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E-IJD® - CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 5  |  Page : 628
Bilateral nevoid telangiectasia: A case report and review of the literature


1 Dermatology and Venereology, Faculty of Medicine, Akdeniz University, Antalya, Turkey
2 Department of Pathology, Faculty of Medicine, Akdeniz University, Antalya, Turkey

Date of Web Publication29-Dec-2022

Correspondence Address:
Ahmet Demir
Dermatology and Venereology, Faculty of Medicine, Akdeniz University, Antalya
Turkey
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_12_22

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How to cite this article:
Demir A, Bilgic A, Bassorgun CI, Alpsoy E. Bilateral nevoid telangiectasia: A case report and review of the literature. Indian J Dermatol 2022;67:628

How to cite this URL:
Demir A, Bilgic A, Bassorgun CI, Alpsoy E. Bilateral nevoid telangiectasia: A case report and review of the literature. Indian J Dermatol [serial online] 2022 [cited 2023 Feb 8];67:628. Available from: https://www.e-ijd.org/text.asp?2022/67/5/628/366101




Sir,

A 49-year-old male patient presented to our outpatient clinic with a widespread rash on the back which began 6 months ago. The patient did not have any complaints like pruritus, pain or dysesthesia. He had no history of cardiovascular, rheumatological, hematological disorders or any liver disease. There was no family history of similar symptoms. Physical examination revealed erythematous macules and patches widespread on the back between T1-L4 dermatome in which superficial telangiectasias were observed in an asymmetrical distribution. [Figure 1] and [Figure 2]. In his dermoscopic examination, no additional features were observed except for telangiectasias. His detailed laboratory workup (complete blood count, comprehensive metabolic panel, thyroid function tests, testosterone, estrogen, luteinising hormone, follicle-stimulating hormone, and complement levels) were within the normal range. Screening tests (ANA, ANCA, RF) for rheumatological diseases and the serology for hepatitis and HIV were negative. A histopathological study of multiple biopsies taken from the lesions revealed multiple capillaries with increased diameter in the dermis [Figure 3]. He was diagnosed with bilateral nevoid telangiectasia (BNT) with clinical and histopathological findings.
Figure 1: Telangiectatic macules on the back in the bilateral and symmetric distribution

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Figure 2: Close view of telangiectatic macules and patches

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Figure 3: Dilated vessels in dermis (H&E×100)

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Before diagnosing BNT, hereditary haemorrhagic telangiectasia and generalised essential telangiectasia were excluded by the absence of epistaxis and family history, and the distribution and morphological features of the lesions. In all diseases mentioned above, histopathology is characterised by many thin-walled dilated vessels in the superficial dermis.[1] Furthermore, telangiectasia macularis eruptiva was excluded with the histopathology in which mast cells stained with toluidine blue were absent.

Intravascular lymphoma (IVL) is a rare and aggressive subtype of extranodal non-Hodgkin lymphoma. Selective growth of lymphoma cells is observed in the lumen of capillaries and small vessels. In approximately 40% of IVL cases, the skin lesions are tender and indurated, they can range from erythematous patches to nodules, usually distributed over the trunk and extremities which can be accompanied by edema. Due to the diversity in skin findings, many diseases are included in the differential diagnosis of IVL. Superficial telangiectasias covering indurated plaques have also been described in IVL.[2] In this respect, BNT can be included in the differential diagnosis with IVL. In our case, the patient's lesions were not infiltrated, and he did not have any symptoms related to a tumor burden. In addition, IVL is more common in older age (with mean age of 70), and no intravascular lymphoma cells were observed in our histopathological work up.[2]

Unilateral nevoid telangiectasia (UNT) is superficial telangiectasias observed in unilateral dermatomal localisation, usually in the upper part of the trunk and typically in the region of the C3-T4 dermatomes. The exact pathogenesis is unknown. However, it is assumed to be associated with increased estrogen levels, as it has a higher prevalence in puberty, pregnancy, and liver disease. Furthermore, there is an increased number of estrogen receptors in the dermatomal area where telangiectasias are located. However, since serum estrogen values were not studied in non-pregnant UNT cases, the relationship between estrogen and UNT has not been fully demonstrated.[1]

BNT is an extremely rare variant of UNT with a bilateral dermatomal distribution.[1] More than 100 cases of nevoid telangiectasia have been described so far and only a few of them had BNT.[3] In 2004, Tang et al.[4] reported the first case of BNT in a pregnant woman with gastrointestinal involvement, which completely regressed after delivery. Turan et al.[5] then reported a similar case in a male patient associated with gastrointestinal involvement, without a known cause. In the case series of Jee et al., two cases out of nine had liver disease and two others were in the postmenopausal period.[1] In 2020, Huang et al. reported a case of BNT with bier spots.[3] In our case, no specific etiological factor has been identified. [Table 1] summarises the general characteristics of previously reported BNT cases together with the current case.
Table 1: The patients' characteristics, underlying co-morbidities at the time of presentation

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Of the total 12 BNT cases reported in the literature, seven cases were male and five were female. The age distribution was between 27 and 65 years. Only four of the cases had an underlying pathological condition. Eleven out of the 12 cases had spider shape telangiectasias. Our patient's demographic and clinical characteristics were generally similar to those in the literature. The telangiectasias of all reported cases were located between C2 and T12 dermatomes. However, in our case, the telangiectasias were observed between T1-L4 dermatomes. This is the first reported case in which telangiectasias were also observed in the lumbosacral dermatomal area.

Although the number of BNT reported in the literature is extremely limited, it should be considered in the differential diagnosis of cases with specific clinical findings as described above and possible underlying diseases should be investigated.

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.



 
   References Top

1.
Jee H, Kim TG, Kim DS, Kim DY, Lee MG. Acquired bilateral nevoid telangiectasia: Report of 9 cases. Eur J Dermatol 2013;23:736-7.  Back to cited text no. 1
    
2.
Vásquez J, Romero V, Vilas P, Serra-Rexach JA, Vidán MT. Progressive edemas and generalized telangiectasia: A presentation of Intravascular B-cell Lymphoma. Clin Case Rep 2019;7:2429-32.  Back to cited text no. 2
    
3.
Huang Y, Zhang L, Li Z, Bi M. A case of Bier spots with bilateral nevoid telangiectasia. Indian J Dermatol Venereol Leprol 2020;86:400-3.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Tang SJ, Faughnan ME, Marcon NE. Bilateral nevoid telangiectasia syndrome. Gastrointest Endosc 2004;60:468-71.  Back to cited text no. 4
    
5.
Turan A, Saricaoglu H, Bulbul Baskan E, Keskin M, Balaban Adim S, Turan H, et al. Acquired bilateral nevoid telangiectasia syndrome with gastrointestinal involvement. Eur J Dermatol 2011;21:629-30.  Back to cited text no. 5
    


    Figures

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    Tables

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