Letters to the Editor

284 Indian Dermatology Online Journal | Volume 8 | Issue 4 | July‑August 2017

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Choudhary M, Sharma I, Kaur M, Dalal V, Singh A. Immunohistochemical expression of wt-1 helps to differentiate cutaneous vascular tumors from vascular malformations. Indian Dermatol Online J 2017;8:282-4.

Received: June, 2016. Accepted: August, 2016.

© 2017 Indian Dermatology Online Journal | Published by Wolters Kluwer - Medknow

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DOI:10.4103/2229-5178.209607

Conflicts of interestThere are no conflicts of interest.

Manisha Choudhary, Ira Sharma, Manveen Kaur, Varsha Dalal1, Avninder Singh

Department of Histopathology, 1Senior Resident, National Institute of Pathology, ICMR, Safdarjung Hospital Campus, New Delhi, India

Address for correspondence: Dr. Avninder Singh,

National Institute of Pathology, ICMR, Safdarjung Hospital Campus, New Delhi ‑ 110 029, India.

E‑mail: avninders@hotmail.com

References1. Wassef M, Blei F, Adams D, Alomari A, Baselga E, Berenstein A,

et al. Vascular Anomalies Classification: Recommendations From

the International Society for the Study of Vascular Anomalies. Pediatrics 2015;136:e203‑14.

2. Hanson J, Gorman J, Reese J, Frazier G. Regulation of vascular endothelial growth factor, VEGF, gene promoter by the tumor suppressor, WT1. Front Biosci 2007;12:2279‑90.

3. Trindade F, Tellechia O, Torrelo A, Requena L, Colmenero I. Wilms tumor 1 expression in vascular neoplasms and vascular malformations. Am J Dermatopathol 2011;33:569‑72.

4. Galfione SK, Ro JY, Ayala AG, Ge Y. Diagnostic utility of WT‑1 cytoplasmic stain in variety of vascular lesions. Int J Exp Clin Pathol 2014;7:2536‑43.

5. Timar J, Meszaros L, Orosz Z, Albini A and Raso E. Wt1 expression in angiogenic tumours of the skin. Histopathology 2005;47:67‑73.

6. Lawley LP, Cerimele F, Weiss SW, North P, Cohen C, Kozakewich HP, et al. Expression of Wilms tumor 1 gene distinguishes vascular malformations from proliferative endothelial lesions. Arch Dermatol 2005;141:1297‑300.

7. Al Dhaybi R, Powell J, Mc Cuaig C, Kokta V. Differentiation of vascular tumors from vascular malformations by expression of Wilms tumor 1 gene: Evaluation of 126 cases. J Am Acad Dermatol 2010;63:1052‑7.

Figure 2: WT-1 expression in (a) portwine stain, (b) angiokeratoma, (c) lymphangioma, (d) and arteriovenous malformation (×200)

dc

ba

A Case of Eruptive Syringoma Mimicking Plane Warts

Sir,Syringoma is a benign tumour arising from the intraepidermal portion of sweat ducts, affectingapproximately0.6% of the general population, of which the generalized eruptive form is a rare clinical variant.[1]

A 26‑year‑old female presented with multiple asymptomatic, skin‑colored lesions of 10 years duration which were progressive in nature. Lesions started from the neck and spread to the chest up to the suprasternal area. Patient was not on any medication. Family history was unremarkable. Cutaneous examination revealed multiple skin‑colored papules over the chest, neck [Figure 1], and infraorbital

region [Figure 2], varying from 1 to 4 mm in size. Few discrete papules were also present over both the arms and abdomen. No mucus membrane, nails, scalp, or palmoplantar involvement was seen. Systemic examination was unremarkable. Biopsy was taken from one of the papules over the chest, withplane warts, acrokeratosis verruciformis, and syringoma as clinical differentials. The section showed histology of a benign adnexal tumour composed of small island and duct‑like structure embedded in collagen in the upper dermis. The overlying epidermis was unremarkable. The tumour cells were monomorphic, having round‑to‑vesicular nuclei and eosinophilic cytoplasm

Letters to the Editor

285Indian Dermatology Online Journal | Volume 8 | Issue 4 | July‑August 2017

[Figure 3]. Small ducts lined with a double row of flattened epithelial cells, with outer layer extending into the surrounding stroma, forming a comma‑like projection (tails), and giving them the appearance of tadpole [Figure 4] were seen. Findings were consistent with the clinical diagnosis of syringoma. Patient was advised for cryotherapy with liquid nitrogen. After 6 to 8 sittings, only minor improvement was seen. Patient is still under follow up. As the patient was in child bearing age group, wedid notadminister isotretinoin.

The word syringoma is derived from the Greek word syrinx meaning pipe or tube. It refers to a group of benign adnexal neoplasms with a tendency to ductal (acrosyringeal) differentiation.[2] Based on Friedman and Butler’s classification scheme, fourvariants of syringoma are recognized, namely, localized, associated with Down syndrome, generalized form having multiple and eruptive syringomas, and a familial form. Estrogen and progesterone receptors have been detected in histochemical studies, explaining its occurrence more commonly in females and during puberty.[3]

There are reports in literature where syringoma is seen to be associated with Down’s syndrome, diabetes mellitus, milium, sarcoidosis, elevated serum carcinoembryonic antigen (CEA), psychiatric disorders, and with heat stimuli.[4]

Classically, syringoma presents with multiple skin‑coloredto slightly yellowish papules which are dome‑shaped to flat with characteristic angulated borders. The papules are 1–5 mm in size over the periorbital area, particularly around the lower eyelid. In the variant of eruptive syringoma, successive crops of disseminated multiple lesions develops involving anterior chest, neck, upper arm, penis, vulva, upper abdomen, axillae, and periumbilical region. The eruptions are generally asymptomatic, although pruritus has been reported in some cases.[5]

Recent studies suggest that eruptive syringoma is not a true neoplasm but a reactive process in hyperplastic eccrine

Figure 1: Multiple skin-colored papules over the chest and neck Figure 2: Multiple skin-coloured papules over infraorbital region

duct, resulting from a previous cutaneous inflammation,[4] although its pathophysiology is not yet completely understood.

Histochemical studies have shown that all eccrine type of enzymes and glycogen are present in the tumor cells of syringoma. Eccrine‑specific monoclonal antibody positively stains syringoma lesions. Hence, formerly thought to be of mixed origin, now it is considered to be a benign appendageal tumor of intraepidermal eccrine sweat duct.[6]

Clinically, syringoma may be mistaken with lichen planus, plane warts, papular mucinosis, xanthoma disseminatum, mastocytosis, acne vulgaris, sebaceous hyperplasia, milia, urticaria pigmentosa, hidrocystoma, and trichoepithelioma.[4] Morphology of lesions in our patient was mimicking plane warts in the form of multiple skin‑coloured flat papules.

Syringomas demonstrate distinctive histopathological features. Dermis shows numerous small ducts lined with a double row of flattened epithelial cells. Often the outer layer extends into the surrounding stroma, forming a comma‑like projection (tails) of epithelial cells giving them the appearance of tadpole,[6] as was seen in our case. Ductal lumina are filled with an amorphous, periodic, acid‑Schiff‑positive material.

Therapy for syringomas is usually unsatisfactory as they are embedded within the dermis; complete removal is often unsuccessful and recurrences are common.Various treatment modalities include dermabrasion, excision, cryosurgery, electrocautery, electrodesiccation, and chemical peeling. Successful treatment of facial syringomas with carbon dioxide laser has been reported.[7] Oral isotretinoin and topical tretinoin and adapalene have also been used.Spontaneous involution of the lesions has also been reported.

Declaration of patient consentThe 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

Letters to the Editor

286 Indian Dermatology Online Journal | Volume 8 | Issue 4 | July‑August 2017

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-ShareAlike 3.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Nair PA, Singhal RR, Gandhi SS. A case of eruptive syringoma mimicking plane warts. Indian Dermatol Online J 2017;8:284-6.

Received: May, 2016. Accepted: July, 2016.

© 2017 Indian Dermatology Online Journal | Published by Wolters Kluwer - Medknow

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 sponsorshipNil.

Conflicts of interestThere are no conflicts of interest.

Pragya A. Nair, Rochit R. Singhal, Shailee S.Gandhi

Department of Dermatology and Venereology, Pramukhswami Medical College, Karamsad, Gujarat, India

Address for correspondence: Dr. Pragya Ashok Nair,

Department of Dermatology and Venereology, Pramukhswami Medical College, Karamsad ‑ 388 325, Gujarat, India.

E‑mail: drpragash2000@yahoo.com

References1. Guitar J, Rosenbaum MM, Requena L. ‘Eruptive syringoma’:

Amisnomer for a reactive eccrine gland ductal proliferation. Cutan J Pathol 2003;30:202‑5.

2. Kumar YH, Keethi S. Eruptive syringoma‑A great mimicker: An uncommon presentation in males. Int J Health Allied Sci 2014;3:270‑2.

3. Timpanidis PC, Lakhani SR, Groves RW. Progesterone receptor‑positive eruptive syringoma associated with diabetes. J Am Acad Dermatol 2003;48:103‑4.

Figure 4: Duct with eosinophilic cuticle lined by elongated cells giving a tadpole like appearance in the dermis (H and E, ×40)

Figure 3: Unremarkable epidermis with monomorphic cells, having round to vesicular nuclei and eosinophilic cytoplasm and duct-like structure embedded in collagen of upper dermis (H and E, ×4)

4. Seirafi HH, Akhyani M, Naraghi ZS, Mansoori P, Dehkordi HS, Taheri A, et al. Eruptive syringomas. Dermatol Online J 2005;11:13.

5. Verma SB. An unusual case of eruptive syringomas presenting as itchy symmetrical lesions on both forearms in a patient of hyperkeratotic eczema. Indian Dermatol Online J2011;2:104‑6.

6. Hashimoto K, DiBella RJ, Borsuk GM, Lever WF. Eruptive hidradenoma and syringoma. Histological, histochemical, and electron microscopic studies. Arch Dermatol 1967;96:500‑19.

7. Cho SB, Kim HJ, Noh S, Lee SJ, Kim YK, Lee JH. Treatment of syringoma using an ablative 10,600‑nm carbon dioxide fractional laser: Aprospective analysis of 35 patients. DermatolSurg 2011;37:433‑8.

Access this article online

Website:www.idoj.in

Quick Response Code

DOI:10.4103/2229-5178.209603