Concurrent localized scleroderma and discoid lupus erythematosus. Cutaneous 'mixed' or 'overlap' syndrome

Umbert, P.

doi:10.1001/archderm.114.10.1473

Cited by 32 publications

(31 citation statements)

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“…Thus, taking into account the above reports [4, 5] similar to ours, it is conceivable to consider the linear distribution as the clinical hallmark of this unique, ‘sclerodermic’ subset of LEP. Segmental loss of fat involving the face and/or the upper part of the body has also been described in partial lipodystrophy, which is a rare disease occurring in children and young adults and predominantly affecting females [18].…”

Section: Discussionsupporting

confidence: 66%

“…Alternatively, as suggested for several disorders commonly or rarely characterized by a linear pattern, including herpes zoster, psoriasis, lichen planus or cutaneous lymphoma [9, 17], various causative factors, such as trauma, Köbner phenomenon, viral agents or immunological pathomechanisms, may be regarded as responsible for this unique clinical presentation. On the other hand, a case with linear scleroderma and lupus developing sequentially in the same lesion has been reported by Umbert and Winkelmann [5]; these authors speculated that either both diseases coexisted for a long time or one evolved into the other, or a new entity of ‘linear’ LE mimicking linear scleroderma developed. The latter may be considered, in our opinion, as the first reported case of sclerodermic linear LEP.…”

Section: Discussionmentioning

confidence: 75%

“…Cutaneous lesions of LE with a linear dermatological pattern are rare [1,2,3,4,5,7,8,9,10,11,12,13,14,15,16]. To the best of our knowledge, only 4 cases of LEP with linear distribution have been reported in the literature [1,2,3,4].…”

Section: Discussionmentioning

confidence: 99%

“…A linear configuration of LEP has very rarely been described [1,2,3,4]. On the other hand, rare descriptions of the coexistence of cutaneous LE and localized scleroderma or morphea have been reported [5, 6]. …”

Section: Introductionmentioning

confidence: 99%

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Sclerodermic Linear Lupus Panniculitis: Report of Two Cases

Marzano¹,

Tanzi²,

Caputo³

et al. 2005

Dermatology

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Lupus erythematosus panniculitis is a rare disease characterized by deep subcutaneous nodules, most commonly localized on the upper limbs and face. Unique clinical presentations, such as linear configuration or ‘overlap’ forms between lupus erythematosus panniculitis and localized scleroderma have been reported. We present here the clinical characteristics, course and laboratory findings of 2 patients having linear lupus erythematosus panniculitis with localized scleroderma-like changes. The 2 patients (of the 14 patients with lupus erythematosus panniculitis seen by us since 1990) were females with a young age at the onset of disease (median, 25 years). In 1 case, evolution into systemic lupus erythematosus with severe renal involvement occurred whereas the other patient, who had a spontaneous abortion and exhibited anticardiolipin antibodies, should be followed and screened for the emergence of antiphospholipid syndrome. Thus, the clinical behavior of this variant seems to be more aggressive, as compared with the usual course of lupus erythematosus panniculitis, which is considered to be a benign disease, although some reports have suggested that its prognosis is not always favorable. The linear distribution could be the clinical hallmark of such a unique, ‘sclerodermic’ subset of lupus erythematosus panniculitis.

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Section: Discussionsupporting

confidence: 66%

Section: Discussionmentioning

confidence: 75%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Sclerodermic Linear Lupus Panniculitis: Report of Two Cases

Marzano¹,

Tanzi²,

Caputo³

et al. 2005

Dermatology

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“…Linear Cutaneous Lupus Erythematosus (LCLE) was first described in 1978 [2]. It is a term proposed for DLE with a linear configuration [3].…”

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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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