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Eccrine spiradenoma of the ear: case report
Int. J. Oral Maxillofac. Surg. 2003; 32: 662–663 doi:10.1054/ijom.2002.0412, available online at http://www.sciencedirect.com
Case Report Clinical Pathology
Eccrine spiradenoma of the ear: case report
C. Amoroso1, E. Grandi2, F. Carinci1 1
ENT Clinic and 2Department of Pathology, University of Ferrara, Italy
C. Amoroso, E. Grandi, F. Carinci: Eccrine spiradenoma of the ear: case report. Int. J. Oral Maxillofac. Surg. 2003; 32: 662–663. 2003 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. A case of eccrine spiradenoma of the left pinna is described. To the best of our knowledge, this is the first report regarding this site. The treatment was a surgical excision in safe margins. This rare benign tumour should be considered for the differential diagnosis of solitary or multiple soft tissue lesions of the head and neck.
Introduction Tumours of sweat glands represent the most frequently encountered adnexal neoplasms, with the majority being benign. Eccrine neoplasms are more numerous than those showing apocrine differentiation8. This fact probably reflects the wider distribution of normal eccrine glands, with those manufacturing apocrine secretions being relatively confined to the head, neck, and genital/ perineal skin8. Eccrine spiradenoma (ES) is defined a benign tumour of the sweat glands. Young to middle-aged adults of both sexes are most often affected. ES was first described in detail by K & H in 19566. These physicians showed a benign neoplasm of the skin with a slow, indolent course. Another 15 years passed before the first malignant transformation of ES (MES) was reported, as stated by B et al.3. Grossly, ES is a lump, firm, and ovoid; it varies in colour from tan to bluish, and is only exceptionally ulcerated8. An ES of the left pinna has not yet been described in the English literature. 0901-5027/03/000662+02 $30.00/0
Case report A 77-year-old female patient was referred for a 7-year-old painless lesion
Fig. 1. Nodule with a maximum diameter of 3 cm arising from the left pinna.
Key words: eccrine spiradenoma; pinna; adnexal tumour; ear tumour. Accepted for publication 12 March 2003
of the left pinna. It was an asymptomatic nodule with a maximum diameter of 3 cm, tender, skin coloured and not ulcerated (Fig. 1). The patient had a clinical history of hypertension, cerebrovascular accident and heart stroke, and she had neither personal nor familial history of skin tumour. The patient underwent local excision with safe margins under local anaesthesia. The postoperative course was uneventful. Histological examination of haematoxylin–eosin-stained sections showed preponderant features of ES (Fig. 2). The lesion was a circumscribed,
Fig. 2. The lesion is over the reticular dermis: there is an organoid proliferation of basaloid cells in nodular patterns, surrounded by fibrosus tissue (EE 50).
2003 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Eccrine spiradenoma of the ear ovoid, lobule within a loose fibrovascular stroma. Three types of epithelial cells form the tumour. The first type is the largest and contains a round to ovoid nucleus with evenly dispersed pale chromatin and a scant rim of cytoplasm. The second contains a round, hyperchromatic nucleus and a virtually undetectable cytoplasm. The third is a cuboidal cell that forms eccrine-type ducts. Today, the patient is well with no recurrence after 1 year follow-up. Discussion Normal skin of the auricle contains hair with sebaceous glands, eccrine sweat glands and a few apocrine ceruminous glands. Benign, appendageal tumours are derived from hair follicles, sebaceous glands, eccrine or apocrine sweat glands. ES arising from the auricle was not found in the English literature4,6,7,9,10. However, it is observed in a wide variety of anatomic locations and almost invariably presents as a solitary tender nodule of the upper body5. In a series of 134 cases of ES the majority of the lesions (97%) were solitary6. R et al. reported a case of multiple ES on the scalp mimicking multiple cylindromas and reviewed nine other cases of multiple ES9. The lesion described here was asymptomatic and soft. In contrast to the majority of other appendage tumours, ES is often tender to palpation and constitutes one of the five painful neoplasms of the skin and soft tissues (spiradenoma, angiolipoma, angioleiomyoma, post-traumatic neuroma, and
glomus tumour)8. However, in the clinical reports6,7, pain or tenderness are not always mentioned as a clinical characteristic. In a series of 49 cases of ES, pain and tenderness were mentioned in only 23% of the lesions7. This was much less common than the 91% previously reported by K & H6. Classification of these skin tumours has been based on routine histologic findings. Microscopically, there are several low-power features of ES that are part of the histological diagnosis. These include circumscription with respect to the surrounding dermis or subcutis, a clustered organoid growth pattern with rounded or ovoid cell nests, a basaloid cell constituency, and dilated lymphatic vessels in and around the neoplasm (Fig. 2). Cellular atypia, pleomorphism, necrosis, loss of nested and trabecular patterns are absent since they have more importance than mitosis in the diagnosis of MES. ES are benign and the literature reports an occasional malignant transformation within a preexisting ES1,2,5. The treatment of choice of ES is surgical excision with safe margins. This benign lesion could be considered in the differential diagnosis of solitary or multiple soft tissue lesions within other skin tumours of the head and neck.
References 1. A ZB, N AV, B K, S JK, W DC. Malignant Eccrine Spiradenoma. Am J Dermatopathol 1992: 13: 381–390. 2. B GJ. Pathology consultation. Dermal eccrine cylindroma. Ann Otol
3.
4.
5.
6.
7.
8.
9.
10.
663
Rhinol Laryngol 1989: 98: 991– 992. B AC, B TA, P C. Malignant Eccrine Spiradenoma: A previously unreported presentation and review of the literature. Am Surg 1999: 65: 236–240. G DK, MC MA, F VR. Cylindroma and Eccrine Spiradenoma coexistent in the same lesion. Arch Dermatol 1982: 118: 273– 274. G SR, S K, C E, B K, MK PH. Malignant Eccrine Spiradenoma (Spiradenocarcinoma). Am J Dermatopathol 2000: 22: 97–103. K DW, H EB. Eccrine Spiradenoma. Arch Dermatol 1956: 73: 199–227. M NC. Eccrine spiradenoma: clinical and pathologic study of 49 tumors. J Cut Pathol 1983: 10: 312–320. M RW, S PE. Cutaneous adnexal tumors: a guide to pathologic diagnosis. Chicago: ASCP Press 1991: 24–30. R P, C J, M M. Multiple eccrine spiradenoma: case report and review. J Cut Pathol 1988: 15: 226–229. Y SS, D RW, N BW. Eccrine spiradenoma: report of a case. J Oral Maxillofac Surg 1988: 46: 905–907.
Address: Francesco Carinci ENT Clinic University of Ferrara Arcispedale S. Anna Corso Giovecca, 803 44100 Ferrara Italy Tel: (+39) 0532 236383 Fax: (+39) 0532 247709 E-mail: [email protected]
Case Report Clinical Pathology
Eccrine spiradenoma of the ear: case report
C. Amoroso1, E. Grandi2, F. Carinci1 1
ENT Clinic and 2Department of Pathology, University of Ferrara, Italy
C. Amoroso, E. Grandi, F. Carinci: Eccrine spiradenoma of the ear: case report. Int. J. Oral Maxillofac. Surg. 2003; 32: 662–663. 2003 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Abstract. A case of eccrine spiradenoma of the left pinna is described. To the best of our knowledge, this is the first report regarding this site. The treatment was a surgical excision in safe margins. This rare benign tumour should be considered for the differential diagnosis of solitary or multiple soft tissue lesions of the head and neck.
Introduction Tumours of sweat glands represent the most frequently encountered adnexal neoplasms, with the majority being benign. Eccrine neoplasms are more numerous than those showing apocrine differentiation8. This fact probably reflects the wider distribution of normal eccrine glands, with those manufacturing apocrine secretions being relatively confined to the head, neck, and genital/ perineal skin8. Eccrine spiradenoma (ES) is defined a benign tumour of the sweat glands. Young to middle-aged adults of both sexes are most often affected. ES was first described in detail by K & H in 19566. These physicians showed a benign neoplasm of the skin with a slow, indolent course. Another 15 years passed before the first malignant transformation of ES (MES) was reported, as stated by B et al.3. Grossly, ES is a lump, firm, and ovoid; it varies in colour from tan to bluish, and is only exceptionally ulcerated8. An ES of the left pinna has not yet been described in the English literature. 0901-5027/03/000662+02 $30.00/0
Case report A 77-year-old female patient was referred for a 7-year-old painless lesion
Fig. 1. Nodule with a maximum diameter of 3 cm arising from the left pinna.
Key words: eccrine spiradenoma; pinna; adnexal tumour; ear tumour. Accepted for publication 12 March 2003
of the left pinna. It was an asymptomatic nodule with a maximum diameter of 3 cm, tender, skin coloured and not ulcerated (Fig. 1). The patient had a clinical history of hypertension, cerebrovascular accident and heart stroke, and she had neither personal nor familial history of skin tumour. The patient underwent local excision with safe margins under local anaesthesia. The postoperative course was uneventful. Histological examination of haematoxylin–eosin-stained sections showed preponderant features of ES (Fig. 2). The lesion was a circumscribed,
Fig. 2. The lesion is over the reticular dermis: there is an organoid proliferation of basaloid cells in nodular patterns, surrounded by fibrosus tissue (EE 50).
2003 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Eccrine spiradenoma of the ear ovoid, lobule within a loose fibrovascular stroma. Three types of epithelial cells form the tumour. The first type is the largest and contains a round to ovoid nucleus with evenly dispersed pale chromatin and a scant rim of cytoplasm. The second contains a round, hyperchromatic nucleus and a virtually undetectable cytoplasm. The third is a cuboidal cell that forms eccrine-type ducts. Today, the patient is well with no recurrence after 1 year follow-up. Discussion Normal skin of the auricle contains hair with sebaceous glands, eccrine sweat glands and a few apocrine ceruminous glands. Benign, appendageal tumours are derived from hair follicles, sebaceous glands, eccrine or apocrine sweat glands. ES arising from the auricle was not found in the English literature4,6,7,9,10. However, it is observed in a wide variety of anatomic locations and almost invariably presents as a solitary tender nodule of the upper body5. In a series of 134 cases of ES the majority of the lesions (97%) were solitary6. R et al. reported a case of multiple ES on the scalp mimicking multiple cylindromas and reviewed nine other cases of multiple ES9. The lesion described here was asymptomatic and soft. In contrast to the majority of other appendage tumours, ES is often tender to palpation and constitutes one of the five painful neoplasms of the skin and soft tissues (spiradenoma, angiolipoma, angioleiomyoma, post-traumatic neuroma, and
glomus tumour)8. However, in the clinical reports6,7, pain or tenderness are not always mentioned as a clinical characteristic. In a series of 49 cases of ES, pain and tenderness were mentioned in only 23% of the lesions7. This was much less common than the 91% previously reported by K & H6. Classification of these skin tumours has been based on routine histologic findings. Microscopically, there are several low-power features of ES that are part of the histological diagnosis. These include circumscription with respect to the surrounding dermis or subcutis, a clustered organoid growth pattern with rounded or ovoid cell nests, a basaloid cell constituency, and dilated lymphatic vessels in and around the neoplasm (Fig. 2). Cellular atypia, pleomorphism, necrosis, loss of nested and trabecular patterns are absent since they have more importance than mitosis in the diagnosis of MES. ES are benign and the literature reports an occasional malignant transformation within a preexisting ES1,2,5. The treatment of choice of ES is surgical excision with safe margins. This benign lesion could be considered in the differential diagnosis of solitary or multiple soft tissue lesions within other skin tumours of the head and neck.
References 1. A ZB, N AV, B K, S JK, W DC. Malignant Eccrine Spiradenoma. Am J Dermatopathol 1992: 13: 381–390. 2. B GJ. Pathology consultation. Dermal eccrine cylindroma. Ann Otol
3.
4.
5.
6.
7.
8.
9.
10.
663
Rhinol Laryngol 1989: 98: 991– 992. B AC, B TA, P C. Malignant Eccrine Spiradenoma: A previously unreported presentation and review of the literature. Am Surg 1999: 65: 236–240. G DK, MC MA, F VR. Cylindroma and Eccrine Spiradenoma coexistent in the same lesion. Arch Dermatol 1982: 118: 273– 274. G SR, S K, C E, B K, MK PH. Malignant Eccrine Spiradenoma (Spiradenocarcinoma). Am J Dermatopathol 2000: 22: 97–103. K DW, H EB. Eccrine Spiradenoma. Arch Dermatol 1956: 73: 199–227. M NC. Eccrine spiradenoma: clinical and pathologic study of 49 tumors. J Cut Pathol 1983: 10: 312–320. M RW, S PE. Cutaneous adnexal tumors: a guide to pathologic diagnosis. Chicago: ASCP Press 1991: 24–30. R P, C J, M M. Multiple eccrine spiradenoma: case report and review. J Cut Pathol 1988: 15: 226–229. Y SS, D RW, N BW. Eccrine spiradenoma: report of a case. J Oral Maxillofac Surg 1988: 46: 905–907.
Address: Francesco Carinci ENT Clinic University of Ferrara Arcispedale S. Anna Corso Giovecca, 803 44100 Ferrara Italy Tel: (+39) 0532 236383 Fax: (+39) 0532 247709 E-mail: [email protected]