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CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 4  |  Page : 423-424
Unilateral nevoid telangiectasia: An overlooked entity


From the Department of D.V.L., Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India

Date of Web Publication2-Nov-2022

Correspondence Address:
Vignesh Nambi Ravi
From the Department of D.V.L., Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_828_21

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How to cite this article:
Sukanya G, Sane RR, Ravi VN, Nehete SS. Unilateral nevoid telangiectasia: An overlooked entity. Indian J Dermatol 2022;67:423-4

How to cite this URL:
Sukanya G, Sane RR, Ravi VN, Nehete SS. Unilateral nevoid telangiectasia: An overlooked entity. Indian J Dermatol [serial online] 2022 [cited 2022 Nov 26];67:423-4. Available from: https://www.e-ijd.org/text.asp?2022/67/4/423/360359




Sir,

Unilateral nevoid telangiectasia (UNT) is a congenital or acquired disorder resulting in abnormal superficial cutaneous vasculature. It is commoner in females.

An 18-year-old female presented with complaints of diffuse red lesions over left side of the body since 1 year. Lesions were gradual in onset, non-progressive persistent and asymptomatic. Patient attained her menarche at 12 years of age. She had irregular menstrual cycles with significant premenstrual syndrome. There was no history of systemic illness. Patient denied history of alcoholism. On examination, multiple, diffuse telangiectatic lesions were distributed unilaterally along the left arm, forearm, palm, trunk and face [Figure 1]. Right side was apparently normal. Heterochromia of irides was noted. Diascopy showed reduced prominence of the telangiectasias. Dermoscopy showed multiple red tortuous capillaries with a reticulated pattern [Figure 2]a and [Figure 2]b. Histopathological examination showed normal epidermis and multiple thin walled dilated capillaries in the upper dermis with extravasations of erythrocytes [Figure 2]c and [Figure 2]d. Patient had hyperestrogenemia with an estradiol level 439.1 pg/mL in luteal phase [Normal: 43.8–211 pg/mL]. Other general and liver parameters were normal. A clinical diagnosis was made with dermoscopic and histopathological confirmation. Heterochromia irides might be considered as coincidental finding in this case. Patient was counselled about the innocuous nature of the condition and was advised pulse dye laser.
Figure 1: Diffuse telangiectatic lesions over left arm (a), back (b), face (c), palm (d)

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Figure 2: (a and b) Dermoscopic images showing multiple red tortuous capillaries (polarised mode, 20 × magnification). (c and d) Histopathology picture showing multiple thin-walled dilated blood capillaries in the upper dermis. (H and E stain, 400× magnification)

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UNT presents more commonly post puberty with telangiectasias distributed along C3–C4 and trigeminal dermatomes unilaterally. Endothelial cells of blood capillaries possess estrogen receptors thus; the effect due to hyperestrogenemia is mainly seen as telangiectasias. One theory proposes the role of somatic mosaicism, which becomes evident in hyperestrogenemia leading to the disorder.[1] The lesions appear unilaterally as estrogen sensitive cells are distributed along the dermatomes congenitally.[2] Its association is also reported in pregnancy, oral contraceptive pills use, chronic alcoholism, hepatitis B and C infection, hyperthyroidism, carcinoid syndrome. The commonest reason for UNT in men is considered to be liver cirrhosis.[3] But UNT was also reported in a male young individual following chemotherapy for Hodgkin's disease without liver dysfunction.[3]

Diagnosis is made clinically with evidence of one of the etiological factors. Complete blood count, liver and thyroid function test, estrogen levels are warranted. Dermoscopy shows multiple tortuous capillaries in reticulated pattern.[4] Histopathological examination shows dilated vessels in upper dermis without any endothelial cell proliferation or angiogenesis. Management mainly includes treating the primary cause of hyperestrogenemia and reassurance for the skin condition. Pulse dye laser can be done for the lesions for cosmetic improvement. Till now a few cases have been reported of UNT.[5] Dadlani reported it in a pregnant lady of 30 years of age.[5] Guedes reported three cases of UNT of which, one was a 8-year-old boy, other was a 33-year-old female. Both had normal estrogen levels. A third case was of a 35-year-old pregnant lady with recurrent episodes of UNT.

UNT is neglected multiple times and is under-reported in literature perhaps due to its asymptomatic character. As it is one of markers of hyperestrogenemia, management should be done to avoid complications. We highlight the use of dermoscopy as a non-invasive tool to clinch the diagnosis due to its distinctive findings.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Happle R. Capillary malformations: A classification using specific names for specific skin disorders. J Eur Acad Dermatol Venereol 2015;29:2295-305.  Back to cited text no. 1
    
2.
Jordão JM, Haendchen LC, Berestinas TC, Faucz LR. Acquired unilateral nevoid telangiectasia in a healthy men. An Bras Dermatol 2010;85:912-4.  Back to cited text no. 2
    
3.
Rodríguez-Martín M, Saez M, Carnerero A, García-Bustínduy M, Guimerá F, Rodríguez García F, et al. Unilateral naevoid telangiectasia in a young man after chemotherapy: A simple coincidence or a new clinical association?. J Eur Acad Dermatol Venereol 2006;20:1001-2.  Back to cited text no. 3
    
4.
Villela-Segura U. Dermoscopy as an important tool for differentiating unilateral nevoid telangiectasia and angioma serpiginosum. Dermatol Pract Concept 2019;9:306-7.  Back to cited text no. 4
    
5.
Dadlani C, Kamino H, Walters RF, Rosenman K, Pomeranz MK. Unilateral nevoid telangiectasia. Dermatol Online J 2008;14:3.  Back to cited text no. 5
    


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