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Case of recurrence of spiradenoma in palpebral conjunctiva
© Oie et al.; licensee BioMed Central Ltd. 2014
Received: 4 November 2013
Accepted: 18 June 2014
Published: 27 June 2014
To report a rare case of a recurrence of spiradenoma that developed in the upper eyelid.
A 49-year-old woman who had a second recurrence of a tumor in the right palpebral conjunctiva underwent local resection of the lesion with adjunctive cryotherapy to the surgical site. The tumor consisted of smooth, round to oval nodular lesions approximately 1–3 mm in size with enlarged blood vessels. Histopathologically, the solid and well-circumscribed nodule was located beneath the conjunctival epithelium. It was composed of cells with slightly basophilic-to-clear cytoplasm and round-to-oval nuclei arranged in a trabecular pattern. Periodic acid-Schiff stain was positive in the cytoplasm, and the staining disappeared after digesting by diastase. Many cells in mitosis were observed throughout the tumor but no necrotic cells. Immunohistochemistry showed that the Ki-67 labeling index was 12%. From these findings, we diagnosed this tumor as a recurrence of the spiradenoma. There has been no recurrence and no signs of malignancy in the 6 months after the surgical excision.
Our findings indicate that a spiradenoma should be completely excised surgically because of malignant transformation after repeated recurrences.
Tumors arising from the sweat glands of the eyelids are uncommon, and the differences in their structure have given rise to much confusion [1–4]. A spiradenoma is a type of sweat gland tumor which has a comparatively good clinically course although it appears to be malignant [1, 5]. A spiradenoma involving the eye or eyelid is rare [2, 3]. We describe our findings in a patient with a recurrence of spiradenoma that developed in the upper eyelid.
A spiradenoma is a tumor of the eccrine sweat glands that arises as a solitary, intradermal, and painful nodule on the chest or face and most frequently in early adulthood. Occasionally, there may be multiple tumors [1, 2, 5]. It has been well-characterized in the dermatological literature but not in the ophthalmological literature probably because it is uncommon in the eyelid [2, 3]. To the best of our knowledge, there has been no report of a primary spiradenoma developing in the palpebral conjunctiva. In our case, the tumor was considered to be a secondary spread of the original tumor at the primary site in the palpebral conjunctiva of the eyelid. The majority of spiradenoma have a benign course , and local recurrences have not been reported. Considering our lesion was a local recurrence because of an inadequate surgical removal, we treated the site of the tumor excision with adjunctive cryotherapy to prevent further recurrences.
Immunohistochemical study demonstrated that cytokeratin expression in eccrine spiradenoma was compared with expression in normal eccrine glands and the tubular structures are CEA positive [6, 7]. In the present case, the CAM5.2, and CEA were negative in the tumor cells and the EMA was weakly positive. The immunohistochemical results suggest that the tumor may inadequate differentiate towards both the ductal and secretory segments of the eccrine sweat glands.
The differential diagnosis includes cylindroma, hidradenoma and sebaceoma. Dermal cylindroma and spiradenoma in the same patient or in the same tumor mass are well-represented in the literature [3, 8]. Cylindroma differs from spiradenoma by displaying islands of cells rimmed by a basement membrane matrix and the presence of few lymphocytes . Immunohistochemical reactivity for CAM5.2, CEA, EMA, S-100, and vimentin in hidradenoma is characteristic . Because of the PAS-positivity and adipophilin expression, our tumor mimicked a low grade malignant tumor including sebaceous differentiations. Sebaceous differentiations is defined as the presence of multiple cytoplasmic fat vacuoles and the positive staining of tumor cells for EMA and negative staining for CEA [10, 11].
Malignant changing of spiradenomas is unusual, and the histology of malignant spiradenomas resembles squamous metaplasia or very poorly differentiated pseudosarcomatous spindle cell elements . In cases of a malignant transformation, the patient might die of systemic metastases several months after the diagnosis . Because our tumor had atypical cells with mitotic activity increasing (from 0.3/10 to 5.5/10 HPF) and had a trabecular or alveolar pattern, it appeared to have a more aggressive pattern. The Ki-67 labeling index that is related to the prognostic features was 12% in our case. The tumor contained PAS-positive material, which was identified as glycogen by its breakdown by diastase. Thus, we diagnosed the tumor as a recurrence spiradenoma from the clinical and pathological features of the tumor and might be progressing to malignant changes.
We describe a recurrence of spiradenoma due to an inadequate surgical excision at a rare site, the palpebral conjunctiva. Although a spiradenoma is usually benign, it may transform to a malignant spiradenoma, and thus we are following this patient carefully.
The study was approved by the Ethics Committees of Gifu University Hospital. Written informed consent was obtained from the patient for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Series Editor of this journal.
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