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: 2813  
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 
E-IJD® - CORRESPONDENCE
Year : 2022  |  Volume : 67  |  Issue : 2  |  Page : 209
A greyish-black nodule on the abdominal skin – Uncommon morphology and location of epithelioid hemangioma


1 From the Nirvan Clinic, Vadodara, Gujarat, India
2 Alo Clinic, Kolkata, West Bengal, India
3 Department of Pathology, Government Medical College, Trivandrum, Kerala, India

Date of Web Publication13-Jul-2022

Correspondence Address:
Shyam B Verma
From the Nirvan Clinic, Vadodara, Gujarat
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijd.ijd_941_21

Rights and Permissions



How to cite this article:
Verma SB, Ghosh K, Gopinath N. A greyish-black nodule on the abdominal skin – Uncommon morphology and location of epithelioid hemangioma. Indian J Dermatol 2022;67:209

How to cite this URL:
Verma SB, Ghosh K, Gopinath N. A greyish-black nodule on the abdominal skin – Uncommon morphology and location of epithelioid hemangioma. Indian J Dermatol [serial online] 2022 [cited 2022 Aug 17];67:209. Available from: https://www.e-ijd.org/text.asp?2022/67/2/209/350855




Sir,

A 62-year-old woman presented to one of us (SV) with a solitary grayish black oval plaque with a surrounding brownish halo on the abdomen for 3 years. It gradually increased in size to 2 cm across at presentation [Figure 1]a. It was rough-surfaced, asymptomatic, and with a firm to hard consistency. There was no history of bleeding on trauma. She had no other comorbidities except ulcerative colitis, for which she was prescribed salazopyrine. Her hematological, biochemical, and radiological profile, including levels of eosinophils, IgE, and abdominal ultrasound, were normal.
Figure 1: a: Greyish black keratotic oval plaque on the abdomen. b: Dermatoscopy of the plaque. Blue arrow showing white reticular lines, yellow star showing red lacunae, red arrow showing delicate reticular pigmentary lines (Polarized View Dermlite DL3N, 3Gen, USA. 10×). c: Hyperplastic epidermis with irregular elongation of rete ridges. Upper dermis shows clustering of blood vessels with thickened walls lined by epithelioid endothelial cells. Low power 100×. d: Thickened walls with Plump epithelioid endothelial cells lining the lumina of thick-walled vessels. Perivascular lymphocytes and eosinophils also spilling into the interstitium. High power 400×. The stain is Hematoxylin& Eosin. (H&E)

Click here to view


Dermoscopic examination showed white reticular lines (suggestive of hyperkeratosis) and red lacunae (suggestive of dilated vascular channels) with a delicate reticular pigmentary network at the periphery [Figure 1]b. Punch biopsy showed hyperplastic epidermis with upper dermis showing clustering of variably sized elongated blood vessels with thickened walls lined by plump endothelial cells [Figure 1]c, [Figure 1]d. Additionally, there was perivascular and interstitial infiltrate of lymphocytes and eosinophils. Immunohistochemical study with positive CD31 and CD34 [Figure 2]a markers confirmed the vascular nature of the tumor. Final diagnosis ruled in the favor of epithelioid hemangioma (EH). The lesion regressed significantly when treated with clobetasol propionate plus 3% salicylic acid ointment and timolol drops for approximately 6 months.
Figure 2: a: Significant regression of the plaque after 6 months treatment

Click here to view


Epithelioid hemangioma (EH) and angiolymphoid hyperplasia with eosinophilia (ALHE) are uncommon benign vasoproliferative disorders of unknown etiology, and both terms are often used interchangeably. It is commonly seen in individuals aged 20–50 years, with a mean age of onset of 30–34 years.[1],[2] The onset at the age of 60 years, as in our patient, is uncommon. Lesions typically are dome-shaped, erythematous, red-brown, violaceous, variably sized papules, plaques, or nodules with a smooth surface.[1],[2],[3],[4],[5],[6] Our case presented as an unusual hard grayish-black nodule with a verrucous surface instead. Head and face region, including scalp, forehead, ear, and periauricular region, are favored locations.[2],[3],[5] EH is rarely reported on the trunk as seen in this case.[2],[3],[4],[5],[6] Twenty percent of patients may have peripheral blood eosinophilia without raised IgE and some may show regional lymphadenopathy. Thick-walled blood vessels, lined by large, plump endothelial cells with ovoid nuclei (called epithelioid, histiocytoid, or hobnail cells) projecting into the lumen, with intracytoplasmic vacuoles is a characteristic feature of EH.[2],[3],[4] Another typical feature is the presence of perivascular and interstitial infiltrates containing eosinophils and lymphocytes with 5%–15% of the infiltrate formed by the former.[2],[3],[4],[7] Kimura's disease, the closest differential diagnosis of EH, was ruled out by the absence of a soft tissue mass, absent lymphadenopathy on ultrasonography, no peripheral blood eosinophilia, normal serum immunoglobulin E, and absence of lymphoid follicles on histopathology.[2],[3],[7] Other differential diagnoses, namely lobular capillary hemangioma, hemangioendothelioma, angiosarcoma, Kaposi's sarcoma, and cutaneous metastasis, were ruled out by histopathology.[2],[3],[7]

EH grows slowly and persists indefinitely, though spontaneous resolution has been uncommonly reported.[2],[3],[7] Surgical excision has a recurrence rate of approximately 40% as incomplete excision is common.[2],[3],[4] Other modalities such as electrosurgery, cryotherapy, ablative laser surgery, photodynamic therapy, systemic, intralesional or topical corticosteroids, imiquimod, topical tacrolimus, and timolol have been used with inconsistent outcomes.[1],[4]

In conclusion, we describe a grayish-black hyperkeratotic variant of EH on the abdomen for its unique clinical and dermoscopic appearance and the rarely described location. The beneficial outcome of treatment with the steroid salicylic acid cream along with timolol drops should be evaluated further.

Acknowledgements

We thank Dr. Shekhar Neema, AFMC, Pune for his expert comments on the dermatoscopic findings in this case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Slimani Y, Hali F, Sid'Ahmed Tolba C, Marnissi F, Chiheb S. Spontaneous regression of angiolymphoid hyperplasia with eosinophilia (AHLE): A case report. Ann Med Surg (Lond) 2021;66:102376.  Back to cited text no. 1
    
2.
Panse G. Angiolymphoid Hyperplasia with Eosinophilia: Background, Pathophysiology. 2021. Available from: https://emedicine.medscape.com/article/1082603-overview. [Last accessed on 2021 Oct 25].  Back to cited text no. 2
    
3.
Guo R, Gavino AC. Angiolymphoid hyperplasia with eosinophilia. Arch Pathol Lab Med 2015;139:683-6.  Back to cited text no. 3
    
4.
Trindade F, Haro R, Requena L. Giant angiolymphoid hyperplasia with eosinophilia on the chest. J Cutan Pathol 2009;36:493-6.  Back to cited text no. 4
    
5.
Olsen TG, Helwig EB. Angiolymphoid hyperplasia with eosinophilia. J Am Acad Dermatol 1985;12:781-96.  Back to cited text no. 5
    
6.
Zaraa I, Mlika M, Chouk S, Chelly I, Mokni M, Zitouna M, et al. Angiolymphoid hyperplasia with eosinophilia: A study of 7 cases. Dermatol Online J 2011;17:1.  Back to cited text no. 6
    
7.
Lazar JA. Tumors of vascular origin. In: Calonje E, Brenn A, Billings S, editors. KcKee's Pathology of the Skin with Clinical Correlations. 5th ed. Elsvier. 2020. p. 1839-42.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2]



 

Top
Print this article  Email this article
 
 
  Search
 
  
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Article in PDF (716 KB)
    Citation Manager
    Access Statistics
    Reader Comments
    Email Alert *
    Add to My List *
* Registration required (free)  


    References
    Article Figures

 Article Access Statistics
    Viewed170    
    Printed6    
    Emailed0    
    PDF Downloaded0    
    Comments [Add]    

Recommend this journal