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| Year : 2014 | Volume
: 57
| Issue : 2 | Page : 281-283 |
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| Bilateral multiple axillary apocrine hidrocystomas accompanied by apocrine hyperplasia: A rare presentation |
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Shailaja Shukla1, Karsing Patiri1, Mukta Pujani2, Singh Bhawana Komal1
1 Department of Pathology, Lady Hardinge Medical College & Smt. Sucheta Kriplani Hospital, New Delhi, India
2 Department of Pathology, Hamdard Institute of Medical Sciences and Research, New Delhi, India
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| Date of Web Publication | 19-Jun-2014 |
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 Abstract | | |
Apocrine hidrocystomas (AHC) are uncommon cystic lesions of apocrine glands and are most often solitary. They are usually found in the head and neck region. Multiple AHC have been rarely reported in the literature usually in the eyelids and face. On extensive search of literature, we came across only a single case report of multiple axillary AHC. We report a case of a 31-year-old female who presented with fullness and discomfort in bilateral axillae, so a clinical diagnosis of bilateral accessory breast was made. However on histopathological examination, it turned out to be bilateral multiple AHC associated with apocrine hyperplasia. Keywords: Apocrine hidrocystoma, apocrine hyperplasia, axilla, bilateral
How to cite this article:
Shukla S, Patiri K, Pujani M, Komal SB. Bilateral multiple axillary apocrine hidrocystomas accompanied by apocrine hyperplasia: A rare presentation. Indian J Pathol Microbiol 2014;57:281-3 |
How to cite this URL:
Shukla S, Patiri K, Pujani M, Komal SB. Bilateral multiple axillary apocrine hidrocystomas accompanied by apocrine hyperplasia: A rare presentation. Indian J Pathol Microbiol [serial online] 2014 [cited 2023 Sep 26];57:281-3. Available from: https://www.ijpmonline.org/text.asp?2014/57/2/281/134712 |
| Introduction | |  |
Apocrine hidrocystomas (AHC) or cystadenomas are benign adenomatous cystic tumors derived from the secretory portion of apocrine sweat glands, first described by Mehregan [1] in 1964 as a benign neoplasm located on the face. The lesions are commonly seen in the head and neck region especially around the eye (lateral to the outer canthus). Other sites of occurrence are ears, scalp, chest and shoulders. AHC is an uncommon cystic lesion and is most often solitary. However, multiple lesions have also been documented. [2],[3],[4],[5],[6] Although axilla is rich in apocrine glands, AHC are rarely reported in this region. [5] We hereby report a rare case of bilateral multiple axillary AHC accompanied by apocrine hyperplasia in a 31 year old female with a clinical diagnosis of bilateral accessory breast.
| Case report | | |
A 31-year-old female patient complained of fullness in both the axillae for 12 years with increase in discomfort for 3 years. On physical examination 8 cm × 7 cm mobile, tender masses were present in both the axillae.
A clinical diagnosis of bilateral accessory breasts was made. Ultrasonography revealed fibro-fatty glandular appearing tissue suggestive of normal breast tissue. Patient was advised fine needle aspiration cytology (FNAC) examination. FNAC of both axillary masses yielded thick yellowish greasy aspirate, which on microscopic examination showed mainly lipoproteinaceous material along with occasional cluster of columnar epithelial cells and adipose tissue fragments. No mammary ductal epithelial cells were seen. In view of inconclusive FNAC, both axillary masses were resected and sent for histopathological examination.
Both axillary masses with overlying skin eclipse measured 7 cm × 5 cm × 4 cm. Multiple small cysts 0.2-0.6 cm in diameter filled with thick yellowish greasy fluid were seen in the subcutaneous tissue. Microscopic examination of both axillary masses showed lesions essentially in the dermis and subcutis and not in the breast parenchyma. Multiple cystic spaces of variable size lined by single or double layer of cuboidal to columnar epithelium above an outer myoepithelial cell layer were seen representing dilated apocrine ducts. The lining epithelium in larger dilated ducts was flattened. Few cysts showed papillary projections into their lumina. Many of these cystic spaces contained eosinophilic secretions (decapitation secretion) [Figure 1] and [Figure 2]. Hyperplastic lobules of benign apocrine glands were also seen in adjoining areas [Figure 3] . No ductal or lobular breast tissue was seen. Immunohistochemistry revealed smooth muscle actin positivity in myoepithelial cell layer while S-100 turned out to be negative. | Figure 1: Photomicrograph showing multiple cystic spaces in apocrine hidrocystoma (H and E, ×100)
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 | Figure 2: Photomicrograph showing characteristic decapitation secretion in lining of apocrine hidrocystoma (H and E, ×400)
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 | Figure 3: Photomicrograph showing apocrine gland hyperplasia (H and E, ×100)
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A final diagnosis of bilateral multiple axillary AHC accompanied by apocrine hyperplasia was rendered.
| Discussion | | |
AHC are benign cystic tumors of apocrine sweat glands. They are usually asymptomatic, solitary, dome shaped bluish black to light brown cystic lesions. They are seen in middle aged to elderly patients with equal frequency in both male and female patients. Anzai et al. [7] reviewed 167 cases of AHC and found that only 7 (4.3%) cases were reported in the axilla. AHC are usually solitary lesions multiple AHC are relatively uncommon and are more commonly seen in head and neck region and those on the face are sometimes referred to as Robinsons variant. [2] Multiple AHC in axilla has been very rarely reported in the literature. [3]
A peculiar feature of AHC is that they seldom occur in areas rich in apocrine glands. [5] Histologically AHC are characterized by a unilocular or multilocular cystic space in the dermis. The cyst wall is lined by outer layer of myoepithelial cells and inner layer of tall columnar or cuboidal cells. These cysts are filled with eosinophillic material known as decapitation secretion which is the hallmark of AHC and not seen in eccrine variant. Also papillary projections growing into the central cavity are seen. AHC show variable S100 positivity. [2] Verma [5] and Warrier et al. [6] have reported S-100 positivity in AHC, however, S-100 was negative in our case.
Multiple AHC may be associated with two rare inherited syndromes: Schopf-Schulz-Passarge syndrome and Goltz-Gorlin syndrome More Details. Schopf-Schulz-Passarge syndrome is autosomal recessive presenting with multiple apocrine hidradenomas associated with palmoplantar hyperkeratosis, hypotrichosis, onychodystrophia, and hypodontia. Goltz-Gorlin syndrome has an association of multiple AHC with bilateral keratoconus, esophageal papillomatosis and hiatus hernia. However in the present case, none of these features were present.
Multiple AHC, although uncommon, should always be biopsied to confirm the diagnosis. [8] The various treatment options available for AHC are simple excision, electrodessication, [8] anticholinergic creams, trichloroacetate acid, CO 2 laser, 1450 nm diode laser. [4]
Multiple AHC in both axilla has been described only in a single case report by Obaidat and Vani [3] where it was accompanied by benign apocrine hyperplasia similar to the present case.
| References | | |
| 1. | Mehregan AH. Apocrine cystadenoma; a clinicopathologic study with special reference to the pigmented variety. Arch Dermatol 1964;90:274-9. 
[PUBMED] |
| 2. | Calonje E, Lazar A, Brenn T, McKee PH. Tumors of the sweat glands. In: Calonje E, Lazar A, Brenn T, McKee PH, editors. Pathology of the Skin. 4 th ed. St. Louis: Elsevier Mosby; 2012. p. 1508-70.
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| 3. | Obaidat NA, Ghazarian DM. Bilateral multiple axillary apocrine hidrocystomas associated with benign apocrine hyperplasia. J Clin Pathol 2006;59:779.
[PUBMED] |
| 4. | Vani D, Dayananda TR, Shashidhar HB, Bharathi M, Kumar HRS, Ravikumar V. Multiple apocrine hidrocystomas: A case report. J Clin Diagn Res 2013;7:171-2.
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| 5. | Verma SB. Multiple apocrine hidrocystomas: A confusing clinical diagnosis. An Bras Dermatol 2010;85:260-3.
[PUBMED] |
| 6. | Warrier S, Mohammed K, Safia B. Multiple apocrine hidrocystoma - An uncommon presentation. Indian J Dermatol Venereol Leprol 2003;69:64-5.
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| 7. | Anzai S, Goto M, Fujiwara S, Da T. Apocrine hidrocystoma: A case report and analysis of 167 Japanese cases. Int J Dermatol 2005;44:702-3.
[PUBMED] |
| 8. | Sarabi K, Khachemoune A. Hidrocystomas - A brief review. MedGenMed 2006;8:57.
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Correspondence Address:
Mukta Pujani
Associate Professor, Department of Pathology, Hamdard Institute of Medical Sciences and Research, New Delhi - 110 062
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0377-4929.134712
[Figure 1], [Figure 2], [Figure 3] |
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