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Only one case of clear-cell syringoma has been reported in the United States, whereas six cases have already been reported in Japan, and ours described herein constitute an eighth and ninth. Eight of the total of nine cases of clear-cell syringoma were associated with diabetes mellitus. In this report, we summarize these nine cases and emphasize the close relationship between clear-cell syringoma and diabetes mellitus.
We report a case of collagenous spherulosis (CS) incidentally found in a chondroid syringoma of the facial skin. The lesion was studied by routine light microscopy, special stains, immunohistochemical methods, x-ray spectrophotometry, and electron microscopy. Light microscopy revealed solitary and confluent eosinophilic globules with radiating fibrillary structures within and around the lumina of the tubuloglandular components of chondroid syringoma. The fibrillary structures stained strongly for collagen and reticulin and less intensely for acidic mucopolysaccharides. Immunohistochemically, the globules were focally positive only for collagen type IV. Electron microscopy revealed radiating collagen fibers surrounded by basal lamina-like material. No inorganic crystals were identified by x-ray spectrophotometry. We conclude that (a) CS is not specific to breast but also occurs in chondroid syringomas; (b) the term collagenous spherulosis is appropriate because collagen fibers were demonstrated histochemically and ultrastructurally in the spherules; (c) CS appears to be associated with tubular epithelial structures; (d) there was no immunohistochemical evidence of myoepithelial differentiation. The etiology and significance of CS remain obscure.
The distribution of cytokeratin (CK) polypeptides expressed in syringomas (12 cases) was compared with that in normal eccrine sweat ducts using immunohistochemical techniques on paraffin-embedded tissue. Intradermal and intraepidermal segments of the eccrine duct showed reactivity with an antibody to CK1/5/10/11 in all cell layers, whereas CK19 expression was restricted to the luminal cell layer. CK14 was expressed in all cells of the eccrine duct except for the peripheral cells of the intraepidermal duct. Expression of CK5/6 was seen in the basal cells of the dermal duct and of the lower intraepidermal duct (sweat duct ridge) exclusively. Reactivity with an antibody to CK1 was found in the intermediate cells of the uppermost part of the eccrine dermal duct. In addition, this antibody gave a strong staining of the peripheral cells of the intraepidermal duct, leaving basal cells of the sweat duct ridge and luminal cells unstained. In syringoma, CK distribution was essentially comparable with that found in the uppermost part of the dermal duct and in the sweat duct ridge. Namely, ductal luminal cells expressed CK1/5/10/11, CK19, and variably CK14. Intermediate cells of ductal structures and solid nests were homogeneously stained by antibodies to CK1 and CK1/5/10/11, whereas CK14 was expressed heterogeneously. The basal or outermost layer of ductal structures and solid nests was reactive with antibodies to CK1/5/10/11, CK5/6, and CK14. With regard to CK expression, the results indicate that syringoma represents a tumor differentiating toward both the uppermost part of the dermal duct and the lower intraepidermal duct (sweat duct ridge) of the eccrine sweat gland.
A 6-year-old girl with Down's syndrome presented milialike whitish small papules on her hands and feet and periorbital syringoma. Histopathological examination of the hand lesion revealed small localized calcium deposits and syringoma in the adjacent upper dermis. This is a very rare but typical case of calcinosis cutis with syringoma in a patient with Down's syndrome.
A case of syringoma of the vulva causing severe vulval pruritus is presented. The patient was thought to have either vulval dystrophy or nonspecific inflammation. Unexpectantly the diagnosis of syringoma was made on pathological examination of biopsy specimens. This unusual case is presented as the presenting symptoms and macroscopic appearance was atypical for syringoma.
We report three cases of clinically unapparent syringoma found incidentally on horizontal frozen tissue sections from patients undergoing Mohs micrographic excision of basal cell carcinoma (BCC). In the first case, the syringoma and BCC were admixed; in the other two cases, they were seen in close approximation to one another. In all cases, the tumors were detected on the faces of elderly women. The potential existence of a benign syringoma in association with BCC should be borne in mind, particularly in connection with Mohs micrographic excision. These tumors must be distinguished from one another in order to avoid excessive surgery beyond adequate resection of the basal cell carcinoma.
The expression of Ca 15-3, KA-93, Ca 19-9, CD44 and BM-1 in normal skin and chondroid syringoma was investigated immunohistochemically using antibodies to these antigens. In the normal skin, the eccrine ducts and/or secretory elements were positive for all these antigens. On the other hand, the apocrine ducts and/or secretory elements were positive for the antibodies to Ca 15-3, KA-93 and CD44. In chondroid syringoma, the luminal cells of tubuloglandular structures were positive for the antibodies to Ca 15-3 and BM-1, and partly positive for those to KA-93 and Ca 19-9. The basal cells of the tubular structures and solid nests were positive only for the antibody to CD44. Stromal cells in the myxoid area were positive for the antibodies to KA-93 and CD44, and the chondroid matrix was positive for the antibody to BM-1. It is suggested that chondroid syringoma might originate from, or differentiate into the ducts and/or secretory elements of the eccrine sweat glands. In addition, the significance of the expression of BM-1 in the chondroid matrix is discussed.
Chondroid syringoma is a benign skin tumour characterized by several histological aspects similar to salivary gland adenomas. It generally affects the head and neck, mainly in the 6th and 7th decade of life. Its incidence in males is twice as high as in females. The neoplasm is usually an asymptomatic subcutaneous swelling that patients want removed for aesthetic reasons. Excision is the elective treatment. A few cases of malignant chondroid syringoma, however, have been reported. The neoplasm tends to produce metastases to both the regional and distant lymph nodes, causing the death of the patient. In these cases, radiation therapy follows the surgical excision. The authors report the case of a woman with chondroid syringoma located in the occipital region of the scalp. After a period of slow growth, the neoplasm suddenly increased in size. The patient asked for it to be removed out of concern for the concomitant hair loss.
The authors report a quite unusual case in which an eruption of syringomas, mongolism, Marfan's disease and Ehlers-Danlos' disease were associated. They emphasize the originality of the intricate appearance of syringomas on the ichthyosiform skin due to mongolism. They also stress the importance of the dysplasia of the connective tissue demonstrated by electron microscopy and the cause-effect relationships between the dysplasia of the connective tissue and the syringomas, the former accounting for the latter.
In summary, chondroid syringoma is a rare sweat gland neoplasm characterized by its slow growth and unpredictable growth phase. When malignant, it has a significant potential for recurrence, metastases, and even death. Due to the rarity of this tumor, an accurate preoperative diagnosis is difficult. Therefore, it is important for the clinician to have a high degree of suspicion for chondroid syringoma when considering the differential diagnosis of solid cutaneous nodules. A patient has been presented with a chondroid syringoma with atypical features, but no unequivocal evidence of malignancy. This poses a therapeutic dilemma. At the present time, the best therapeutic option appears to be accurate diagnosis, wide local excision, and close follow-up. Further research will elucidate the role of adjunctive treatment modalities.
A case of unilateral naevoid syringoma is reported. Syringoma is an adnexal benign neoplasia originating from the ducts of eccrine sweat glands. Localized (neck, anterior portion of the chest, genitalia, fingers) and generalized forms are known, but unilateral naevoid syringoma is quite uncommon. The existence of this variety can be explained with reference to the theory of "genetic mosaicism".
It appears justified to isolate under the term syndrome of Nicolau and Balus', a group of deformities that include eruptive syringomas of the disseminated micropapular type, milium cysts and atrophoderma vermiculata. We present here a highly typical observation of an 11-year-old child whose mother also presented with eruptive syringomas. This syndrome accentuates the semiological value of disseminated syringomas and atrophoderma vermiculata, which often occur in the context of a complex syndrome. In addition, an associated elastopathy was discovered by light- and electron-microscopic observations.
A case of malignant chondroid syringoma on a foot with multiple bone metastases is presented. An 18-year-old male patient first noticed a protruding mass on the plantar surface of his right foot at 9 years of age. A sweat gland tumor of a benign nature was diagnosed and excised at that time. The tumor recurred three times during the 10 years after surgery and was finally diagnosed as malignant chondroid syringoma. Multiple bone metastases involving the calcaneus, talus and fibula of the lesion side were found after extensive radiologic survey. The patient underwent below-knee amputation with total removal of the fibula. However, pelvic bone metastasis developed 1 year after the amputation. He died of this disease due to brain and diffuse bony metastasis 36 months after the amputation. This is a rare case of malignant chondroid syringoma with a long history but ominous outcome. We recommend that sweat gland tumors be carefully examined and treated more radically when there is a suspicion of malignancy.
Although syringoma is a relatively common adnexal tumor, syringoma of the hands has not been reported previously. We report a unique case of multiple acral syringomas that appeared as symmetrical grouped erythematous papules limited to the dorsum of the proximal and middle phalanges of both hands in a young man. This condition should be considered in the diagnosis of papular lesions of the hands.
A 42-year-old male with Down's syndrome suddenly developed disseminated red-brown papules. Histological studies revealed eruptive syringomas. We discuss the association of Down syndrome with different skin diseases, in particular with syringomas.
Syringoma is a benign eccrine tumor that generally forms asymptomatic papules on facial skin. Eruptive forms of the tumor are uncommon and of uncertain cause. We report a case in which eruptive syringoma with clear cell morphology was found to express progesterone receptors, suggesting the possibility of hormonal control of the tumor.
Syringomas are benign tumors of the eccrine ducts that appear as small skin-colored or yellow papules. They usually occur in the periorbital area, but have also been found on the scalp, forehead, cheeks, axillae, abdomen, extremities, genitalia, and buttocks. We describe a patient with an unusual presentation of syringomas on the moustache area.
Syringomas are benign tumors derived from the intraepidermal portion of eccrine sweat ducts. They usually occur on the periobital area, but have also been found on the scalp, forehead, cheeks, axillae, abdomen, extremities, genitalia, and buttocks. We describe a patient with an unusual presentation of unilateral syringoma of the face associated with hyperthyroidism.