Linear Childhood Discoid Lupus Erythematosus Following the Lines of Blaschko: A Case Report with Review of the Linear Manifestations of Lupus Erythematosus

Green, Justin J.; Baker, Donald J.

doi:10.1046/j.1525-1470.1999.00030.x

Cited by 51 publications

(60 citation statements)

References 41 publications

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“…12,13 These conditions are believed to be caused by genetic mosaicism. 2 The explanation suggested is the occurrence of post-zygotic mutation in the keratinocytes, giving rise to abnormal cells that are cloned and migrate in a single direction during embryogenesis.…”

Section: Discussionmentioning

confidence: 99%

“…2 The explanation suggested is the occurrence of post-zygotic mutation in the keratinocytes, giving rise to abnormal cells that are cloned and migrate in a single direction during embryogenesis. 5,6,12 The cells arising from these mosaicisms express neoantigens capable of eliciting local immune response. 3 The trigger for the onset of the disease may be trauma, primary irritation or exogenous agents such as ultraviolet light, drugs, pesticides, heavy metals or other elements.…”

Section: Discussionmentioning

confidence: 99%

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Lúpus eritematoso crônico discoide nas linhas de Blaschko

Daldon

Lage

2011

An. Bras. Dermatol.

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Linear chronic discoid lupus erythematosus is a rare manifestation of cutaneous lupus in which erythematous, atrophic, dyschromic lesions are located along the lines of Blaschko. This report describes the case of a 15-year old boy with a 2-year history of discoid, erythematous, hyper and hypopigmented lesions with central atrophy, situated along the lines of Blaschko on his right arm. Histopathology showed epidermal atrophy, hyperkeratosis, follicular plugging, thickening of the basement membrane zone, and superficial and deep chronic perivascular and periadnexal inflammatory infiltrate, with dermal mucin deposit, thus confirming diagnosis. A total of 14 cases have been described of this variant, the onset of which is often in childhood. There is no difference in incidence between genders. Lesions most commonly develop on the face. There have been no reports of any association with systemic disease. Keywords: Lupus erythematosus, cutaneous; Lupus erythematosus, discoid; Mosaicism Resumo: O lúpus eritematoso crônico discoide linear é manifestação rara da doença lúpica cutânea, em que lesões eritêmato-atrófico-discrômicas dispõem-se nas linhas de Blaschko. Descrevemos o caso de um menino de 15 anos, com dois anos de história de lesões discoides eritêmato-atróficas, hipo e hiperpigmentadas, dispostas nas linhas de Blaschko do membro superior direito. O exame histopatológico revelou atrofia da epiderme, hiperqueratose, rolhas córneas, espessamento da zona da membrana basal, infiltrado inflamatório crônico perianexial e perivascular superficial e profundo, depósito de mucina na derme, confirmando o diagnóstico. Há, no total, 14 casos descritos dessa variante que se inicia frequentemente na infância e que não apresenta predomínio quanto ao sexo. As lesões ocorrem preferencialmente na face. Não há relatos de associação com doença sistêmica. Palavras-chave: Lupus eritematoso cutâneo; Lupus eritematoso discóide; Mosaicismo

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Lúpus eritematoso crônico discoide nas linhas de Blaschko

Daldon

Lage

2011

An. Bras. Dermatol.

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“…20 Cases with localized lesions are on record. 21,22 Other disorders with telangiectasia on the face such as rosacea, carcinoid, seborrheic dermatitis, hereditary hemorrhagic telangiectasia (history of oro-nasal bleed and involvement of mucosa), and bloom syndrome (history of photosensitivity) may be differentiated by the absence of skin atrophy. Facial telangiectasia due to prolonged steroid application (history of topical application of high potency steroid) and angiolupoid sarcoidosis can be differentiated by classical histologic findings.…”

Section: Secondary Telangiectasia With Skin Atrophymentioning

confidence: 99%

A clinical approach to diagnose patients with localized telangiectasia

et al. 2015

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Telangiectasia, or dilated blood vessels, may represent a benign condition or a marker of a much more serious internal or cutaneous malignancy (e.g. mycosis fungoides). They can be generalized or localized in distribution. ‘Localized telangiectasia? can be macular or papular. Macular ones can be further divided into two major categories: primary and secondary. They can occur on their own (primary), after skin injury (e.g. sun damage, post‐radiation therapy, after prolonged topical steroid application), or may arise due to an underlying condition (secondary). While telangiectases can spring from a multitude of possible causes, the location of the lesions, in combination with a careful history and exmination, often helps in elucidating the correct diagnosis. A patient with localized telangiectasia as a manifestation of malignancy may be difficult to diagnose, however the diagnosis is one of exclusion, and a thorough evaluation should be performed before diagnosing this condition. This article reviews possible conditions to consider in these patients and suggests an approach to their evaluation.

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“…Till now, fewer than 30 cases in both adults and children have been published [4,5]. Among them, only 15 cases of childhood diagnosed as facial LCLE have been reported (Table 1) [3,[6][7][8][9][10][11][12][13][14][15][16]. Nearly half of the cases presented with lesions before the age of 7 years.…”

mentioning

confidence: 99%

“…Previously reported therapies for facial LCLE of childhood include topical steroids [7,8,10,[14][15][16], pimecrolimus [11], tarcrolimus [12][13][14], oral steroids [16], dapsone [3,8], hydroxychloroquine [6,9,11,13], isotretinoin [11], and intense pulsed light [11]. However, patients show various responses to these therapies.…”

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confidence: 99%

Functional Regulation Triad of Mitosis, Apoptosis and Thigmotropism of Survivin in Cutaneous Malignant Melanoma

Chen¹

2016

CRDOA

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A 5-year-old girl went to our clinic presented with a more than 1 year history of linear and annular erythematous patches and telangiectasia from right cheek to right forehead (Figure1a), which was consistent with the implemented lines of Blaschko on the head (Figure1b). Some mild atrophic center over right forehead was noted (Figure1c). She was treated with topical steroids by other dermatologists for more than 1 year but in vain. There were no oral ulcer, alopecia, muscular weakness, or arthralgia, but photosensitivity was noted. The clinical impression was Discoid Lupus Erythematosus (DLE). The initial screening laboratory examination results revealed negative Antinuclear Antibodies (ANA), and positive anti-ENA antibodies. The complement C3, C4, and complete blood count were all within normal limits. The further laboratory examination confirmed positive anti-nDNA antibodies (45.3 IU/ml, >10 IU/ml), and negative anti-Sm/RNP, anti-SSA or anti-SSB antibodies. A skin biopsy was performed on the right forehead and the sample was sent for histological and Direct Immuno fluorescent Examination (DIF). Microscopic features showed papillary dermal edema and perivascular and periappendageal infiltration in both superficial and deep dermis. There were increased dermal mucin deposition and perivascular lymphocytic infiltration (Figure 2). The higher magnification showed interface dermatitis in the epidermis characterized by presence of basal cell vacuolar change, lymphocyte exocytosis, and dyskeratotic cells (Figure 3). Pigment incontinence was present in the papillary dermis. The DIF of the skin biopsy demonstrated a thick linear-granular IgG deposition in the basement membrane zone (Figure 4). There were also granular IgA, IgM, and C3 deposition at the dermo-epidermal junction. These findings were consistent with cutaneous lupus erythematosus. The systemic workup revealed normal chest x ray, and urianalysis. The repeated laboratory workup showed complete blood count, renal profile, total protein and albumin were within normal limits. The ANA, anti-dsDNA antibody, anticardiolipin IgM antibody, C3, C4, and direct Coombs test were all negative. Oral hydroxychloroquine 100mg a day was then prescribed. The skin lesions improved after treatment. DiscussionThe clinical presentation of childhood DLE is similar to that of the adult form. Over 50% of patients presented with lesions before the age of 10 years. The remainder presented between the ages of 10 and 16 years [1]. Unlike adults with a female predominance, it seems that there is no sex association in children. Some authors AbstractWe report on a 5-year-old girl with a pruritic, erythematous cutaneous eruption on the right side of the face for more than 1 year. The lesion had a linear distribution following the lines of Blaschko. Histopathological findings and direct immunofluorescence were compatible with cutaneous lupus erythematosus. We consider this unusual clinical presentation of childhood facial "Blaschko-linear cutaneous lupus erythematosus" to be a distinct subtyp...

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Linear Childhood Discoid Lupus Erythematosus Following the Lines of Blaschko: A Case Report with Review of the Linear Manifestations of Lupus Erythematosus

Cited by 51 publications

References 41 publications

Lúpus eritematoso crônico discoide nas linhas de Blaschko

Lúpus eritematoso crônico discoide nas linhas de Blaschko

A clinical approach to diagnose patients with localized telangiectasia

Functional Regulation Triad of Mitosis, Apoptosis and Thigmotropism of Survivin in Cutaneous Malignant Melanoma

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