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unilateral nevoid telangiectasia ( unt ) is a rare , usually acquired condition described for the first time by blaschko in 1899 and counted among the primary telangiectases .
the exact pathogenesis still remains unknown although some authors believe in a strong association with a hyperestrogenic state .
endothelial cells possess estrogen receptors , and estrogens are felt to play a role in angiogenesis .
indeed unt is commonly reported in association with puberty , pregnancy , the use of oral contraceptives , hormonal therapy , chronic alcoholism with or without cirrhosis , chronic liver diseases , hepatitis b and c infections , portal hypertension , carcinoid syndrome with liver metastases , and hyperthyroidism .
however , there have been reported cases in otherwise healthy individuals , especially male patients .
independently of the underlying mechanism , there seems to be a somatic mosaicism , which becomes obvious in environments with excess of the feminine hormone [ figure 1 ] .
other theories claim that hyperestrogenemic states or estrogen receptor abnormalities may not play a major role in the pathogenesis of unt , especially in adult male patients .
these theories propose hemodynamic disturbances , neural alterations , angiogenic factors , and aberrations in perivascular supportive connective tissue as causative agents .
a 9-year - old boy presented with a 1-year history of multiple , scattered , asymptomatic red spots , distributed in a linear and unilateral pattern that involved his right upper extremity ( c6 , c7 , and c8 dermatomes ) . on closer inspection , blanchable telangiectases were evident surrounded by a pale ring [ figure 2 ] .
laboratory results revealed no abnormalities in complete blood cell count , serum chemistry panel and liver function test .
testosterone and estrogen levels were within normal limits and antibodies to hepatitis a , b , and c were absent .
characteristic anemic halo ( red ring ) a 33-year - old man presented with an erythematosus eruption with months of evolution . on physical examination , there were multiple , blanching , scattered , discrete 1 - 3 mm telangiectases with a peripheral anemic halo involving the right lateral inferior portion of his neck , his right shoulder , and right chest [ figure 3 ] .
the rest of the skin , scalp , genitalia , nails , and mucous membranes were clear of lesions , and there was no associated physiological or pathological condition .
he denied any alcohol consumption , and laboratory tests were normal ( chemistry , hormone , and infectious panel ) .
a skin biopsy was performed , which revealed thin wall dilated capillaries in the papillary and reticular dermis .
telangiectases distributed along c4 , c5 , c6 , and t1 dermatomes a 35-year - old woman was referred to dermatology with telangiectatic , scattered , pinpoint , erythematosus , and smooth papules over her anterior left chest [ figure 4 ] .
she had delivered a child 2 weeks before and reported that the lesions began during the last 6 months of her pregnancy .
she had another 4-year - old child and referred similar cutaneous findings during the last months of her previous pregnancy , which disappeared a few months after delivery .
a 9-year - old boy presented with a 1-year history of multiple , scattered , asymptomatic red spots , distributed in a linear and unilateral pattern that involved his right upper extremity ( c6 , c7 , and c8 dermatomes ) . on closer inspection , blanchable telangiectases were evident surrounded by a pale ring [ figure 2 ] .
laboratory results revealed no abnormalities in complete blood cell count , serum chemistry panel and liver function test .
testosterone and estrogen levels were within normal limits and antibodies to hepatitis a , b , and c were absent .
characteristic anemic halo ( red ring ) a 33-year - old man presented with an erythematosus eruption with months of evolution . on physical examination , there were multiple , blanching , scattered , discrete 1 - 3 mm telangiectases with a peripheral anemic halo involving the right lateral inferior portion of his neck , his right shoulder , and right chest [ figure 3 ] .
the rest of the skin , scalp , genitalia , nails , and mucous membranes were clear of lesions , and there was no associated physiological or pathological condition .
he denied any alcohol consumption , and laboratory tests were normal ( chemistry , hormone , and infectious panel ) .
a skin biopsy was performed , which revealed thin wall dilated capillaries in the papillary and reticular dermis .
telangiectases distributed along c4 , c5 , c6 , and t1 dermatomes a 35-year - old woman was referred to dermatology with telangiectatic , scattered , pinpoint , erythematosus , and smooth papules over her anterior left chest [ figure 4 ] .
she had delivered a child 2 weeks before and reported that the lesions began during the last 6 months of her pregnancy .
she had another 4-year - old child and referred similar cutaneous findings during the last months of her previous pregnancy , which disappeared a few months after delivery .
a 9-year - old boy presented with a 1-year history of multiple , scattered , asymptomatic red spots , distributed in a linear and unilateral pattern that involved his right upper extremity ( c6 , c7 , and c8 dermatomes ) . on closer inspection , blanchable telangiectases were evident surrounded by a pale ring [ figure 2 ] .
laboratory results revealed no abnormalities in complete blood cell count , serum chemistry panel and liver function test .
testosterone and estrogen levels were within normal limits and antibodies to hepatitis a , b , and c were absent .
a 33-year - old man presented with an erythematosus eruption with months of evolution . on physical examination , there were multiple , blanching , scattered , discrete 1 - 3 mm telangiectases with a peripheral anemic halo involving the right lateral inferior portion of his neck , his right shoulder , and right chest [ figure 3 ] .
the rest of the skin , scalp , genitalia , nails , and mucous membranes were clear of lesions , and there was no associated physiological or pathological condition .
he denied any alcohol consumption , and laboratory tests were normal ( chemistry , hormone , and infectious panel ) .
a skin biopsy was performed , which revealed thin wall dilated capillaries in the papillary and reticular dermis .
a 35-year - old woman was referred to dermatology with telangiectatic , scattered , pinpoint , erythematosus , and smooth papules over her anterior left chest [ figure 4 ] .
she had delivered a child 2 weeks before and reported that the lesions began during the last 6 months of her pregnancy .
she had another 4-year - old child and referred similar cutaneous findings during the last months of her previous pregnancy , which disappeared a few months after delivery .
blaschko first description of unt dates from 1899 . since then many alternative designations have been proposed
nowadays , however the two terms most frequently used are  unilateral nevoid telangiectasia  and  unilateral dermatomal superficial telangiectasia ,  this last one described by wilkin in 1978 .
although it is thought to be a rare condition with only fewer than 100 reported cases , we believe that it is more common . however , until today , there is no published article concerning this issue in our country .
the rare congenital form occurs more frequently in males in an autosomal dominant fashion and appears during or shortly after the neonatal period . on the other hand
, the acquired form occurs almost exclusively in young female patients associated with physiologic conditions .
clinically , unt is characterized by multiple , unilateral , linearly arranged , blanching telangiectases in a dermatomal ( most frequently involving the third and fourth cervical dermatomes ) or blaschkoid pattern .
predilection sites are the face , neck , shoulder - arm region , and thorax .
a pale ring , referred as an anemic halo , may be observed surrounding the telangiectases . why the pathology is distributed in a strict unilateral manner
is unclear ; however , it is thought that a localized increase in estrogen receptors caused by a chromosomal mosaicism , that is unmasked at times of relative estrogen excess , accounts for the distribution that follows blaschko 's lines .
histologically , there are multiple , dilated , thin - walled vessels lined by plump endothelial cells in the papillary and upper reticular dermis , characteristic of unt .
other laboratory findings recently described are the functional impairments at the blood vessel level in laser doppler image .
lastly , capillaroscopy showed morphologically altered vascular structures with megacapillaries in the fingers nail grooves .
unt typically persists but rarely , in acquired cases , resolves spontaneously if the eliciting factor is removed .
the use of pulsed dye laser has been a useful alternative in aesthetic improvement of the condition ; however , none of the two adult patients or the parents of the child presented in this article accepted to be submitted to such treatment , advocating the lack of symptoms of the disease and the minor aesthetic disarrangement .
the three patients described in this article were observed in a short period of time ( 3 months ) , which emphasizes the theory advocated by some authors , that unt is not a rare condition , but is only subdiagnosed and subreported in the literature , perhaps due to its asymptomatic character .
the occurrence of two idiopathic cases points out to the importance of this subtype , with few reported cases to date , and raises again the question whether estrogen receptors do play a significant role in the pathogenesis of this disorder .
unilateral nevoid telangiectasia is a rare congenital or acquired disorder . 
 the majority of the authors propose that it is probably related to elevated estrogen receptors , although other authors advocate different theories . 
 we report the occurrence of lesions clinically compatible with this diagnosis in a child and two adults . 
 only one of the patients had a background of estrogen elevation . 
 the other two patients were healthy . 
 we stress the occurrence of this disease in a rare idiopathic form and point out to the relative elevated incidence of it , because the patients discussed in this article were observed for a short period of time .