DISCUSSION
Angioma serpiginosum (AS) is a rare vascular disorder described first by
Hutchinson in 1889 as a “serpiginous or infective nevus” and then
named by Crocker in 1894.2,3 Cases are usually
sporadic, however familial cases with an autosomal dominant or X linked
dominant inheritance and few cases with PORCN gene mutation or deletion
have been reported .4 Angioma serpiginosum results
from the proliferation of endothelial cells resulting dilated
capillaries. There are various theories regarding its pathogenesis. One
of the theories suggesting the role of estrogen while another theory
calming the role of cold temperature in its pathogenesis. However, both
of the theories are not accepted universally.7,8
The condition usually starts in childhood or birth with a female
predominance.5,6 This condition presents as
asymptomatic multiple, pinpoint violaceous or erythematous macules,
non-blanching and clustered in an area or form a large sheets
distributed in serpiginous, linear (Blaschkoid distribution) or annular
pattern.1 Lesions are typically unilateral and located
predominantly on the lower limbs and extremities but can be
extensive.5,9 However, palms, soles, mucosal, and
truncal involvement is rare. Few cases with segmental truncal and
mono-lateral plantar area involvement have been
reported.6,10,11,,12,13,14 This condition progresses
slowly and usually attains stability at puberty with some partial
spontaneous resolution in late adulthood. The diagnosis is usually made
clinically that is confirmed by histological findings of distended
ectatic capillaries lined by flattened endothelium cells of normal
appearance and the absence of inflammation, erythrocyte extravasations,
and hemosiderin deposition.15 Treatment is only
indicated for cosmetic reasons for which vascular laser is the best
option.
In our case, the patient presented with localized telangiectasia without
skin atrophy. The differentials for the condition include Angioma
serpiginosum, capillary malformation, unilateral nevoid telangiectasia,
and purpuric conditions. 17 Presence of the lesions
since birth as unilateral, asymptomatic erythematous, pinpoint to
pinhead-sized non blanchable macules and papules in the zosteriform
pattern (T8-T10) without atrophy and perilesional halo supports the
diagnosis of angioma serpiginosum. The histological findings confirmed
the diagnosis. The peculiarity about our case is the site of lesion.
This is the first case as per our knowledge with presentation of the
lesion on abdomen. In our case, the parents were not concerned about the
lesions so, no treatment was given and asked for follow-up later in case
of cosmetic concerns.