|Year : 2021 | Volume
| Issue : 2 | Page : 113-114
Acquired unilateral nevoid telangiectasia as a novel postherpetic Wolf's isotopic response
Hao Wu, Fang Qiu, Meng-Tong Li, Ying Zhou, Yi-Ming Fan
Department of Dermatology, Affiliated Hospital of Guangdong Medical University, Zhanjiang, Guangdong, China
|Date of Submission||22-Sep-2020|
|Date of Decision||27-Nov-2020|
|Date of Acceptance||27-Dec-2020|
|Date of Web Publication||23-Jun-2021|
Dr. Yi-Ming Fan
Department of Dermatology, Affiliated Hospital of Guangdong Medical University, 57 Southern Renmin Avenue, Xiashan District, Zhanjiang 524001, Guangdong
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Wu H, Qiu F, Li MT, Zhou Y, Fan YM. Acquired unilateral nevoid telangiectasia as a novel postherpetic Wolf's isotopic response. Dermatol Sin 2021;39:113-4
|How to cite this URL:|
Wu H, Qiu F, Li MT, Zhou Y, Fan YM. Acquired unilateral nevoid telangiectasia as a novel postherpetic Wolf's isotopic response. Dermatol Sin [serial online] 2021 [cited 2021 Jul 24];39:113-4. Available from: https://www.dermsinica.org/text.asp?2021/39/2/113/319155
Wolf's isotopic response (WIR) refers to the occurrence of a new dermatosis at the same site of a previously healed, unrelated skin disorder. The most common primary dermatosis is herpes zoster (HZ), while secondary dermatoses are miscellaneous. However, vascular disorders are rarely reported. Only two cases of postherpetic tufted angioma have been documented in English literature., We describe a patient who developed acquired unilateral nevoid telangiectasia (UNT) on C4 dermatome of recently healed HZ.
A 65-year-old postmenopausal woman presented with painful reddish telangiectatic macules on the left side of upper chest, back, and shoulder (C4 dermatome) for 7 days. Fifteen days ago, grouped vesicles with severe neuralgia developed on the same dermatome. She was diagnosed with HZ and treated with oral famciclovir (1 g/d), prednisone (20 mg/d), analgesics, and moxibustion in a local hospital. The telangiectatic macules occurred as soon as the vesicular eruption healed with mild scarring. Past history included well-controlled hypertension. Physical examination revealed numerous, blanching, reddish telangiectatic macules on C4 dermatome with allodynia and several scars [Figure 1]a. There were no skin atrophy, palmar erythema, mucous telangiectasia, and positive Darier's sign. Dermoscopy showed red arborizing and tortuous vessels with a reticulated appearance [Figure 1]b. Laboratory results including full blood count, liver and renal function, coagulation profile, serum sex and thyroid hormones, and hepatitis B and C were unremarkable. Chest radiograph and abdominal ultrasonography showed no pathological findings. H and E and Giemsa stain of lesional biopsy displayed unremarkable epidermis and dilated capillaries with perivascular infiltrate of lymphohistiocytes and mastocytes in the superficial dermis [Figure 2]a. Immunohistochemically, dermal lymphocytes were predominantly positive for CD4 [Figure 2]b, and less for CD8 and CD45RO [Figure 2]c. The mean number of CD117+ mastocytes in the superficial dermis was 7.9/high-power field (HPF;×400) in 10 serial fields [Figure 2]d. These results were consistent with the diagnosis of acquired UNT following HZ. The neuralgia was remarkably alleviated after oral mecobalamin and ibuprofen/codeine sustained tablets, but the telangiectasia remained stable during 1-month follow-up.
|Figure 1: Clinico-dermoscopic images of acquired unilateral nevoid telangiectasia following herpes zoster. (a) Numerous, blanching, reddish telangiectatic macules with some scars on the left side of upper chest, back, and shoulder (C4 dermatome). Note multiple moxibustion-induced scars on the left lateral neck and nucha. (b) Dermoscopy showing red arborizing and tortuous vessels with a reticulated appearance (original magnification, ×30).|
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|Figure 2: Histo- and immunopathological images of acquired unilateral nevoid telangiectasia following herpes zoster. (a) H and E stain revealing dilated capillaries with perivascular lymphohistiocytic infiltrate in the upper dermis (original magnification, ×100). (b-d) Immunohistochemical staining displaying CD4+ (b) and CD45RO+ (c) lymphocytes, and CD117+ mastocytes (d) in the papillary dermis (original magnification, ×200).|
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Our patient developed unilateral C4 dermatomal telangiectasia following acute HZ. The suspected disorders include acquired UNT, telangiectasia macularis eruptiva perstans (TMEP), and serpiginous angioma. UNT is congenital or acquired entity characterized clinically by telangiectatic patches in a unilateral dermatomal or linear distribution, and histopathologically by dermal dilated capillaries with unconspicuous mastocyte infiltrate. UNT commonly affects the trigeminal nerve divisions as well as cervical and upper thoracic segments (C3-T2). Although the exact pathogenesis of UNT remains undefined, the main hypothesis is related to physiological or pathological hyperestrogenemia. Even though some UNT cases are lack of hyperestrogenemia and abnormal liver function, the estrogen receptors of endothelial cells and vascular endothelial growth factor are linked to the pathogenesis of UNT. TMEP is a rare form of cutaneous mastocytosis that typically presents as symmetrically and bilaterally distributed telangiectatic macules with perivascular mastocyte infiltrate in adulthood, while unilateral TMEP is uncommon., Angioma serpiginosum is a rare unilateral vascular condition characterized by pinpoint red maculopapules in linear, serpiginous, or gyrate patterns and predominantly affects the lower limbs in young females. In this case, although the number of dermal mastocytes was mildly increased, it cannot accord the diagnostic criteria (>20 mastocytes/HPF) of TMEP. Meanwhile, CD117+ mastocytes were seen in nine of ten patients with postherpetic WIR. Furthermore, dermoscopic features of red arborizing and tortuous vessels with a reticulated appearance in our patient agree with UNT but not angioma serpiginosum (multiple red oval lagoons). Therefore, acquired UNT could be the preferable diagnosis for the present case.
The pathomechanism of WIR remains unclear, possibly involving neuroimmune instability at viral infection sites. The healed skin of the first disease may leave scar, pigment/color changes, or other minimal changes. Persisting varicella-zoster virus (VZV) infection should be considered in our case because of successive occurrence of secondary UNT. The different ratio of dermal CD4/CD8 lymphocytes may be owing to various postherpetic WIRs. Mastocyte infiltrate may reflect the local excess immune response in VZV-infected dermatomes. Our literature review, including a paper from Ise's group, suggests that memory CD8+ lymphocytes could contribute to the pathogenesis of postherpetic WIR. In addition, secondary vascular lesions might be caused by local deregulation via both angiogenic and neuroimmunal mechanisms.,
UNT tends to be asymptomatic and generally requires no treatment. Pulsed-dye laser therapy may be feasible for cosmetic reasons, but lesional relapse is common.
To our knowledge, this case may represent the first case of acquired UNT following HZ, which enlarges the spectrum of postherpetic WIR.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initial will not be published and due efforts will be made to conceal her identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2]