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Juvenile localized scleroderma with port wine stain: coincidental or possible common pathogenetic association.

Kacar SD, Ozuguz P, Polat S, Kacar E, Polat O, Tokyol C - Indian J Dermatol (2015 Mar-Apr)

Bottom Line: Port wine stain and juvenile localized scleroderma are two different dermatoses usually encountered in pediatric age group.Coexistence of both diseases is not found yet.We herein present a case of juvenile localized scleroderma on the left side of trunk, with congenital port wine stain located on the ipsilateral face at V1-V2 distribution.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, School of Medicine, Afyon Kocatepe University, Afyonkarahisar, Turkey.

ABSTRACT
Port wine stain and juvenile localized scleroderma are two different dermatoses usually encountered in pediatric age group. Up to now, there are reports of morphea patients initially diagnosed and treated as port wine stain. Coexistence of both diseases is not found yet. We herein present a case of juvenile localized scleroderma on the left side of trunk, with congenital port wine stain located on the ipsilateral face at V1-V2 distribution.

No MeSH data available.


Related in: MedlinePlus

Firm, erythematous-brown lesions on the left forearm (a) and thigh (b)
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Figure 1: Firm, erythematous-brown lesions on the left forearm (a) and thigh (b)

Mentions: A 21-year-old female patient was attended to our clinic for lesions which have started on anterior aspect of the left thigh 10 years before and new ones which have recently appeared on the left forearm and the lateral aspect of the left breast. The patient received various topical treatments, narrow-band UVB, hydroxychloroquine and dapsone previously with a diagnosis of morphea. She did not receive any treatment for 6 months and the breast lesions occurred during this time. On dermatological examination, erythematous-brown firm plaques on the anterior aspect of the left thigh, left forearm, medial aspect of the upper arm and lateral aspect of the left breast were observed [Figure 1a and b]. Furthermore she had congenital dark red telangiectatic macular plaques localized in ipsilateral V1 and V2 distribution on the face [Figure 2]. The punch biopsy from the new plaque on the breast was compatible with localized scleroderma [Figure 3], with dermal perivascular lymphomononuclear infiltration and focal sclerosis in subkutis and deep dermis. The lesion located on the face diagnosed as a PWS. Ophthalmologic and neurological examinations were normal. There was also no pathology in cranial computerized tomography scan and magnetic resonance imaging. On laboratory examination, complete blood count, urinalysis, renal and liver function tests were within the normal range and antinuclear antibodies (ANA) were positive at 1/400 titer. Further autoimmune panel were negative. Methotrexate treatment (15 mg/week) was started for progressive disease and she is still on follow ups.


Juvenile localized scleroderma with port wine stain: coincidental or possible common pathogenetic association.

Kacar SD, Ozuguz P, Polat S, Kacar E, Polat O, Tokyol C - Indian J Dermatol (2015 Mar-Apr)

Firm, erythematous-brown lesions on the left forearm (a) and thigh (b)
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4372961&req=5

Figure 1: Firm, erythematous-brown lesions on the left forearm (a) and thigh (b)
Mentions: A 21-year-old female patient was attended to our clinic for lesions which have started on anterior aspect of the left thigh 10 years before and new ones which have recently appeared on the left forearm and the lateral aspect of the left breast. The patient received various topical treatments, narrow-band UVB, hydroxychloroquine and dapsone previously with a diagnosis of morphea. She did not receive any treatment for 6 months and the breast lesions occurred during this time. On dermatological examination, erythematous-brown firm plaques on the anterior aspect of the left thigh, left forearm, medial aspect of the upper arm and lateral aspect of the left breast were observed [Figure 1a and b]. Furthermore she had congenital dark red telangiectatic macular plaques localized in ipsilateral V1 and V2 distribution on the face [Figure 2]. The punch biopsy from the new plaque on the breast was compatible with localized scleroderma [Figure 3], with dermal perivascular lymphomononuclear infiltration and focal sclerosis in subkutis and deep dermis. The lesion located on the face diagnosed as a PWS. Ophthalmologic and neurological examinations were normal. There was also no pathology in cranial computerized tomography scan and magnetic resonance imaging. On laboratory examination, complete blood count, urinalysis, renal and liver function tests were within the normal range and antinuclear antibodies (ANA) were positive at 1/400 titer. Further autoimmune panel were negative. Methotrexate treatment (15 mg/week) was started for progressive disease and she is still on follow ups.

Bottom Line: Port wine stain and juvenile localized scleroderma are two different dermatoses usually encountered in pediatric age group.Coexistence of both diseases is not found yet.We herein present a case of juvenile localized scleroderma on the left side of trunk, with congenital port wine stain located on the ipsilateral face at V1-V2 distribution.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, School of Medicine, Afyon Kocatepe University, Afyonkarahisar, Turkey.

ABSTRACT
Port wine stain and juvenile localized scleroderma are two different dermatoses usually encountered in pediatric age group. Up to now, there are reports of morphea patients initially diagnosed and treated as port wine stain. Coexistence of both diseases is not found yet. We herein present a case of juvenile localized scleroderma on the left side of trunk, with congenital port wine stain located on the ipsilateral face at V1-V2 distribution.

No MeSH data available.


Related in: MedlinePlus