Linear lupus erythematosus profundus on the face, following the lines of Blaschko

Tamiya, Hisashi; Sowa, Junko; Nakanishi, Takeshi; Adachi, M.; Ishii, Masamitsu; Kobayashi, Hiromi

doi:10.1111/j.1365-4632.2009.04285.x

Cited by 12 publications

(17 citation statements)

References 4 publications

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“…In our patient, linear LEP was an initial manifestation of SLE at a young age. Of the eight other cases of linear LEP reported in the literature, none progressed to SLE during the follow‐up period reported (1–7 years) . The authors report this case to illustrate an unusual superimposed segmental manifestation of an inflammatory polygenic disorder.…”

Section: Discussionmentioning

confidence: 72%

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Linear Lupus Erythematosus Profundus as an Initial Manifestation of Systemic Lupus Erythematosus in a Child

Fernandes

Santos²,

Freitas

et al. 2012

Pediatric Dermatology

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The case of a 13-year-old Caucasian girl with a 1-year history of a linear plaque on her thigh is reported. Histopathologic examination of the incisional biopsy tissue established the diagnosis of lupus erythematosus panniculitis. Six months later she developed the clinical and laboratory criteria for the diagnosis of systemic lupus erythematosus (SLE). She was treated with azathioprine and oral prednisolone, with a favorable clinical outcome. Our case illustrates a child with linear lupus erythematosus profundus as an initial manifestation of SLE. To our knowledge, only eight other cases of linear lupus erythematosus profundus have been reported in the literature, five in children. In contrast to our patient, none of those cases progressed to SLE during the reported follow-up period. The authors report this case to illustrate an unusual superimposed segmental manifestation of an inflammatory polygenic disorder.

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Section: Discussionmentioning

confidence: 72%

“…The linear form of LEP is extremely uncommon. To our knowledge, only eight other cases have been reported in the English literature, five in children, but none of those cases progressed to SLE during the follow-up period reported (3)(4)(5)(6)(7)(8)(9)(10).…”

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confidence: 95%

Linear Lupus Erythematosus Profundus as an Initial Manifestation of Systemic Lupus Erythematosus in a Child

Fernandes

Santos²,

Freitas

et al. 2012

Pediatric Dermatology

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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.…”

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confidence: 99%

“…The lesions are usually asymptomatic, with a low incidence of photosensitivity. This unique presentation can be considered as DLE following the lines of Blaschko, but other types of lupus with a linear distribution, such as Lupus Erythematosus Profundus (LEP), have also been described in some pediatric patients [15][16][17][18][19][20]. Most reported findings in DIF are variable and DIF was not performed in nearly half of the cases.…”

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confidence: 99%

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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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Arc-Shaped Furrow on the Forehead Extending From Linear Alopecia on the Vertex

2022

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A man in his 50s presented to our dermatology department with an asymptomatic, arcshaped, depressed skin groove on the left side of his forehead, frontal scalp, and vertex area. Localized linear hair loss and hair thinning on the left scalp area were observed. The patient reported that when the lesion had begun to appear, approximately 10 years earlier, he had received 2 doses of intralesional steroid via injection on a small bald patch on the vertex. Thereafter, the lesion gradually progressed to the frontal scalp and eventually made a linear furrow on the left side of his forehead.Physical examination revealed 2 long, curved-line, depressed, alopecic patches on the left side of the vertex and frontal scalp area extending to the left eyebrow (Figure , A and B). The lesions measured (length × width) 7 × 2 cm and 6 × 1 cm, respectively. Dermoscopic examination of the alopecic area revealed several short vellus hairs, yellow dots, and honeycomb hyperpigmentation; however, broken hairs were not seen. The result of a hair pull test was negative. The patient denied any trauma history and had no history of connective tissue disease nor any evidence of systemic involvement, such as arthralgia, photosensitivity, or skin rashes. Skin biopsies were performed on the left side of the vertex and the forehead. Histopathologic findings of the 2 biopsy specimens were identical (Figure, C and D).

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Linear lupus erythematosus profundus on the face, following the lines of Blaschko

Cited by 12 publications

References 4 publications

Linear Lupus Erythematosus Profundus as an Initial Manifestation of Systemic Lupus Erythematosus in a Child

Linear Lupus Erythematosus Profundus as an Initial Manifestation of Systemic Lupus Erythematosus in a Child

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

Arc-Shaped Furrow on the Forehead Extending From Linear Alopecia on the Vertex

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