Literature DB >> 15918232

Progressive facial hemiatrophy: a complex disorder not only affecting the face. A report in a monozygotic male twin pair.

C V Hulzebos1, T W de Vries, W Armbrust, P J J Sauer, W S Kerstjens-Frederikse.   

Abstract

UNLABELLED: Progressive facial hemiatrophy (PFH) is a ubiquitous disease, characterized by hyperpigmentation of skin followed by unilateral craniofacial atrophy of subcutaneous tissues, including fat, muscle and bone. Hereditary factors have been postulated to be involved in the aetiology of PFH. Yet, the occurrence of PFH in one of two identical male twins reported here makes this possibility unlikely. PFH usually occurs in the first two decades of life, and the clinical presentation resembles linear scleroderma. PFH may be complicate by autoimmune, neurological, ocular and dental disorders. Management of PFH comprises a long term follow-up of somatic disorders, and prevention of psychological problems. Treatment of PFH is symptomatic and consists of plastic surgery after the disease activity has stopped.
CONCLUSION: The occurrence of PFH in one of monozygotic twin pair suggests that genetic factors are not involved in its aetiology. Early diagnosis of PFH and accurate follow-up is essential to disclose the occurrence of complications.

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Year:  2004        PMID: 15918232

Source DB:  PubMed          Journal:  Acta Paediatr        ISSN: 0803-5253            Impact factor:   2.299


  4 in total

1.  [Hemiatrophia faciei progressiva (Parry-Romberg-syndrome) ].

Authors:  E Roller; J Reifenberger; B Homey; D Bruch-Gerharz
Journal:  Hautarzt       Date:  2006-10       Impact factor: 0.751

Review 2.  The central nervous system manifestations of localized craniofacial scleroderma: a study of 10 cases and literature review.

Authors:  Ezekiel Maloney; Sarah J Menashe; Ramesh S Iyer; Sarah Ringold; Amit K Chakraborty; Gisele E Ishak
Journal:  Pediatr Radiol       Date:  2018-07-03

3.  A significant proportion of children with morphea en coup de sabre and Parry-Romberg syndrome have neuroimaging findings.

Authors:  Yvonne E Chiu; Sheetal Vora; Eun-Kyung M Kwon; Mohit Maheshwari
Journal:  Pediatr Dermatol       Date:  2012 Nov-Dec       Impact factor: 1.588

4.  Progressive facial hemiatrophy - a case series.

Authors:  Feroze Kaliyadan; Kaberi Biswas; A D Dharmaratnam
Journal:  Indian J Dermatol       Date:  2011 Sep-Oct       Impact factor: 1.494

  4 in total

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