10.5826/dpc.1301a71
text/xml
2160-9381
2569
en
Mario Linares Barrios
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
tamoxifen
Acquired Bilateral Nevoid Telangiectasia Induced by Tamoxifen
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2023-04-29
2022-04-30
2023-04-29
2023-06-05
e2023071
Dermatol Pract Concept
Mario Linares Barrios
2023/04/29
10.5826/dpc.1301a71
e2023071
Unidad de Gestión Clínica de Dermatología Médico-Quirúrgica y Venereología, Hospital Universitario Puerta del Mar, Cádiz, España
2160-9381
tamoxifen
en
e2023071
Rodríguez-Martín M, Sáez M, Carnerero A, et al. Unilateral naevoid telangiectasia in a young man after chemotherapy: a simple coincidence or a new clinical association? J Eur Acad Dermatology Venereol. 2006;20(8):1001-1002. DOI: 10.1111/j.1468-3083.2006.01593.x. PMID: 16922953.
Acquired Bilateral Nevoid Telangiectasia Induced by Tamoxifen
https://dpcj.org/index.php/dpc/article/download/2569/2059
               Acquired Bilateral Nevoid Telangiectasia
                        Induced by Tamoxifen
              Irene Navarro-Navarro1, Alejandro Ortiz-Prieto1, Gonzalo Gallo-Pineda1,
              Myriam Viedma-Martínez1, David Jiménez-Gallo1, Mario Linares-Barrios1


1 Unidad de Gestión Clínica de Dermatología Médico-Quirúrgica y Venereología, Hospital Universitario Puerta del Mar, Cádiz, España



Key words: bilateral nevoid telangiectasia, tamoxifen
Citation: Navarro-Navarro I, Ortiz-Prieto A, Gallo-Pineda G, Viedma-Martínez M, Jiménez-Gallo D, Linares-Barrios M. Acquired Bilateral
Nevoid Telangiectasia Induced by Tamoxifen. Dermatol Pract Concept. 2023;13(2):e2023071. DOI: https://doi.org/10.5826/dpc.1302a71
Accepted: July 5, 2023; Published: April 2023
Copyright: ©2023 Navarro-Navarro et al. This is an open-access article distributed under the terms of the Creative Commons
Attribution-NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted
noncommercial use, distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding author: Irene Navarro Navarro, MD, Hospital Universitario Puerta del Mar. Unidad de Gestión Clínica de Dermatología,
2ª planta. Avenida Ana de Viya 21, 11009, Cádiz, Spain Phone: +34629924839 E-mail: irenen.navarro@gmail.com




Introduction                                                             radiotherapy and starting tamoxifen treatment. Physical ex-
                                                                         amination showed symmetric purpuric linear macules on the
Unilateral nevoid telangiectasia (UNT) is a superficial tel-             inner arms, chest and upper back (Figure 1). The macules
angiectasia of the upper body characterized by showing a                 had a dermatomal distribution following the T1 dermatome
unilateral dermatomal distribution, typically affecting der-             (Figure 2). She was interrogated for extracutaneous mani-
matomes from C3 to T3 [1]. Although the exact cause is un-               festations such as digestive bleeding or epistaxis, without
known, the association with hyperestrogenic states has been              reporting any systemic symptoms. A skin biopsy showed
described [2]. Bilateral nevoid telangiectasia (BNT) is a rare           dilated blood vessels in the upper dermis with mild perivas-
variant of UNT affecting both sides of the body. We present              cular inflammation and normal number of mastocyte cells.
a case of BNT in relation with tamoxifen treatment.                      Immunohistochemistry showed positivity to estrogen recep-
                                                                         tors in fibroblastic cells. Blood tests including biochemistry,
Case Presentation                                                        blood count, coagulation study, estrogen and progesterone
                                                                         levels and viral hepatitis serology were normal or negative.
A 41-year-old female attended our dermatology department                 Stool blood test was negative. She was diagnosed with BNT
due to the appearance of asymptomatic cutaneous lesions on               secondary to tamoxifen.
the upper trunk and arms. She had a history of ductal cancer
of the right breast stage IA with positivity for estrogen and
                                                                         Conclusions
progesterone receptors, treated with surgery, radiotherapy
and tamoxifen. She did not have any other drug history. The              BNT, although bilateral in its presentation, is analogous
cutaneous lesions had appeared several weeks after finishing             to UNT in that it follows the characteristic dermatomal




Research Letter | Dermatol Pract Concept. 2023;13(2):e2023071                                                                         1
Figure 1. Clinical image of bilateral nevoid telangiectasia. Symmet-
                                                                       Figure 2. Clinical image of bilateral nevoid telangiectasia. Linear
ric purpuric linear macules on the arms and chest following the T1
                                                                       purpuric – brownish macules following the T1 dermatome.
dermatome.



distribution of the telangiectasias. However, this variant             also for BNT. However, more studies directed to explain the
seems to be less frequent. Over a hundred cases of UNT                 underlying mechanisms causing UNT and BNT are needed
have been described in the literature, and only twenty-four            to validate our observations.
cases of BNT [3]. Although both entities are similar in their
presentation, some epidemiological differences have been               References
described between the two variants. BNT is more frequent
                                                                        1. Duong MH, Raymond CP. Unilateral dermatomal superficial tel-
in male patients whereas UNT occurs predominantly in                       angiectasia. Can Med Assoc J. 1983;129(10):1117-1118. PMID:
women. Patients with BNT are usually older than those with                 6627172.PMCID: PMC1875470.
UNT, with a later onset of presentation, and higher associa-            2. Uhlin SR, McCarty KS Jr. Unilateral nevoid telangiectatic
tion with underlying diseases such as liver diseases, diabetes             syndrome. The role of estrogen and progesterone receptors.
                                                                           Arch Dermatol. 1983;119(3):226-228. DOI: 10.1001/arch-
and smoking [4].
                                                                           derm.119.3.226. PMID: 6824361.
    The association of UNT with hyperestrogenic states has
                                                                        3. Jee H, Kim TG, Kim DS, Kim DY, Lee MG. Acquired bilateral
been proposed, owing to the high prevalence in patients                    nevoid telangiectasia: Report of 9 cases. Eur J Dermatology.
during puberty and pregnancy [5]. Tamoxifen is a selective                 2013;23(5):736-737. DOI: 10.1684/ejd.2013.2141. PMID:
estrogen receptor modulator used in breast cancer with pos-                24211825.
itivity for hormonal receptors. This therapy has an antag-              4. Kim EJ, Park HS, Yoon HS, Kim KH, Cho S. Demographic
                                                                           and clinical differences between unilateral and bilateral forms
onist effect on breast tissue, but an agonist effect on bone,
                                                                           of naevoid telangiectasia: A retrospective study with review of
liver, skin and endometrium. Association between UNT and
                                                                           the literature. Br J Dermatol. 2015;172(6):1651-1653. DOI:
other drugs, such as chemotherapy agents, has also been                    10.1111/bjd.13615. PMID: 25495722.
­proposed [6].                                                          5. Sánchez Conejo-Mir J, Ortega Resina M CMF. Unilateral nev-
    We hypothesize that tamoxifen led to an increase of estro-             oid telangiectasia syndrome. Study of cutaneous estrogen recep-
gen receptors on the dermatomal skin of our patient, which                 tors. Med Cutan Ibero Lat Am. 1984;12(6):469-475. PMID:
                                                                           6397668.
may have been the cause for the presentation of BNT in this
                                                                        6. Rodríguez-Martín M, Sáez M, Carnerero A, et al. Unilat-
case. This is the first case reported, to our knowledge, of nev-
                                                                           eral naevoid telangiectasia in a young man after chemother-
oid telangiectasia (UNT or BNT) in relation with tamoxifen.                apy: a simple coincidence or a new clinical association? J Eur
This contributes to the hypothesis that hyperestrogenic states             Acad Dermatology Venereol. 2006;20(8):1001-1002. DOI:
may be important not only for the pathogenesis of UNT but                  10.1111/j.1468-3083.2006.01593.x. PMID: 16922953.




2                                                                          Research Letter | Dermatol Pract Concept. 2023;13(2):e2023071
