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 Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 3  |  Issue : 2  |  Page : 75-76

Multiple eccrine spiradenomas in zosteriform distribution involving three dermatomes


1 Department of Dermatology, Silchar Medical College, Silchar, Assam, India
2 Department of Pathology, Silchar Medical College, Silchar, Assam, India

Date of Web Publication5-Dec-2016

Correspondence Address:
Mahimanjan Saha
Department of Dermatology, Silchar Medical College, Silchar - 788 014, Assam
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2349-6029.195222

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  Abstract 


Eccrine spiradenoma is an uncommon benign tumor of the sweat glands, most frequently characterized by a solitary, painful, deep-seated nodule. A case of multiple spiradenomas in a zosteriform distribution involving three dermatomes is described with its clinico-pathological features.

Keywords: Spiradenoma, three, zosteriform


How to cite this article:
Saha M, Madhab V, Das SS, Begum C Z. Multiple eccrine spiradenomas in zosteriform distribution involving three dermatomes. Indian J Dermatopathol Diagn Dermatol 2016;3:75-6

How to cite this URL:
Saha M, Madhab V, Das SS, Begum C Z. Multiple eccrine spiradenomas in zosteriform distribution involving three dermatomes. Indian J Dermatopathol Diagn Dermatol [serial online] 2016 [cited 2020 Oct 28];3:75-6. Available from: https://www.ijdpdd.com/text.asp?2016/3/2/75/195222




  Introduction Top


Eccrine spiradenoma (ES), an uncommon benign adnexal tumor of sweat gland origin, occurs mainly in young adults, equally in both sexes.[1] It is characterized by a solitary, painful, deep-seated nodule on the trunk or proximal limbs.[1] Multiple ES in a zosteriform distribution is extremely rare and only few cases have been reported in literature.[2],[3],[4] Here, we describe a patient with multiple ES in a zosteriform distribution involving three dermatomes.


  Case Report Top


A 35-year-old woman presented with multiple, elevated lesions on the lower part of the face, trunk, and left upper extremity for the last 4 years. The lesions were insidious in onset but had become painful and increased in size for the last 2 years. Pain was intermittent and dull aching in nature.

On examination, multiple, erythematous to dusky, firm, tender nodules of different sizes, varying from 0.5 to 4 cm in diameter, were arranged in a linear pattern starting from the midline and extending along the chin and left submandibular region as well as over the front and back of the left side of chest and over the flexor aspect of the left arm and forearm [Figure 1]a,[Figure 1]b,[Figure 1]c. The nodules were along the distribution of mandibular branch of trigeminal nerve (V3), C3, and T1 dermatomes, respectively. Palms, soles, mucous membranes, hair, and nails were normal. Differential diagnoses considered were ES, neurofibroma, leiomyoma cutis, dermatofibroma, and angiolipoma.
Figure 1: (a) Multiple solid, firm, elevated nodules of varying size present over the chin, left submandibular region, left side of the chest, (b) lesions involving left scapular region, (c) nodules over the flexor aspect of the left arm and forearm

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Routine hematological and biochemical investigations were normal. Mantoux test was negative. Punch biopsy of a 1 cm sized nodule on hematoxylin and eosin stain revealed variable sized lobular aggregates surrounded by condensed hyaline connective tissue in the dermis [Figure 2]a. At higher magnification, the lobules were observed to be containing ductal structures with pale cells arranged around the lumina and dark cells in the periphery [Figure 2]b.
Figure 2: (a) Variable sized lobular aggregates surrounded by condensed hyaline connective tissue in the dermis (H and E, ×10), (b) lobules consisting of dual cells where larger, paler cells are grouped around lumina and smaller, darker cells arranged in the periphery (H and E, ×40)

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A final diagnosis of zosteriform spiradenoma was made based on clinical and characteristic histopathologic findings. Surgical excision of the tumors was advised, but the patient refused.


  Discussion Top


ES is a benign tumor arising from the transitional area between the secretory portion and the coiled duct of the eccrine sweat gland.[4]

Clinically, it presents as a solitary, rounded, bluish, firm, and painful dermal nodule about 3–50 mm in diameter.[5] The usual site is front of trunk and proximal limbs.[1] In our patient, tumors were distributed along specific dermatomes. Zosteriform and linear distribution of ESs is extremely rare where the tumors have been proposed to develop from an organic hamartomatous process frequently associated with different appendageal components.[4],[6] Shelley and Wood [2] suggested neural origin for zosteriform spiradenomas.

Histologically, the tumor commonly consists of several sharply demarcated lobules in the dermis without connections to the epidermis. The lobules may display a fibrous capsule and are often deeply basophilic due to dense packing of nuclei.[7] Two types of epithelial cells are present in the tumor lobules; those with small, dark nuclei arranged in the periphery and those with large, pale nuclei arranged in the center of the cellular aggregates.[1],[7] Markedly atypical cells with high mitotic figures, necrosis, loss of dual cell population, and lobular architecture indicate malignant transformation.[8] No evidence of a malignant transformation was noted in our case.

Spontaneous episodes of pain may occur in spiradenoma which has been attributed to contraction of myoepithelial cells, but electron microscopy shows the absence of myoepithelial cells in the tumor.[3] The exact explanation for pain is thus lacking.

Malignant transformation, though rare, can occur in long-standing lesions.[3] Complete surgical excision is the treatment of choice.[1] Recurrence may occur if lesions are partially excised.[1] If there are multiple tumors, surgical excision may not be practicable.[9] Radiotherapy and CO2 laser are the alternative treatment modalities available in such cases.[4]

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Calonje E. Tumours of the skin appendages. In: Burns T, Breathnach S, Cox N, Griffiths C, editors. Rook's Textbook of Dermatology. 8th ed. Oxford: Blackwell Publishing; 2010. p. 53.1-53.44.  Back to cited text no. 1
    
2.
Shelley WB, Wood MG. A zosteriform network of spiradenomas. J Am Acad Dermatol 1980;2:59-61.  Back to cited text no. 2
    
3.
Criton S, Aravindan KP. Zosteriform network of spiradenoma. Indian J Dermatol Venereol Leprol 1996;62:185-6.  Back to cited text no. 3
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4.
Gupta S, Jain VK, Singh U, Gupta S. Multiple eccrine spiradenomas in zosteriform distribution in a child. Pediatr Dermatol 2000;17:384-6.  Back to cited text no. 4
    
5.
Kersting DW, Helwig EB. Eccrine spiradenoma. AMA Arch Derm 1956;73:199-227.  Back to cited text no. 5
    
6.
Tsur H, Lipskier E, Fisher BK. Multiple linear spiradenomas. Plast Reconstr Surg 1981;68:100-2.  Back to cited text no. 6
    
7.
Ahmed TS, Priore JD, Seykora JT. Tumours of the epidermal appendages. In: Elder DE, editor. Lever's Histopathology of the Skin. 10th ed. Philadelphia: Lippincott Williams and Wilkins; 2009. p. 851-909.  Back to cited text no. 7
    
8.
Braun-Falco M, Bonel H, Ring J, Hein R. Linear spiradenoma with focal malignant transformation. J Eur Acad Dermatol Venereol 2003;17:308-12.  Back to cited text no. 8
    
9.
Bedlow AJ, Cook MG, Kurwa A. Extensive naevoid eccrine spiradenoma. Br J Dermatol 1999;140:154-7.  Back to cited text no. 9
    


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  [Figure 1], [Figure 2]



 

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