Old lines tell new tales: Blaschko linear lupus erythematosis

Jin, Hui; Zhang, Guiying; Zhou, Ying; Chang, Christopher; Lu, Qianjin

doi:10.1016/j.autrev.2015.11.014

Cited by 32 publications

(55 citation statements)

References 70 publications

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“…To date, less than 100 cases of LCLE have been described in the literature, and approximately half of these occurred in childhood. The youngest patient reported to present with LCLE an 18‐month‐old girl . Analysis of the published cases reveals similar incidence in both sexes and no ethnic predilection …”

Section: Clinical Course and Discussionmentioning

confidence: 84%

“…Linear cutaneous lupus erythematosus represents a rare form of cutaneous lupus erythematosus, usually presenting with erythematous or brownish papules and plaques, atrophic or hyperkeratotic lesions, distributed along the lines of Blaschko, translating the migration pattern of keratinocytes during embryogenesis . Similar to other blaschkolinear dermatoses, these mosaic keratinocytes arise from somatic (postzygotic) mutation creating an aberrant clone which remains silent until a certain triggering event occurs . The differential diagnosis includes other dermatoses with linear pattern along the lines of Blaschko, such as lichen planus, lichen striatus, lichen nitidus, lichen sclerosus, linear granuloma annulare, psoriasis, or blaschkitis.…”

Section: Clinical Course and Discussionmentioning

confidence: 99%

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Unilateral erythematous linear lesions on the face of a 2‐year‐old infant

Kieselova

Santiago

Cunha

et al. 2019

Pediatric Dermatology

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A healthy 2-year-old girl presented to dermatology with an asymptomatic cutaneous eruption on the left cheek. According to her parents, the skin lesions had begun as small papules and pustules 4 months prior.On physical examination, the eruption consisted of confluent linear erythematous plaques and papules on the chin and upper margin of the mandible extending to the left mandibular angle, following along the lines of Blaschko (Figures 1 and 2). The patient had no previous history of trauma or photosensitivity. Her family history was negative for similar rashes. The parents had applied several topical creams, including sertaconazol, fucidic acid, hydrocortisone, with no improvement.Complete blood count, hepatic, and renal function panels were normal.Antinuclear antibodies were negative. A punch biopsy from the left mandibular area was performed (Figure 3).

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Section: Clinical Course and Discussionmentioning

confidence: 84%

Section: Clinical Course and Discussionmentioning

confidence: 99%

Unilateral erythematous linear lesions on the face of a 2‐year‐old infant

Kieselova

Santiago

Cunha

et al. 2019

Pediatric Dermatology

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“…This explains the focal nature of LBLE, the lack of systemic manifestation, and, often, seronegativity. Treatment is similar to that for other types of cutaneous lupus, but the response rate varies .…”

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confidence: 96%

Progressive linear blaschkoid lupus erythematosus

Campos-Muñoz

Fueyo-Casado

Conde‐Taboada

et al. 2017

Arthritis & Rheumatology

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“…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.…”

mentioning

confidence: 99%

“…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.…”

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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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Old lines tell new tales: Blaschko linear lupus erythematosis

Cited by 32 publications

References 70 publications

Unilateral erythematous linear lesions on the face of a 2‐year‐old infant

Unilateral erythematous linear lesions on the face of a 2‐year‐old infant

Progressive linear blaschkoid lupus erythematosus

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

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