Nevus oligemicus is a recently described dermatologic entity, first mentioned by Davies et al.1 in 1981; few cases have been reported to date.

We report 2 cases of women aged 66 and 81 years, identified as patient 1 and patient 2, respectively; the women were evaluated in our department in an interval of approximately 1 year due to the appearance of macules on both breasts. Physical examination revealed erythematous-violaceous telangiectatic macules on the breasts; the macules measured several centimeters and had irregular, poorly defined edges. Palpation caused the macules to turn white and revealed a local temperature that was markedly lower than that of the adjacent healthy tissue, with no signs of arterial ischemia (Fig. 1). The lesions had appeared a year earlier and were asymptomatic. The patients stated that they had not experienced previous local trauma or an infection, or applied topical products. Patient 2 was hypertensive and had osteoporosis and early-stage Alzheimer disease, for which she had been receiving drug therapy for many years. The analyses carried out included a full blood count, biochemistry, coagulation assay, kidney and liver function tests, anticardiolipin antibody assay, antinuclear antibody and cryoglobulin assay, and serology for Borrelia burgdorferi, and all results were normal or negative. A skin biopsy was performed only in patient 2 and showed vascular ectasia in the papillary dermis (Fig. 2). The lesions were diagnosed as nevus oligemicus and remained stable during a follow-up period of approximately 2 years; no new lesions appeared in this time.Nevus oligemicus is rarely reported in the literature; this may be because it is asymptomatic and sometimes not easily noticed, and is therefore underreported by patients or underdiagnosed. The etiology and pathogenesis are unknown, but an abnormality of the adrenergic receptors with increased sympathetic tone in the deep dermal vascular plexus has been suggested. This would lead to reduced deep vascular flow, which is responsible for regulating skin temperature, thereby causing the lesion to be cold to the touch, and relative vasodilation of the superficial vascular plexus, which feeds the tissue, thereby causing the erythema. The term nevus oligemicus may be translated as poorly vascularized nevus, and some authors consider it to be a functional rather than an anatomical disorder.1–3

Figure 1.

Patient 2. Erythematous-violaceous telangiectatic macules, more evident on the right breast.

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Figure 2.

Hematoxylin-eosin, original magnification x100. Vascular ectasia in the papillary dermis.

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The disease manifests clinically as fixed, acquired erythematous-violaceous macules and sometimes in the form of whitish telangiectatic macules,4 with poorly defined, irregular edges. Palpation of the lesions causes them to turn white and, when measured using a thermometer, their temperature is at least 2°C lower than that of adjacent healthy skin. The lesions are usually located on the abdomen and thighs,1,4–6 and less frequently on the breasts7,8 and hands.2 They are asymptomatic and only 1 case of abnormal local heat sensitivity has been reported.2 Although diagnosis is essentially clinical, histology, if performed, shows dilation of the vessels in the papillary dermis and occlusion of the vessels in the reticular dermis, with a normal number of vessels. Results of vasomotor tests are normal in both the uninvolved and involved skin1–3,5 and the finding supports the hypothesis that this is a functional anomaly. The only recognized trigger is prolonged bathing in cold water, which has led to the appearance of nevus oligemicus on the hands.2 Contributing factors such as obesity, a sedentary lifestyle, and proximal pressure (such as that caused by a tight belt) have been suggested for lesions located on the abdomen, but weight loss and removing the pressure failed to resolve the lesions.5 Our patients were overweight, predominantly around the abdomen, and had large, sagging breasts.

The differential diagnosis should include inflammatory erythema (mastitis, cellulitis, erysipelas), which involves warm lesions that tend to resolve, capillary malformations with no changes in local temperature and with characteristic histologic signs, and livedo reticularis, which is characterized by an erythematous-violaceous reticular pattern.5,6,8

In our patients, the lesions remained stable over time, with no changes in size or appearance, and no new lesions developed. Treatment with systemic corticosteroids was ineffective.5

In conclusion, we report 2 cases of nevus oligemicus on the breasts, which is a rare site; the only previously reported case on this site involved a single breast.7 The key to diagnosis is the finding of permanent local hypothermia; it is therefore important to palpate the lesion to detect a reduction of between 2 and 2.5°C in local temperature. We consider that the frequency of this entity is probably greater than indicated by a review of the literature, but that it may go unnoticed or unreported by patients.