Indian Journal of Dermatology
  Publication of IADVL, WB
  Official organ of AADV
Indexed with Science Citation Index (E) , Web of Science and PubMed
 
Users online: 2021  
Home About  Editorial Board  Current Issue Archives Online Early Coming Soon Guidelines Subscriptions  e-Alerts    Login  
    Small font sizeDefault font sizeIncrease font size Print this page Email this page


 
Table of Contents 
CASE REPORT
Year : 2018  |  Volume : 63  |  Issue : 5  |  Page : 409-411
Idiopathic eruptive macular pigmentation - uncommon presentation of an uncommon condition


Kingsway Clinic, Matunga, Mumbai, Maharashtra, India

Date of Web Publication31-Aug-2018

Correspondence Address:
Dr. Resham Jayvant Vasani
C-1, 22, Karmakshetra, Near Shanmukhanada Hall, Sion, Mumbai 400037, Maharashtra
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.IJD_12_18

Rights and Permissions

   Abstract 


Idiopathic eruptive macular pigmentation (IEMP) is an asymptomatic condition with dark brown, nonconfluent lesions chiefly occurring in children and adolescents. The usual sites involved are face, trunk, extremities, and the lesions resolve over few months to years. We report an unusual presentation of this uncommon condition. A 22-year-old male presented with multiple dark-colored asymptomatic lesions on the scalp and forehead for the past 3 years. There was no history of preceding symptoms, drug, or applications, past or family history of such lesions. Examination revealed multiple hyperpigmented macules and barely elevated nonscaly plaques with a velvety feel akin to acanthosis nigricans (AN). Dermoscopy was similar to AN. Blood sugar, thyroid, and lipid profiles were normal. Histology showed “pigmented papillomatosis” concluding the final diagnosis of IEMP. Confluence of lesions is a rarely described phenomenon, with the scalp being a hitherto unreported site. The velvety feel of lesions, the dermoscopic, and histopathological findings further substantiate the hypothesis of this entity being an eruptive variant of AN.


Keywords: Eruptive acanthosis nigricans, idiopathic eruptive macular pigmentation, Pigmented papillomatosis


How to cite this article:
Vasani RJ. Idiopathic eruptive macular pigmentation - uncommon presentation of an uncommon condition. Indian J Dermatol 2018;63:409-11

How to cite this URL:
Vasani RJ. Idiopathic eruptive macular pigmentation - uncommon presentation of an uncommon condition. Indian J Dermatol [serial online] 2018 [cited 2020 Sep 22];63:409-11. Available from: http://www.e-ijd.org/text.asp?2018/63/5/409/240090





   Introduction Top


Idiopathic eruptive macular pigmentation (IEMP) is an uncommon pigmentary disorder of an unknown etiology, characterized by the presence of asymptomatic pigmented macules that involve the face, trunk, and proximal extremities in children and adolescents.[1] Herein, we report an unusual presentation of IEMP, with lesions limited to the scalp and forehead with a velvety feel on touch, coupled with dermoscopic and histologic features similar to acanthosis nigricans (AN) reverberating the question of whether IEMP is an eruptive variant of AN.


   Case report Top


A 22-year-old male presented with multiple dark-colored lesions over the scalp and forehead for the past 3 years. There was an insidious progression of lesions with increase in the number and tendency toward coalescence on the forehead, for the past 3 months which prompted him to seek medical attention. The lesions were essentially asymptomatic and appeared de novo without any preceding event. The patient denied any associated comorbidity or medication. The past, personal, and family histories were not contributory.

Cutaneous examination revealed multiple ill-defined, dark brown to black-colored macules and minimally elevated plaques with a velvety feel on touch. The smaller 0.5 cm sized plaques located on the scalp were discrete [Figure 1]a and [Figure 1]b but coalesced to form a larger irregular 3 cm×6 cm plaque extending from the frontal aspect of the scalp to the forehead [Figure 1]c and [Figure 1]d. The plaque had a velvety feel. Darier's sign was negative. Rest of the cutaneous examination was normal. The palms, soles, mucosae, hair, and nails were normal. Systemic examination was unremarkable. On the basis of history and clinical examination, a differential diagnosis of IEMP, urticaria pigmentosa, and lichen planus pigmentosus were considered.
Figure 1: (a and b) Multiple hyperpigmented discrete plaques on the scalp. (c) Hyperpigmented minimally elevated plaques tending to coalesce on forehead. (d) Close-up of the hyperpigmented plaque on forehead showing accentuation of the skin markings

Click here to view


Hematological examination including the complete blood count, serum insulin level, blood sugar and lipid profile was normal. Dermoscopy of the affected areas showed linear cristae cutis and sulci cutis in addition to an accentuation of the reticular pattern similar to that noticed in AN [Figure 2]. Histopathological examination of hyperpigmented lesion from forehead showed an increase in the basal layer melanization without any increase in number of melanocytes. There was prominent papillomatosis with elongated dermal papillae [Figure 3]a. No significant dermal infiltrate and melanin incontinence were seen, and no interface changes were evident [Figure 3]b. Fontana Masson stain for melanin was positive with negative staining of toluidine blue for mast cells.
Figure 2: Dermatoscopy findings using DermLite DL4-Linear cristae cutis with sulci cutis and accentuation of reticular pattern

Click here to view
Figure 3: (a) Mild acanthosis with prominent papillomatosis and increased melanin in the basal layer with sparse perivascular infiltrate in the upper dermis (H and E, ×10); (b) Absence of basement membrane changes or melanin incontinence (H and E, ×40)

Click here to view


The clinical picture of multiple insidiously progressing asymptomatic hyperpigmented minimally raised plaques with the distinct velvety feel coupled with the histopathological finding of pigmented papillomatosis and a normal basement membrane ruled out other differentials such as urticaria pigmentosa, lichen planus pigmentosus, and fixed drug eruption and hence the final diagnosis was IEMP. The patient was counseled about the possibility of spontaneous regression of lesions and was prescribed topical tretinoin 0.025% cream as a night time application on the forehead lesions. The patient had been applying tretinoin for 3 months till reporting with minimal decrease in the thickness of the plaques.


   Discussion Top


Idiopathic eruptive macular hyperpigmentation is a rare entity with less than 60 cases reported worldwide.[2] The term IEMP was first proposed by Degos et al. in 1978 and several cases have been described in French literature.[3]

The etiopathogenesis of this entity is still unclear. All cases reported till date have had a sporadic occurrence.[1] The possible role of hormonal factors in pathogenesis should be considered as lesions appears in puberty and adolescence. In this case, the lesions were noticed at 19 year of age, which could be a possible delay in reporting, on account of the masking of the asymptomatic lesions on the scalp by hair. One report of aggravation of IEMP during pregnancy further supports the hormonal hypothesis.[4]

Sanz de Galdeano et al. proposed the diagnostic criteria. The first criterion was “eruption of brownish black discrete nonconfluent asymptomatic macules involving the neck, trunk and proximal extremities in children and adolescents.”[5] Based on a critical review of the existing literature on the topic, Joshi et al. suggested a modification of this criterion to – “eruption of brownish black discrete, nonconfluent asymptomatic macules, and slightly elevated plaques that resemble AN and involve face, neck, and proximal extremities.”[2] The current patient majorly fulfilled this modified criterion but had lesions on the scalp and the lesions did tend to confluence on the forehead. Lesions on the scalp could have been easily missed in the earlier reports of this disorder because of the masking by hair and confluence of lesions was also mentioned as a feature in the report by de Souza DF et al.[4]

The absence of preceding inflammation and drug intake in this patient rules out postinflammatory and drug-induced causes as fulfillment of the 2nd and 3rd criteria.

The histopathology of this case showing epidermal hypermelanosis with papillomatosis and absence of significant dermal changes reaffirmed the histopathological finding seen in the case series of Indian patients described by Joshi et al.[6] Normal mast cells ruled out urticaria pigmentosa completing the 5th criterion for diagnosis. Clinically, lesions look and feel like AN with dermoscopic picture showing linear cristae cutis and sulci cutis; corresponding to papillomatosis on histology, further substantiate the hypothesis of IEMP being an eruptive variant of AN. A single case described earlier did have mild concomitant AN of bilateral axillae without associated biochemical insulin resistance.[7]

None of the other cases reported till date, nor the one being reported currently had concomitant AN of the other sites or associated obesity. Considering the fact that this condition is commonly seen in adolescence or childhood, associated biochemical hyperinsulinism or malignancy is also thought to be unlikely. Hence, although morphologically and on dermoscopy and histopathology this eruption resembles AN, further studies with larger number of patients and long-term follow-up are needed to further conclusively define this terminology as a variant of AN or eruptive AN as suggested by Joshi et al.

Observing the natural course of IEMP, it is a chronic disorder with spontaneous resolution in months or years. The patient in question had the disease for 3 years and did not show any signs of regression. There exists a single case report outlining the chronic course of IEMP over 21 years with episodic regressions and recurrences.[8]

This case has been reported for its rarity and depicts a hitherto unreported presentation of the disease on the scalp. More dermoscopically and histopathologically backed reports of this entity are needed to know whether this entity represents eruptive AN.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Komorowska O, Szczerkowska-Dobosz A, Roziewska D, Czapiewski P. Idiopathic eruptive macular pigmentation – A rare pigmentary disorder. Postepy Dermatol Alergol 2011;28:149-52.  Back to cited text no. 1
    
2.
Joshi RS, Rohatgi S. Idiopathic eruptive macular pigmentation: A critical review of published literature and suggestions for revision of criteria for diagnosis. Indian J Dermatol Venereol Leprol 2015;81:576-80.  Back to cited text no. 2
[PUBMED]  [Full text]  
3.
Degos R, Civatte J, Belaïch S. Idiopathic eruptive macular pigmentation (author's transl). Ann Dermatol Venereol 1978;105:177-82.  Back to cited text no. 3
    
4.
de Souza DF, Cunha AC, Piñeiro-Maceira J, Gomes MK, Sodré CT, Ramos-e-Silva M, et al. Idiopathic eruptive macular pigmentation associated with pregnancy. Int J Dermatol 2010;49:810-2.  Back to cited text no. 4
    
5.
Sanz de Galdeano C, Léauté-Labrèze C, Bioulac-Sage P, Nikolic M, Taïeb A. Idiopathic eruptive macular pigmentation: Report of five patients. Pediatr Dermatol 1996;13:274-7.  Back to cited text no. 5
    
6.
Joshi R. Idiopathic eruptive macular pigmentation with papillomatosis: Report of nine cases. Indian J Dermatol Venereol Leprol 2007;73:402-5.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Subhadarshani S, Singh A, Ramateke PP, Verma KK. Idiopathic eruptive macular pigmentation in an Indian male. Indian Dermatol Online J 2017;8:367-70.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Mehta S, Aasi S, Cole R, Chu P, Weinberg JM. Idiopathic eruptive macular pigmentation: A case of 21 years' duration. J Am Acad Dermatol 2003;49:S280-2.  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Article in PDF (1,736 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    Abstract
   Introduction
   Case report
   Discussion
    References
    Article Figures

 Article Access Statistics
    Viewed2567    
    Printed25    
    Emailed0    
    PDF Downloaded74    
    Comments [Add]    

Recommend this journal