Indian Journal of Dermatology
DERMA-QUEST
Year
: 2006  |  Volume : 51  |  Issue : 1  |  Page : 60--61

Mutiple asymptomatic papules on face showing summer exacerbation


Nilay Kanti Das, A Coondoo, Raghubir Banerjee, Swati Worah 
 Department of Dermatology, RK Mission Seva Pratishthan, India

Correspondence Address:
Nilay Kanti Das
Devitala Road, Majerpara, Ishapore, North 24 Paraganas - 743 144
India




How to cite this article:
Das NK, Coondoo A, Banerjee R, Worah S. Mutiple asymptomatic papules on face showing summer exacerbation.Indian J Dermatol 2006;51:60-61


How to cite this URL:
Das NK, Coondoo A, Banerjee R, Worah S. Mutiple asymptomatic papules on face showing summer exacerbation. Indian J Dermatol [serial online] 2006 [cited 2020 Sep 24 ];51:60-61
Available from: http://www.e-ijd.org/text.asp?2006/51/1/60/25204


Full Text

 Case Report



A 45-year-old lady presented with multiple asymptomatic eruptions on face, present for the last 3 years and gradually increasing in number. The rashes exhibited seasonal variation becoming more prominent in the summer but flattening out in winter. Working in hot environment (especially, cooking) also increased size of the lesions. She gave no family history of the disorder neither was she suffering from any other systemic ailment.

On examination there were numerous skin-colored translucent papules, ranging in size from 1mm to 3mm on the nose, both cheeks, forehead and on and around the naso-labial furrow. In addition a few small vesicles of size 3-4 mm were also noted on the nose, which showed clear watery fluid on pricking with a needle [Figure 1]. Hairs, nails and teeth were found to be normal. CBC was within normal limits, with the blood urea 22 mg/dl and creatinine 0.9 mg/dl. LFT was normal and Chest X-ray showed no abnormality.

Histopathology of the lesion showed a thin flattened epidermal lining to which a few hair follicles are attached [Figure 2]. A unilocular dermal cyst is seen in intimate association with eccrine structures. The cyst is lined by double layer of cuboidal epithelium [Figure 3]. Faint eosinophilic material is seen in its lumen. No evidence of decapitation secretion of the cells lining the cyst was found even on serial sectioning. PAS staining showed no evidence of any PAS positive granules in the lining epithelium of the cyst.

 Questions



Q1. What is the diagnosis?

Q2. What is the closest histo-pathological entity that mimics this condition?

Q3. Could immunohistochemistry play some role in further analysis of this disorder?

Q4. How is one supposed to manage this condition?

 Answer to Derma-Quest



Ans 1. Eccrine hidrocystoma ("Classic" Robinson type)

This entity was first described by Robinson in 1893 when he noted numerous minute vesicular eruption on the face of middle aged women working in hot environment.[1] The present case typically mimics the classical manifestation of eccrine hidrocystoma showing summer exacerbation which served as the most important diagnostic clue in our case. The clinical findings along with the histopathology showing a unilocular cyst lined by two layers of cuboidal epithelium with no evidence of decapitation secretion or PAS positive granules in the lining epithelium is diagnostic of the condition. It should be noted that an eccrine hidrocystoma need not always be multiple - it may also present as solitary (or only a few) lesion, as described by Smith and Chernosky.[2] It is proposed that eccrine hidrocystoma represents mature, deformed eccrine gland units where occlusion of the intradermal portion of the eccrine duct leads to dilatation of the ducts.[2] Much of the light on the pathogenesis of this disorder was shed by the study of S-100 protein, which is found exclusively in the secretory portion of eccrine glands and not in the any other parts of eccrine glands or at any levels of the structure of apocrine glands.[3] It was found that S-100 protein was absent in the "classic", multiple-lesion type of eccrine hidrocystoma but detected in the cells of solitary eccrine hidrocystoma suggesting that the former arose from the eccrine ductal cells and the latter from the secretory portion of the eccrine gland.[3],[4]

Ans 2. Apocrine hidrocystoma

This condition, which is regarded as adenomatous proliferation of apocrine unit, is the closest differential diagnosis of eccrine hidrocystoma. Apocrine hidrocystoma differs from its eccrine counterpart by the presence of decapitation secretion and PAS positive granules in the cells lining the cyst and presence of myoepithelial cells surrounding the cyst.[5] There are views questioning the existence of eccrine hidrocystoma[6] and absence of decapitation secretion is being attributed to the increased intraluminal pressure. But studies on the keratin expression clearly demarcates the two condition as separate pathological entity.[7]

Ans 3. Immuno-histochemistry, though not routinely used, may be utilized for differentiating eccrine from apocrine hidrocystoma. Staining with monoclonal antibody IKH-4 points towards eccrine origin;[8] whereas presence of gross cystic disease fluid proteins 15 and 24 (GCDFP-15, -24) are more suggestive of apocrine differentiation.[9],[10] Immuno-histochemical analysis of lysozymes also helps in determining the direction of differentiation of these tumors, with positive staining indicating apocrine origin.[11]

Ans 4. Eccrine hidrocystoma, if solitary, does not pose much of a problem since it can surgically excised with good cosmetic result. But the present case having numerous lesions, surgery was out of question. The few vesicles on the nose were punctured with needle under aseptic condition and then topical application of 1% atropine sulfate in aqueous solution three times daily was tried along with repeated cold compress with good results. The patient was satisfied with the decrease in the size and number of the lesion.

Current literature also recommends the use of atropine sulfate for multiple lesions though the results are not always found to be satisfactory.[12],[13] Among other potential therapeutic agents, Botulinum toxin (type A) has proved to be successful by blocking the cholinergic sweat secretion.[14] Lasers are also being tried in the treatment of this condition with good results obtained from pulsed-dye laser[15] and argon laser.[16] Nevertheless none of the treatment options are yet been standardized in this rare condition so it is recommended that one must individualize the therapy according the response of the patient.

References

1Robinson AR. Hidrocystoma. J Cutan Gen-Urin Dis 1893:11:293-303
2Smith JD, Chernosky ME. Hidrocystomas. Arch Dermatol 1973: 108;676-9
3Tokura Y, Takigawa M, Inoue K, Matsumoto K, Yamada M. S-100 protein-positive cells in hidrocystomas. J Cutan Pathol 1986;13:102-10.
4Kato N, Ueno H. Eccrine hidrocystoma: two cases of Robinson and Smith types. J Dermatol 1992;19:493-7.
5Lever WF, Schaumburg-Lever G. Histopathology of the skin. 7th ed. Philadelphia: J. B. Lippincott Company, 1990: 599-601
6Simon RS, Sanches Yus E. Does eccrine hidrocystoma exist? J Cutan Pathol 1998; 25:182-4.
7de Viragh PA, Szeimies RM, Eckert F. Apocrine cystadenoma, apocrine hidrocystoma and eccrine hidrocystoma: three distinct tumors defined by expression of keratins and human milk fat globulin 1. J Cutan Pathol 1997;24:249-55.
8Ishihara M, Mehregan DR, Hashimoto K, Yotsumoto S, Toi Y, Pietruk T, Mehregan AH, Mehregan DA. Staining of eccrine and apocrine neoplasms and metastatic adenocarcinoma with IKH-4, a monoclonal antibody specific for the eccrine gland. J Cutan Pathol 1998;25:100-5.
9Mazoujian G, Margolis R. Immunohistochemistry of gross cystic disease fluid protein (GCDFP-15) in 65 benign sweat gland tumors of the skin. Am J Dermatopathol 1988;10:28-35.
10Mazoujian G. Immunohistochemistry of GCDFP-24 and zinc alpha2 glycoprotein in benign sweat gland tumors. Am J Dermatopathol 1990; 12:452-7.
11Katsumata M, Ezoe K. Immunohistochemical study of lysozyme in various benign sweat apparatus tumors. J Dermatol 1990; 17: 307-11.
12Lee MR, Ryman W. Multiple eccrine hidrocystomas. Australas J Dermatol 2004;45:178-80.
13Khunger N, Mishra S, Jain RK, Saxena S. Multiple eccrine hidrocystomas: Report of two cases treated unsuccessfully with atropine ointment. Indian J Dermatol Venereol Leprol 2004;70:367-9.
14Blugerman G, Schavelzon D, D'Angelo S. Multiple eccrine hidrocystomas: A new therapeutic option with botulinum toxin. Dermatol Surg 2003;29:557-9.
15Tanzi E, Alster TS. Pulsed dye laser treatment of multiple eccrine hidrocystomas: A novel approach. Dermatol Surg 2001;27:898-900.
16Baum U, Konigsdorffer E, Bocker T, Strobel J, Wollina U. Argon laser therapy of multiple eccrine cysts of sweat gland efferent ducts (eccrine hidrocystomas). Klin Monatsbl Augenheilkd. 1996;209:249-51.