autoimmunity in Segmental Vitiligo

et al. 2020

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“…Many reports describe cases of concomitant generalized vitiligo and morphea. Instead, the association between SV and segmental morphea had been rarely reported[ 2 3 4 ] and is summarized in Table 1 .…”

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confidence: 99%

Concomitant Segmental Vitiligo and Segmental Morphea in an 8-year-old boy

Magri

Miraglia

Soda

et al. 2021

Indian Journal of Dermatology

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“…У пациентки с линейной склеродермией, расположенной в области лба («en coup de saber») слева, наблюдалось витилиго, локализованное в левой периорбитальной области с полиозом бровей и ресниц, а также имелась левосторонняя расщелина губы при рождении [35]. Изучая данный клинический случай, можно видеть, что между половинами лица больной существует дискретная асимметрия без признаков подкожной, мышечной или костной атрофии, помимо той, которая наблюдается в области поражения ЛС [35,36]. В этом клиническом случае гемифациальные проявления витилиго, ЛС и расщелины губы были расценены как редкие [35].…”

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“…Изучая данный клинический случай, можно видеть, что между половинами лица больной существует дискретная асимметрия без признаков подкожной, мышечной или костной атрофии, помимо той, которая наблюдается в области поражения ЛС [35,36]. В этом клиническом случае гемифациальные проявления витилиго, ЛС и расщелины губы были расценены как редкие [35]. Данная клиническая ситуация подтверждает возможную связь этих состояний с явлениями эмбриологического деления клеток, подчеркивая мозаицизм.…”

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“…Эта гипотеза может объяснить как изменения в миграции клеток, вызвавших расщелину губы, так и изменения в некоторых эпидермальных клетках, которые становятся более восприимчивыми к аутоиммунным атакам. Витилиго или склеродермия могут также чаще проявляться после воздействия неопределенных триггеров [35,36], вследствие чего гипотеза участия мозаицизма в аутоиммунных дерматологических заболеваниях, которые проявляются в локальных формах, таких как склеродермия и витилиго, подтверждается [36,37].…”

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Etiopathogenetic Similarities of Combined Forms of Localized Scleroderma and Vitiligo

Мурашкин

Савелова

Иванов

et al. 2020

Vopr. sovr. pediatr.

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Vitiligo is a common skin disease characterized by idiopathic progressive skin hypomelanosis. Vitiligo is associated with several comorbid autoimmune diseases such as localized scleroderma. This article demonstrates the general development mechanism of these pathologies, as well as the key aspect of cross-effect between autoimmune diseases on the molecular level. Recently, dermatologists have noted the increasing number of patients suffering from combined pathologies. Such patients (in pediatrics) have torpid course of disease and no pathognomonic symptoms. That exaggerates the diagnostics and adequate therapy prescription. This leads to increased awareness among physicians of different specialties on possible combinations, clinical presentation and pathogenesis aspects of such conditions.

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Coexistence of segmental vitiligo, scleroderma en coup de sabre and cleft lip on the same hemiface: association with mosaicism?

Cited by 6 publications

References 5 publications

Autoimmunity in Segmental Vitiligo

Autoimmunity in Segmental Vitiligo

Concomitant Segmental Vitiligo and Segmental Morphea in an 8-year-old boy

Etiopathogenetic Similarities of Combined Forms of Localized Scleroderma and Vitiligo

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