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Linear atrophoderma of Moulin: a case report and review of the literature.
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PMID:  23785629     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Linear atrophoderma of Moulin is a rare, acquired, linear dermatosis. We present a 17-year-old girl with multiple asymptomatic brownish atrophic plaques in a zosteriform distribution on the left side of the trunk. Clinical presentation and dermatopathology was compatible with the diagnosis of linear atrophoderma. Twenty years after its initial description by Moulin, there are yet a limited number of case reports and unanswered questions regarding this entity.
Authors:
Aikaterini Patsatsi; Aikaterini Kyriakou; George Chaidemenos; Dimitrios Sotiriadis
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Publication Detail:
Type:  Journal Article     Date:  2013-01-31
Journal Detail:
Title:  Dermatology practical & conceptual     Volume:  3     ISSN:  2160-9381     ISO Abbreviation:  Dermatol Pract Concept     Publication Date:  2013 Jan 
Date Detail:
Created Date:  2013-06-20     Completed Date:  2013-06-21     Revised Date:  2013-06-25    
Medline Journal Info:
Nlm Unique ID:  101585990     Medline TA:  Dermatol Pract Concept     Country:  United States    
Other Details:
Languages:  eng     Pagination:  7-11     Citation Subset:  -    
Affiliation:
Second Dermatology Department, Aristotle University School of Medicine, Papageorgiou Hospital, Thessaloniki, Greece.
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Journal Information
Journal ID (nlm-ta): Dermatol Pract Concept
Journal ID (iso-abbrev): Dermatol Pract Concept
Journal ID (publisher-id): DP
ISSN: 2160-9381
Publisher: Derm101.com
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Copyright: ©2013 Patsatsi et al.
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Received Day: 14 Month: 6 Year: 2012
Accepted Day: 25 Month: 11 Year: 2012
collection publication date: Month: 1 Year: 2013
Electronic publication date: Day: 31 Month: 1 Year: 2013
Volume: 3 Issue: 1
First Page: 7 Last Page: 11
PubMed Id: 23785629
ID: 3663377
DOI: 10.5826/dpc.0301a03
Publisher Id: dp0301a03

Linear atrophoderma of Moulin: a case report and review of the literature
Aikaterini Patsatsi, M.D.1
Aikaterini Kyriakou, M.D. MSc1
George Chaidemenos, M.D.2
Dimitrios Sotiriadis, M.D.1
1Second Dermatology Department, Aristotle University School of Medicine, Papageorgiou Hospital, Thessaloniki, Greece
2Dermatology Practice, Thessaloniki, Greece
Correspondence: Corresponding author: Aikaterini Patsatsi, M.D., Plagiari PO Box 461, 57500 Thessaloniki, Greece. Tel. +306944500285; Fax. +302310991583. Email: katerinapatsatsi@gmail.com.

Case presentation

A 17-year-old girl presented with a six-month history of multiple asymptomatic brownish atrophic plaques in a zosteriform distribution on the left side of her trunk (Figure 1). There was no family history of a similar skin disease. Laboratory studies were unremarkable for any chronic or autoimmune disorder. Biopsy of a lesion showed a normal epidermis with increased pigmentation of the basal layer and a broad dermis with thickened collagen fibers and diminished periadnexal and subcutaneous fat tissue (Figures 2, 3, 4).

Clinical and dermatopathologic findings were compatible with atrophoderma of Moulin. The use of topical steroids and tacrolimus as a second-line regimen did not result in any improvement of lesions.


Discussion

Linear atrophoderma is a rare, acquired, linear dermatosis. It is named after Moulin, who, in 1992, reported on five patients with pigmented and more or less atrophic bands along Blaschko’s lines [1].

The age of onset in the first described cases ranged from 6 to 20 years. Lesions were unilateral, forming a recumbent “S” pattern, and the intensity of pigmentation and atrophy was variable. They remained stable throughout an observation period of 2 to 30 years. Of the skin biopsies performed on three patients, there was only irregular and moderate hyperpigmentation of the basal layer. In the dermis, there was no distinct pigment incontinence, no inflammation or alteration of connective tissue texture, and the clinical impression of skin atrophy was attributed to atrophy of the subcutaneous tissue [1].

Up to now there have been 30 reported cases of linear atrophoderma of Moulin [222]. In 2005, Ang et al mentioned that many cases of linear dermatoses were grouped under the umbrella of linear atrophoderma [23]. In Table 1, 30 reported cases that resemble the initial description of linear atrophoderma of Moulin are listed.

Now, twenty years after the initial description of this entity, there are still some unresolved issues. The differential diagnosis of zosteriform or linear scleroderma is not clear clinically nor dermatopathologically. It may well be that linear atrophoderma of Moulin and zosteriform or linear scleroderma belong to the spectrum of a single disease.

With the growing literature it has been demonstrated that age of onset is not limited to childhood or adolescence. The disease may also present later in life. Lesions are not always unilateral. They may be bilateral but in a linear distribution along Blaschko’s lines (Table 1). Distribution mainly along the Blaschko lines reflects mosaicism.

Dermatopathologic findings of linear atrophoderma of Moulin vary. The epidermis is normal in the majority of cases. However, in two reports there was vacuolar degeneration of the basement membrane [2,7]. Few reports suggest an inflammatory early stage is suggested. The question of a transient inflammatory early stage that is no longer present by the time of clinical evaluation and biopsy remains.

Although the alteration of the connective tissue is not described in the original paper, most of the reviewed papers describe an increase of collagen. Unaltered, fragmented or decreased collagen fibers have been also reported in isolated cases, as well as edema of the dermis (Table 1). Another issue that has not been addressed is the cause of the clinical presentation of atrophic plaques. Does the loss of subcutaneous fat result in atrophy?

In conclusion, even now, 20 years after the first description of atrophoderma of Moulin this rare disease is puzzling. It seems to occur at any age. It is characterized clinically by the presence of atrophic patches distributed along Blaschko lines and dermatopathologically by a normal epidermis with a hyperpigmented basal layer, a dermis with thickened collagen fibers and loss of subcutaneous fat. Etiology and pathogenesis of this disease remains still unclear.


Notes

Funding: None.

Competing interests: The authors have no conflicts of interest to disclose.

All authors have contributed significantly to this publication.

References
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Article Categories:
  • Observation

Keywords: linear, atrophoderma, Moulin, linear scleroderma.

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