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J Clin Aesthet Dermatol 2020 Jan 1;13(1):38-40. Epub 2020 Jan 1.

All authors are with the Department of Dermatology and Cutaneous Surgery at the University of Miami, Miller School of Medicine in Miami, Florida.

Oral mucosal involvement in patients with morphea is quite rare. Unilateral nevoid telangiectasia is a rare vascular dermatosis characterized by unilateral telangiectasia distributed in a dermatomal pattern, especially on the upper trunk and extremities. We describe a case of a 10-year-old female patient that presents with morphea on her left chin and upper lip extending to the inner oral mucosa as well as a unilateral nevoid telangiectasia on her left cheek as an early presentation of localized scleroderma. Read More

Dermatol Pract Concept 2019 Oct 31;9(4):306-307. Epub 2019 Oct 31.

Dermatology Department, Hospital Regional Licenciado Adolfo López Mateos ISSSTE, Mexico City, Mexico.

Chin Med J (Engl) 2019 Sep;132(17):2125-2126

Department of Dermatology, Union Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, Hubei 430022, China.

An Bras Dermatol 2019 Mar-Apr;94(2):211-213. Epub 2019 May 9.

Electron Microscopy Center of the South, Fundação Universidade de Rio Grande, Rio Grande (RS), Brazil.

Cutaneous collagenous vasculopathy is a rare acquired idiopathic microangiopathy characterized by progressive development of diffuse asymptomatic telangiectasias and histologically by accumulation of collagen type IV around the affected vessels. It is diagnosed by its clinical history, confirmed by light microscopy with collagen-specific immunostaining. We report a case of a patient with extensive acquired telangiectasias on the left arm, clinically resembling unilateral nevoid telangiectasia. Read More

J Dtsch Dermatol Ges 2019 04;17(4):459-460

Dermatol Online J 2017 Aug 15;23(8). Epub 2017 Aug 15.

Department of Dermatology, Dermatologist MD PhD, Complejo Hospitalario de Granada, Granada, Spain.

Essential generalized telangiectasia as the result of postcapillary venule dilatation, is characterized by the sudden development of generalized telangiectasias, sometimes involving the conjunctiva. A few pediatric cases have been reported. The main condition in the differential diagnosis includes syndromes with telangiectasia such as hereditary hemorrhagic telangiectasia of Rendu-Osler, unilateral nevoid telangiectasia, and neonatal lupus erythematous. Read More

J Dtsch Dermatol Ges 2017 Jun 17;15(6):660-662. Epub 2017 May 17.

Department of Dermatology, Heidelberg University Hospital, Heidelberg.

Eur J Dermatol 2017 08;27(4):439-441

Institute of Dermatology, Fondazione IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy.

Ann Dermatol 2016 Jun 25;28(3):404-5. Epub 2016 May 25.

Department of Dermatology, Soon Chun Hyang University Bucheon Hospital, Bucheon, Korea.

An Bras Dermatol 2015 Sep-Oct;90(5):769-70

Centro Hospitalar e Universitário de Coimbra, Coimbra, Portugal.

Turk J Pediatr 2015 Nov-Dec;57(6):635-638

Division of Pediatric Oncology, Department of Pediatrics, Yıldırım Beyazıt University, Faculty of Medicine, Ankara, Turkey.

Unilateral nevoid telangiectasia (UNT) is characterized by the dermatomal distribution of telangiectasia, which are distributed unilaterally and frequently found in C3-C4 or the trigeminal dermatomal areas. It may be congenital or acquired and has a 2:1 female: male ratio. Several theories where developed in order to explain its etiopathogenesis. Read More

Dermatol Online J 2014 Jun 15;20(6). Epub 2014 Jun 15.

University of California, Davis.

Unilateral Nevoid Telangectasia Syndrome (UNTS) is characterized by superficial telangiectasias in a unilateral distribution. Vascular endothelial growth factor (VEGF) may play a role in the pathogenesis of UNTS in patients with underlying hepatic disease. We report a case of a patient with UNTS accompanied by chronic hepatitis C virus infection, with a normal serum estrogen profile and strong positive immunohistochemical staining of lesional skin with VEGF. Read More

J R Coll Physicians Edinb 2013 ;43(3):222-4

JJ Park, University of Edinburgh Medical School, The Chancellor's Building, The University of Edinburgh College of Medicine and Veterinary Medicine 49 Little France Crescent, Edinburgh EH16 4SB, UK.

A 55-year-old Caucasian man presented with unilateral right facial spider nevi. Relevant medical history included a right-sided cerebrovascular accident (CVA) in 1997 and decompensated alcoholic liver disease, diagnosed in 2007. The literature describes approximately 100 cases of unilateral spider nevi associated with different aetiologies, grouped under 'unilateral nevoid telangiectasia syndrome' (UNTS). Read More

Acta Dermatovenerol Croat 2013 ;21(2):133-4

An Bras Dermatol 2013 Mar-Apr;88(2):299-301

Dermatology Service, Santo Antonio dos Capuchos Hospital, Central Lisbon Hospital Center, Lisbon, Portugal.

A 27 year-old-man, with no known personal or familial history of disease, mentioned a 10-year history of asymptomatic groups of telangiectasias, with a Blaschko lines distribution on the right lateral aspect of the neck and asymptomatic. He denied any episodes of disease or drug intake that could be associated with the disease. Blood work had no changes, namely of liver enzymes or infectious serologies. Read More

Int J Dermatol 2013 May;52(5):608-10

Department of Dermatology, School of Anti-Aging and Regenerative Medicine, Mae Fah Luang University Hospital, Bangkok, Thailand.

Int J Dermatol 2014 Jan 3;53(1):e32-3. Epub 2013 Mar 3.

Department of Dermatology, Centro Hospitalar do Porto - Hospital de Santo António, Edifício das Consultas Externas (Ex-CICAP), Porto, Portugal.

Indian J Dermatol 2012 Mar;57(2):138-40

Dermatology Department, Centro Hospitalar de Vila Nova de Gaia, Vila Nova de Gaia, Portugal.

Unilateral nevoid telangiectasia is a rare congenital or acquired disorder. The majority of the authors propose that it is probably related to elevated estrogen receptors, although other authors advocate different theories. We report the occurrence of lesions clinically compatible with this diagnosis in a child and two adults. Read More

Ann Dermatol 2012 May 26;24(2):206-8. Epub 2012 Apr 26.

Department of Dermatology and Cutaneous Biology Research Institute, Yonsei University College of Medicine, Seoul, Korea.

Melorheostosis is a rare disorder characterized by irregular, flowing hyperostosis in long bones, commonly described on radiographs as wax flowing down a candle. In addition to bony sclerosis, cutaneous manifestations overlying the involved bones have been reported including linear scleroderma, neurofibromatosis, and vascular and lymphatic malformations. Unilateral nevoid telangiectasia (UNT) is a rare primarily cutaneous condition characterized by linearly arranged small dilated blood vessels in dermatomal or Blaschkoid patterns on the skin. Read More

J Dtsch Dermatol Ges 2012 Feb;10(2):140; author reply 141

J Eur Acad Dermatol Venereol 2011 Nov 17;25(11):1356-9. Epub 2011 Jan 17.

Department of Dermatology and Venerology, Akdeniz University, School of Medicine Antalya, Turkey.

Background: Unilateral nevoid telangiectasia (UNT) is a unique vascular dermatosis of ambiguous aetiology.

Objective: To investigate the role of neurological disorder in pathogenesis of the UNT.

Methods: We investigated eight consecutive patients with unilateral nevoid telangiectasia. Read More

Dermatol Online J 2011 May 15;17(5). Epub 2011 May 15.

Department of Pathology, Beth Israel Deaconess Medical Center and Harvard Medical School, Boston, MA, USA.

We report on a 43-year-old Caucasian female who presented with bright red macules in a unilateral distribution in the left C5-8 and L3-5 dermatomes. Histopathologic examination showed superficial papillary dermal telangiectasia with minimal chronic inflammation. Immunohistochemical stains for estrogen and progesterone receptors (ER/PR) were negative. Read More

An Bras Dermatol 2010 Nov-Dec;85(6):912-4

Evangelical University Hospital of Curitiba, Evangelical Faculty of Parana, Curitiba, PR, Brasil.

Unilateral nevoid telangiectasia is a rare vascular dermatosis, characterized by areas of superficial telangiectases distributed in a linear unilateral pattern. It was described in 1899 by Zeisler and Blascko. Several theories where developed in order to explain its etiopathogenesis. Read More

J Dtsch Dermatol Ges 2011 May 13;9(5):389-90. Epub 2010 Dec 13.

Arch Dermatol 2010 Oct;146(10):1167-72

Department of Dermatology, University of Wisconsin, Madison, USA.

J Am Acad Dermatol 2010 Mar;62(3):528-30

J Mal Vasc 2009 Sep 10;34(4):272-4. Epub 2009 Jun 10.

Clinique de médecine vasculaire, Service de dermatologie, CHU Michallon, BP 217, 38043 Grenoble cedex 09, France.

Patients presenting skin disorders with a probably vascular origin are often addressed to the vascular medicine unit. In general, the diagnosis of telangiectasia is straightforward but in certain rare clinical forms may be misleading. We report the case of a female patient presenting unilateral circumscribed telangiectasia on the left arm. Read More

Actas Dermosifiliogr 2009 Jan-Feb;100(1):82-3

Servicio de Dermatología, Hospital Clínico San Cecilio, Granada, España.

Int J Dermatol 2008 Dec;47(12):1331-3

Dermatol Online J 2008 Oct 15;14(10). Epub 2008 Oct 15.

Department of Dermatology, New York University, USA.

A 30-year-old woman, who was six months pregnant, presented with multiple, blanching, asymptomatic telangiectasies on her right upper extremity for two years. At the onset of her pregnancy, her lesions increased in number and redness. Given the unilateral distribution and worsening during pregnancy, a diagnosis of unilateral nevoid telangiectasia was made. Read More

Yonsei Med J 2008 Jun;49(3):509-13

Department of Dermatology, Taipei Municipal Wan-Fang Hospital, Taipei Medical University, Taipei, Taiwan.

Angioma serpiginosum is an uncommon, acquired vascular nevoid disorder with capillary dilation and proliferation in the papillary dermis. The eruptions are asymptomatic and characterized by grouped, erythematous to violaceous, serpiginous and punctate macules. The condition usually appears in females during adolescence on unilateral lower extremities and the buttocks. Read More

Indian J Dermatol Venereol Leprol 2008 Mar-Apr;74(2):163-4

J Dermatol 2006 Nov;33(11):825-6

Int J Dermatol 2006 Aug;45(8):960-4

Department of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India.

Background: Unilateral nevoid telangiectasia (UNT) is a unique vascular dermatosis of ambiguous etiology. The therapeutic role of pulsed dye laser in this condition, especially in individuals with skin types III and IV, has not been elucidated completely. The aim of this study was to assess the response to flash-lamp pulsed dye laser of UNT in Indian patients. Read More

Dermatology 2004 ;209(3):215-7

Department of Dermatology and Venereology, Martin Luther University Halle-Wittenberg, Halle/Saale, Germany.

We report on a 29-year-old patient with a unilateral nevoid telangiectasia syndrome (UNTS). Based on microcirculation examinations, both functional impairments at the blood vessel level (laser-Doppler flowmetry) and morphologically altered capillaries (capillary microscopy) could be observed. We could demonstrate that microcirculatory examinations are suitable to demonstrate pathophysiological defects at the vascular level, especially since subclinical functional defects can also be recorded. Read More

J Dermatol 2004 May;31(5):411-4

Abant Izzet Baysal University, Duzce Medical School, Department of Dermatology, Duzce, Turkey.

Telangiectasia and spider angioma were distributed in a unilateral, dermatomal pattern in a female patient. Physical examination and laboratory studies revealed hyperthyroidism. The possible effects of thyroid hormones on formation of vascular nevi were considered to be noteworthy. Read More

J Dermatol 2004 Feb;31(2):109-12

Department of Dermatology, Cukurova University, School of Medicine, Adana, Turkey.

Unilateral nevoid telangiectasia is a cutaneous condition consisting of congenital or acquired patches of superficial telangiectases in a unilateral linear distribution. Unilateral nevoid telangiectasia has been associated with elevations of blood estrogen levels and/or an increased number of estrogen receptors in the involved skin. We present a hepatitis-B carrier case with unilateral nevoid telangiectasia on the face and the right side of the neck; she had normal blood estrogen and a normal number of estrogen receptors in the involved skin. Read More

J Dermatol 2001 Aug;28(8):453-4

Vasa 2000 Aug;29(3):225-8

Dermatologische Klinik, Universitätsspital Zürich, Schweiz.

We report the development of an acquired nevus flammeus following a ski accident. This disorder was first described by Fegeler 1949, who reported the case of a 43-year-old soldier who acquired a nevus flammeus in the face following a cranial trauma. Since then, a number of similar case reports have been published. Read More

J Am Acad Dermatol 1998 Jul;39(1):138-9

J Am Acad Dermatol 1997 May;36(5 Pt 2):819-22

Division of Dermatology and Cutaneous Surgery, University of South Florida, Tampa 33612, USA.

Unilateral nevoid telangiectasia, a rarely reported disorder, has been said to be related to increased estrogen receptors in involved skin. The lesions may be congenital or acquired. When acquired it arises almost exclusively during periods of relatively increased estrogen levels such as during pregnancy or puberty, or in association with alcoholic cirrhosis. Read More

Ann Dermatol Venereol 1997 ;124(5):407-8

Service de Dermatologie, Hôpital Robert Debré, Reims.

Acta Derm Venereol 1997 Jan;77(1):62-3

Department of Dermatology and Allergy, Gülhane Military Medical Academy, Haydarpaşa Teaching Hospital, Istanbul, Turkey.

We describe a man with acquired unilateral nevoid telangiectasia syndrome, in whom no underlying disease, alcohol abuse or physiological conditions causing hormonal changes are demonstrable. To our knowledge, this is the first case of acquired unilateral nevoid telangiectasia syndrome seen in a healthy adult male and not associated with a hyperestrogenaemic state and estrogen receptor abnormality. This case report casts doubt on the commonly held view that unilateral nevoid telangiectasia syndrome is an estrogen-sensitive nevoid anomaly. Read More

Cutis 1994 Jan;53(1):53-4

Division of Dermatology, Georgetown University Medical Center, Washington, DC 20007.

Unilateral nevoid telangiectasia syndrome can be classified into two major categories: congenital and acquired. There have been reports showing an increase in skin estrogen and progesterone receptors in patients with this syndrome. We report a case of acquired unilateral nevoid telangiectasia syndrome related to pregnancy with no estrogen and progesterone receptors. Read More

Vasa 1993 ;22(1):82-5

Dermatologische Klinik, Universitätsspital Zürich.

A case of unilateral spider telangiectasia on the left side of the face initiating at puberty is reported. With respect to its therapy and prognosis this alteration must be distinguished from other telangiectasias. With unilateral nevoid telangiectasia it shares the unilateral distribution of the lesions and the predilection for the female sex during hormonal instability periods. Read More

G Ital Dermatol Venereol 1988 Sep;123(9):448

G Ital Dermatol Venereol 1987 Oct;122(10):519-22

Hautarzt 1985 Feb;36(2):107-8

A case of unilateral nevoid teleangiectatic syndrome (UNTS) occurring during pregnancy is described. Biopsy specimens of the normal skin and of the UNTS were investigated histologically. Read More

Med Cutan Ibero Lat Am 1984 ;12(6):469-75

We present four observations of unilateral nevoid telangiectasia syndrome, pointing out its common cervicothoracic distribution and its relation with hyper-estrogenic conditions, both physiological (puberty, pregnancy, use of contraceptive pills), and pathologic (post-alcoholic hepatic cirrhosis). We also comment on its differential diagnosis with similar diseases. A study of estrogenic receptors in the affected skin was carried out, thus supplying further data for the ethiopathogenesis of the process. Read More

Arch Dermatol 1983 Mar;119(3):226-8