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A Rare Association of Childhood Alopecia Areata and Blepharophimosis-Ptosis-Epicanthus Inversus Syndrome: Successfully Treated with Diphenylcyclopropenone.

Jagadeesan S, Balasubramanian P, Panicker VV, Anjaneyan G, Thomas J - Int J Trichology (2015 Apr-Jun)

Bottom Line: The genetic background of alopecia areata has only recently begun to get unraveled.We report the association of a case of pediatric alopecia areata with a rare genetic syndrome-blepharophimosis-ptosis-epicanthus inversus syndrome (BPES), which responded well to topical immunotherapy with diphenylcyclopropenone.In the background of increasing evidence surfacing on the genetic basis of alopecia areata, this association may be of significance.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, Amrita Institute of Medical Sciences, Kochi, Kerala, India.

ABSTRACT
The genetic background of alopecia areata has only recently begun to get unraveled. We report the association of a case of pediatric alopecia areata with a rare genetic syndrome-blepharophimosis-ptosis-epicanthus inversus syndrome (BPES), which responded well to topical immunotherapy with diphenylcyclopropenone. In the background of increasing evidence surfacing on the genetic basis of alopecia areata, this association may be of significance.

No MeSH data available.


Related in: MedlinePlus

Treatment response: Frontal view (a) after 4 weeks of treatment, (b) after 4 months. Bilaterally symmetrical ptosis, blepharophimosis, telecanthus (increased distance between the medial canthi) can be appreciated in the figure. The hypopigmented scars along the medial canthi are following surgical correction of epicanthus inversus (a fold of skin of the lower eyelid seen along the inner canthi in blepharophimosis-ptosis-epicanthus inversus syndrome)
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Figure 3: Treatment response: Frontal view (a) after 4 weeks of treatment, (b) after 4 months. Bilaterally symmetrical ptosis, blepharophimosis, telecanthus (increased distance between the medial canthi) can be appreciated in the figure. The hypopigmented scars along the medial canthi are following surgical correction of epicanthus inversus (a fold of skin of the lower eyelid seen along the inner canthi in blepharophimosis-ptosis-epicanthus inversus syndrome)

Mentions: She was started on topical immunotherapy with diphenylcyclopropenone (DPCP). Within 4 weeks of starting, re-growth of hairs was noted. Within 4 months, there was full thickness growth of hair covering entire scalp [Figures 2 and 3]. The treatment was tapered and stopped in 10 months. She is under follow-up since 1-month, and her hairs are continuing to grow with no abnormal shedding.


A Rare Association of Childhood Alopecia Areata and Blepharophimosis-Ptosis-Epicanthus Inversus Syndrome: Successfully Treated with Diphenylcyclopropenone.

Jagadeesan S, Balasubramanian P, Panicker VV, Anjaneyan G, Thomas J - Int J Trichology (2015 Apr-Jun)

Treatment response: Frontal view (a) after 4 weeks of treatment, (b) after 4 months. Bilaterally symmetrical ptosis, blepharophimosis, telecanthus (increased distance between the medial canthi) can be appreciated in the figure. The hypopigmented scars along the medial canthi are following surgical correction of epicanthus inversus (a fold of skin of the lower eyelid seen along the inner canthi in blepharophimosis-ptosis-epicanthus inversus syndrome)
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4502480&req=5

Figure 3: Treatment response: Frontal view (a) after 4 weeks of treatment, (b) after 4 months. Bilaterally symmetrical ptosis, blepharophimosis, telecanthus (increased distance between the medial canthi) can be appreciated in the figure. The hypopigmented scars along the medial canthi are following surgical correction of epicanthus inversus (a fold of skin of the lower eyelid seen along the inner canthi in blepharophimosis-ptosis-epicanthus inversus syndrome)
Mentions: She was started on topical immunotherapy with diphenylcyclopropenone (DPCP). Within 4 weeks of starting, re-growth of hairs was noted. Within 4 months, there was full thickness growth of hair covering entire scalp [Figures 2 and 3]. The treatment was tapered and stopped in 10 months. She is under follow-up since 1-month, and her hairs are continuing to grow with no abnormal shedding.

Bottom Line: The genetic background of alopecia areata has only recently begun to get unraveled.We report the association of a case of pediatric alopecia areata with a rare genetic syndrome-blepharophimosis-ptosis-epicanthus inversus syndrome (BPES), which responded well to topical immunotherapy with diphenylcyclopropenone.In the background of increasing evidence surfacing on the genetic basis of alopecia areata, this association may be of significance.

View Article: PubMed Central - PubMed

Affiliation: Department of Dermatology, Amrita Institute of Medical Sciences, Kochi, Kerala, India.

ABSTRACT
The genetic background of alopecia areata has only recently begun to get unraveled. We report the association of a case of pediatric alopecia areata with a rare genetic syndrome-blepharophimosis-ptosis-epicanthus inversus syndrome (BPES), which responded well to topical immunotherapy with diphenylcyclopropenone. In the background of increasing evidence surfacing on the genetic basis of alopecia areata, this association may be of significance.

No MeSH data available.


Related in: MedlinePlus