Unilateral nevoid telangiectasia: case report and proposal for new classification system

Tanglertsampan, Chuchai; Chanthothai, Jetchawa; Phichawong, Thanaphat

doi:10.1111/j.1365-4632.2011.05279.x

Cited by 20 publications

(15 citation statements)

References 26 publications

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“…Until today, unilateral naevoid telangiectasia of the patchy type is a poorly defined disorder . It is included here for heuristic reasons.…”

Section: Capillary Malformations That Perhaps Represent Naevimentioning

confidence: 99%

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Capillary malformations: a classification using specific names for specific skin disorders

Happle

2015

Acad Dermatol Venereol

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The name capillary malformation has caused much confusion because it is presently used to designate numerous quite different disorders such as naevus flammeus, the salmon patch, the vascular naevus of the hereditary 'megalencephaly-capillary malformation syndrome' and the skin lesions of non-hereditary traits such as 'capillary malformation-arteriovenous malformation' and 'microcephaly-capillary malformation'. To avoid such bewilderment, the present review describes the distinguishing clinical and genetic criteria of 20 different capillary malformations, and a specific name is given to all of them. The group of capillary naevi includes naevus flammeus, port-wine naevus of the Proteus type, port-wine naevus of the CLOVES type, naevus roseus, rhodoid naevus, cutis marmorata telangiectatica congenita, congenital livedo reticularis, segmental angioma serpiginosum, naevus anaemicus, naevus vascularis mixtus and angiokeratoma circumscriptum. Capillary lesions that perhaps represent naevi are the mesotropic port-wine patch, Carter-Mirzaa macules, unilateral punctate telangiectasia and unilateral naevoid telangiectasia of the patchy type. Capillary malformations that do not represent naevi include X-linked angiokeratoma corporis diffusum (Fabry disease), autosomal dominant angiokeratoma corporis diffusum, hereditary haemorrhagic telangiectasia, hereditary angioma serpiginosusm and the salmon patch. In this way, we are able to discriminate between various non-hereditary capillary naevi such as naevus roseus and the hereditary rhodoid naevus and several hereditary traits that do not represent naevi such as angiokeratoma corporis diffusum and hereditary haemorrhagic telangiectasia; between four different types of port-wine stains, three of them being lateralized and one being mesotropic; between cutis marmorata telangiectatica congenita and congenital livedo reticularis; between telangiectatic naevi and the vasoconstrictive naevus anaemicus; and between two different types of angiokeratoma corporis diffusum. Finally, arguments are presented why the salmon patch ('stork bite', 'naevus simplex') cannot be categorized as a naevus.

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“…Until today, unilateral naevoid telangiectasia of the patchy type is a poorly defined disorder . It is included here for heuristic reasons.…”

Section: Capillary Malformations That Perhaps Represent Naevimentioning

confidence: 99%

“…107,112,113 Unilateral naevoid telangiectasia of the patchy type Until today, unilateral naevoid telangiectasia of the patchy type is a poorly defined disorder. [114][115][116] It is included here for heuristic reasons. The disorder is characterized by light red macules of various sizes.…”

Section: Figurementioning

confidence: 99%

Capillary malformations: a classification using specific names for specific skin disorders

Happle

2015

Acad Dermatol Venereol

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“…Unilateral Nevoid Telangiectasia Syndrome (UNTS) is a rare vascular dermatosis characterized by linearly arranged telangiectasias in a unilateral, dermatomal, or blaschkoid pattern (1)(2)(3). UNTS can be congenital or acquired.…”

Section: Introductionmentioning

confidence: 99%

“…UNTS can be congenital or acquired. Many cases of UNTS have been described in association with pregnancy, puberty, liver disease (including alcohol abuse, HBV infection, and HCV infection), and the use of hormonal contraceptive medication (1,2). The disease is noted more often in women during hyperestrogenic states (4).…”

Section: Introductionmentioning

confidence: 99%

Unilateral nevoid telangiectasia syndrome of pregnancy

Zanniello¹

2014

CDERM

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SummaryUnilateral Nevoid Telangiectasia Syndrome (UNTS) is a rare vascular dermatosis characterized by linearly arranged telangiectasias in a unilateral, dermatomal, or blaschkoid pattern that can be congenital or acquired. We report on a 36-year-old Caucasian female who presented UNTS localized in the supraclavicular dermatomal region. The lesions were present since puberty, but suddenly worsened during pregnancy, and completely resolved after delivery. Histological examination revealed ectatic blood vessels in the dermis. Immunochemistry showed the expression of estrogen receptors limited to perivascular cells of ectatic vessels but not on endothelial cells. UNTS associated with pregnancy may be due to excessive estrogenic stimulation of pericytes, which in turn allow vasodilatation. This may also explain the spontaneous resolution of the lesions following delivery.

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“…The notion of an underlying mutation that results in a localized increase Wilkin et al [ 9 ] established a classifi cation system that divided UNT into a congenital and an acquired form, with further subclassifi cation of the latter into variants linked to "states of physiologic estrogen increase" and those that are "liver-related". In 2013, Tanglertsampan et al [ 10 ] , proposed a new classifi cation system that differentiated acquired "estrogen-related" forms from "liver-related", "skin-related", or "other" types (those without an association to any specifi c conditions). In 2015, Happle [ 2 ] argued in favor of distinguishing the punctate from the patchy type.…”

mentioning

confidence: 99%