Between Light and Dark, the Chimera Comes Out

Lipsker, Dan; Flory, Elisabeth; Wiesel, Marie-Louise; Hanau, D.; Salle, Henri

doi:10.1001/archderm.144.3.327

Cited by 30 publications

(16 citation statements)

References 12 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…In addition, there have been some cases of pigmentary mosaicism with both hyperpigmentation and hypopigmentation. However, all of these reported cases (3)(4)(5)(6)(7)(8)(9)(10) displayed hyperpigmentation that was present apart from hypopigmentation in each line or macule, unlike our present patient.…”

Section: Discussioncontrasting

confidence: 76%

See 1 more Smart Citation

A Finely Wrinkled, Band-like Naevus Associated with Hyper- and Hypopigmented Spots: A New Entity?

Mizuashi¹,

Tsunoda²,

Aiba³

et al. 2009

Acta Derm Venerol

View full text Add to dashboard Cite

We report here a case of a Japanese man with an unusual congenital band-like naevus, consisting of a finely wrinkled skin area with a mosaic of hyper-and hypopigmented spots involving only the right side of his chest. To our knowledge, there are no previous reports of this presentation of congenital naevus. CASE REPORTA 36-year-old Japanese man presented with a band-like pigmentedareaononly the right side of his chest, which had been present since birth. As there had been no particular change in either its colour or size, he had not consulted a dermatologist previously. There was no family history of a similar skin change.On his first visit, he had a well-suntanned skin due to his hobby of marine sports., together with a roughly S-shaped. band-like, and mildly pigmented., lesion localized only on the right side ofthe mid-chest ( Fig. la). Although the skin texture appeared rough due to the presence of fine wrinkles, the lesions were in fact smooth on palpation. On closer inspection, the mildly pigmented plaque appeared more wrinkled than the surrounding normal skin, presenting a reticular pattern dotted with hyper-and hypopigmented spotty areas (Fig. lb). The spotty changes were composed of variously sized, darkly hyperpigmemed, hypopigmented or almost nornial looking, finely wrinkled areas, which did not appear to conform to any ofthe so-far reported cases of congenital skin lesions. He had no other abnormalities in the skin, hair or teeth, and no clinically observable sexual ambiguity or family history of similar skin changes.Histologically, in the darkly hyperpigmented spot, we found atrophie epidermis and basal hypermelanosis that was prominently noted at the tips of elongated rete ridges whose covering stratum corneum was somewhat thickened and compact containing scattered melanin granules. Moreover, there were scattered melanophages among a mild mononuclear cell infiltrate at the perivascular portion ofthe upper dermis. Neither a nest of melanocytes nor sclerosis ofthe dermis was detectable (Fig. 2a). Other mildly hyperpigmented portions showed thin epidermis, mild basal hypermelanosis and a few melanophages in the upper dermis. On the other hand, the hypopigmented spot revealed a thin epidermis covered by rather thick and compact stratum corneum with no changes suggestive of scar or amyloid deposition in the dermis (Fig. 2b). There were neither hair follicles nor eccrine sweat glands in the specimens. He did not require any further treatment for his skin lesion. DISCUSSIONTo our knowledge, the skin changes found in this patient are unique among the types of congenital naevus described to date. Its clinical feature had been stable since birth, showing a well-defined, band-like, finely wrinkled, mildly pigmentary skin area confined to the right side ofthe chest. On a closer examination, this consisted of hyper-, hypo-, and even depigmented spotty areas. If there had been any telangiectatic component, we could have described it as poikilodermatous. Although there was no clinical sign of inflammation, histolog...

show abstract

Section: Discussioncontrasting

confidence: 76%

“…chimerism. chromosomal mosaicism, somatic mutations, and half-chromatid mutations (3)(4)(5)(6)(7)(8)(9)(10). Thus, this group of disorders has been suggested to be "pigmentary mosaicism" in general (5).…”

Section: Discussionmentioning

confidence: 99%

A Finely Wrinkled, Band-like Naevus Associated with Hyper- and Hypopigmented Spots: A New Entity?

Mizuashi¹,

Tsunoda²,

Aiba³

et al. 2009

Acta Derm Venerol

View full text Add to dashboard Cite

show abstract

“…1,2,7 Thus, the characteristics of this disease will be distributed along the Balschko lines or other mosaic patterns, corresponding to cells containing the mutation. 2,5,8 The rest of the skin will be normal genotypically and phenotypically. In general, this type of mosaicism is not inherited, except when the mutation affects the gonads.…”

Section: Classification Of Cutaneous Mosaicismsmentioning

confidence: 99%

Cutaneous mosaicisms: concepts, patterns and classifications

Kouzak

Mendes

Costa

2013

An. Bras. Dermatol.

View full text Add to dashboard Cite

A mosaic is an organism composed of two or more genetically distinct cell populations derived from a genetically homogeneous zygote. Cutaneous mosaicisms are the clinical expressions of these disorders. The main event which allows the existence of mosaicism is a genetic mutation, either structural or functional. Cutaneous mosaicisms usually manifest by specific patterns on the skin and the archetypic pattern is the system of Blaschko lines, but others include checkerboard, phylloid, large patches without midline separation and lateralization. Since 1901, when Blaschko lines were first described, the study of mosasicism has helped to elucidate the behavior of numerous genetic diseases, generating therapeutic perspectives for these pathologies, including the promising gene therapy.

show abstract

“…3 The topographic co-localisation of both segmental vitiligo and DH could be explained by mosaic genetic variation, for example in the HLA-alleles, for both conditions. 4 There has been only one prior case report in which DH localised mostly, but not exclusively, within depigmented areas of vitiligo in a patient with autoimmune thyroiditis. …”

Section: Photoletter To the Editor Abstractmentioning

confidence: 99%

Dermatitis herpetiformis co-localised with vitiligo in a patient with autoimmune polyglandular syndrome

Macbeth

Lee²,

Levell

et al. 2013

J Dermatol Case Rep

View full text Add to dashboard Cite

A 37-year-old female presented with a 3-day history of a severe, pruritic, bullous eruption on the trunk and limbs associated with malaise. Her past medical history included Addison's disease, autoimmune hypothyroidism, type 1 diabetes mellitus, coeliac disease and vitiligo. Coeliac disease was diagnosed following distal duodenal biopsy which demonstrated subtotal villous atrophy. The patient reported strict adherence to a gluten-free diet. Vitiligo on the limbs, trunk, and neck was treated with moderate potency topical corticosteroids fourteen years prior to this acute presentation. Her non-identical twin sister also suffered from autoimmune hypothyroidism and vitiligo. On examination, multiple, well-demarcated, segmental areas of depigmentation were present on the forearms, neck, groin, and upper thighs. There were vesicles and bullae of up to 20 mm in diameter with adjacent crusted, erythematous papules exclusively within the depigmented areas but not within the pigmented patches (Fig. 1).Indirect immunofluorescence, ANA, rheumatoid factor, and viral and bacterial swabs were unremarkable. Biopsy from DOI: http://dx.doi.org/10.3315/jdcr.2013.1153 101 PHOTOLETTER TO THE EDITOR AbstractWe report a case of dermatitis herpetiformis co-localised with segmental vitiligo in a 37-year-old woman with a background history of autoimmune polyglandular syndrome type 2. We propose genetic mosaicism as a possible mechanism. There has only been one previous case report in which dermatitis hepetiformis co-localised in close proximity but not exclusively within vilitigo in a patient with autoimmune thyroiditis. To our knowledge, this is the first case report of dermatitis herpetiformis co-localised exclusively to segmental vitiligo in the presence of autoimmune polyglandular syndrome. (J Dermatol Case Rep. 2013; 7(3): 101-102)

show abstract

Between Light and Dark, the Chimera Comes Out

Abstract: Background: Chimerism, especially in the absence of sexual ambiguity, is extremely rare in humans. We report the case of a 6-year-old boy whose skin pigmentary abnormalities revealed chimerism.

Cited by 30 publications

References 12 publications

A Finely Wrinkled, Band-like Naevus Associated with Hyper- and Hypopigmented Spots: A New Entity?

A Finely Wrinkled, Band-like Naevus Associated with Hyper- and Hypopigmented Spots: A New Entity?

Cutaneous mosaicisms: concepts, patterns and classifications

Dermatitis herpetiformis co-localised with vitiligo in a patient with autoimmune polyglandular syndrome

Contact Info

Product

Resources

About