Trichoepitheliomas and eccrine spiradenomas with spiradenoma/cylindroma overlap

Bumgardner, Amy C.; Hsu, Sylvia; Nunez-Gussman, Janna; Schwartz, Mary R.

doi:10.1111/j.1365-4632.2005.02044.x

Cited by 30 publications

(21 citation statements)

References 16 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…About 50 cases of malignant spiradenomas have been described in literature. Spiradenomas have been known to occur in Brooke Spiegler syndrome which manifests with combined lesions like cylindromas, spiradenomas and trichoepitheliomas [4,5]. But an eccrine spiradenoma co-existing with lichen planus in an old and treated case of basal cell carcinoma, as in the present case is an extremely uncommon occurrence.…”

mentioning

confidence: 73%

Inexplicable Co- Existence of Eccrine Spiradenoma and Lichen Planus in an old Case of Basal Cell Carcinoma

Hemalatha

et al. 2015

JCDR

View full text Add to dashboard Cite

An 80-year-old woman presented with history of a painless raised blue to blackish lesion over the right upper part of the thigh measuring 2 x 1 cms. On local examination, the nodule was nontender, with a smooth surface. The overlying skin was pinchable. She also complained of another raised swelling over the medial side of the left breast which was present since 10 years which on examination was a plaque-like lesion measuring 1 x 0.5 cms [Table/ Fig-1]. General examination did not reveal any significant findings. Her past history and clinical records revealed that she had been treated for basal cell carcinoma 10 years ago.The provisional clinical diagnosis offered for the nodule over the thigh was "Benign Adnexal tumour" and that for the lesion over the breast was "Seborrheic Keratosis." Both the lesions were excised and submitted for histopathological examination. HIStopAtHoLoGICAL FINDINGSGross examination-Biopsy from the breast showed a skin covered grey-white tissue mass measuring 0.75 x 0.4 cms. Biopsy from the thigh showed a skin covered grey-white tissue mass measuring 1.75 x 0.75 cms. Cut section showed a well encapsulated greywhite, skin covered nodule.Excisional biopsy from the lesion over the breast showed focal hyperkeratosis, acanthosis, focal spongiosis and melanin incontinence. Papillary dermis showed diffuse and dense band-like lymphocytic infiltrates [Table/ Fig-2].Biopsy from the nodule over the thigh showed thinned out epidermis and a well-encapsulated, lobulated appendageal tumour Pathology SectionInexplicable Co-Existence of Eccrine Spiradenoma and Lichen Planus in an old Case of Basal Cell Carcinoma ABStRACtEccrine spiradenoma is a rare benign adnexal tumour usually seen in the head, neck or upper trunk in young adults. It is rare in the lower extremities and in the elderly. Co-existence of eccrine spiradenoma with other lesions like cylindroma, trichoepithelioma, hidradenoma and chondroid syringoma has been documented in literature. But, concomitant occurrence of eccrine spiradenoma with a non-neoplastic lesion like lichen planus in an old and treated case of basal cell carcinoma has not been documented in literature till date.We present a rare case of eccrine spiradenoma occurring on the right thigh in an elderly female along with lichen planus over the left breast. The lady had been diagnosed with basal cell carcinoma 10 years ago and treated with wide local excision. A brief review of literature is also included.in the dermis composed of two types of epithelial cells arranged in cords and sheets with evidence of ductal differentiation at places [Table /Fig-3]. The cells in the center were large and basaloid with large pale nuclei. The cells in the periphery were smaller with hypechromatic nuclei and scanty cytoplasm. Some blood vessels showed perivascular palisading by tumour cells [Table /Fig-4].Final diagnosis-Eccrine spiradenoma over the thigh co-existent with lichen planus over the breast. DISCUSSIoNEccrine spiradenoma, an uncommon benign tumour of sweat gland origin with debatable ...

show abstract

mentioning

confidence: 73%

Inexplicable Co- Existence of Eccrine Spiradenoma and Lichen Planus in an old Case of Basal Cell Carcinoma

Hemalatha

et al. 2015

JCDR

View full text Add to dashboard Cite

show abstract

“…4 Cylindromas, unlike spiradenomas, have few or no lymphocytes. 1 Spiradenomas, also termed ''eccrine'' spiradenomas and first described by Kerstin and Helwig 5 in 1956, often present during early adulthood as solitary, firm, intradermal lesions that can be painful or tender. 1,2 Although lesions are generally found on the trunk and ventral extremities, they may also present on the scalp or face.…”

Section: Introductionmentioning

confidence: 99%

“…1 Spiradenomas, also termed ''eccrine'' spiradenomas and first described by Kerstin and Helwig 5 in 1956, often present during early adulthood as solitary, firm, intradermal lesions that can be painful or tender. 1,2 Although lesions are generally found on the trunk and ventral extremities, they may also present on the scalp or face. Similar to cylindromas, differentiation from and relationship to eccrine dermal ducts, secretory coils, and apocrine structures is supported.…”

Section: Introductionmentioning

confidence: 99%

“…These slow-growing adnexal neoplasms typically develop during adulthood in the facial or scalp region and form variably sized, sometimes painful dermal papules or nodules. 1,2 Cylindroma origin has been linked to the coiled duct with apocrine differentiation, the secretory coil, and eccrine and pilar structures. 1,3 Classic cylindromas histologically present in the upper dermis and grow in a ''mosaic'' or ''jigsaw puzzle'' lobular pattern comprising small, variably shaped, well-demarcated islands, each surrounded by a prominent hyaline membrane (periodic acid-Schiff positive).…”

Section: Introductionmentioning

confidence: 99%

“…1,3 Classic cylindromas histologically present in the upper dermis and grow in a ''mosaic'' or ''jigsaw puzzle'' lobular pattern comprising small, variably shaped, well-demarcated islands, each surrounded by a prominent hyaline membrane (periodic acid-Schiff positive). 1,4 These neoplasms contain 2 distinct cell populations: larger pale cells found peripherally and smaller, basophilic cells with scant cytoplasm and hyperchromatic nuclei, found centrally. Ductal differentiation is common.…”

Section: Introductionmentioning

confidence: 99%

See 2 more Smart Citations

Carcinoma Ex Spiradenoma/Cylindroma Confirmed by Immunohistochemical and Molecular Loss-of-Heterozygosity Profiling

Jukic

Drogowski²,

Davie³

2009

The American Journal of Dermatopathology

View full text Add to dashboard Cite

We present a case of spiradenoma/cylindroma with admixed carcinoma of unknown origin, resolved using immunohistochemical and molecular loss-of-heterozygosity (LOH) profiling. The patient, a woman in her mid-70s, initially presented with separate mammary (ductal) carcinomas of the right and left breasts that were treated with radical mastectomies. For 9 years, the patient remained disease free until complaining of a slow-growing skin nodule on the lower back that was excised under clinical suspicion of metastatic mammary carcinoma. Histopathological exam revealed a benign eccrine spiradenoma/cylindroma and an intermixed carcinoma, with a differential diagnosis of either primary eccrine carcinoma or mammary carcinoma metastatic to the spiradenoma/cylindroma. Histological features and immunohistochemical staining favored eccrine carcinoma but not unequivocally; therefore, LOH profiles were performed on archival paraffin block tissue from the 3 neoplastic lesions (4 components). The mammary carcinomas showed disparate LOH at 5 of 7 (right breast) and 4 of 7 (left breast) informative genetic loci, establishing these carcinomas as separate primary neoplasms. Both the spiradenoma/cylindroma and eccrine carcinoma revealed no LOH at the tested loci, establishing the unknown carcinoma as an independent carcinoma arising within a spiradenoma/cylindroma. This neoplasm is referred to in the literature as carcinoma ex spiradenoma/cylindroma and spiradenocylindrocarcinoma.

show abstract

Tumor Mapping in 2 Large Multigenerational Families With CYLD Mutations

Rajan¹,

Langtry²,

Ashworth³

et al. 2009

Arch Dermatol

View full text Add to dashboard Cite

Objective To comprehensively ascertain the extent and severity of clinical features in multiple affected individuals from two large families with proven heterozygous mutations in the CYLD locus and to correlate these findings with the three appendageal tumor predisposition syndromes, familial cylindromatosis (FC), Brooke-Spiegler syndrome (BSS), and multiple familial trichoepitheliomas (MFT) known to be associated with such germline mutations. Design Inter- and intra-familial observational study. Setting Tertiary genetic and dermatology referral centre. Participants 32 individuals were recruited from two large multigenerational families with CYLD mutations. Clinical details, history and tumor maps were obtained from all participants whilst 18 were further corroborated with detailed clinical examination. Main outcome measures Severity of tumor density, distribution and histology, associated medical conditions, patient symptoms and impact of disease on quality of life. Results We demonstrate a wide variation in clinical presentation seen in individuals from the same family. In addition, we provide clinical evidence that correlates with hormonally stimulated hair follicles being particularly vulnerable to loss of heterozygosity and tumor induction. Conclusion In view of our findings, we propose that the burden of disease at sites other than the head and neck is underreported in the literature, but impacts greatly on quality of life. The differentiation between the clinical diagnoses has little prognostic or clinical utility in genetic counselling even within individuals from the same family. Thus, we suggest an encompassing diagnosis of “CYLD cutaneous syndrome”. Finally, our results relating to the clinical distribution of tumors suggest hormonal factors may play an important role in tumor induction in these patients.

show abstract

Trichoepitheliomas and eccrine spiradenomas with spiradenoma/cylindroma overlap

Cited by 30 publications

References 16 publications

Inexplicable Co- Existence of Eccrine Spiradenoma and Lichen Planus in an old Case of Basal Cell Carcinoma

Inexplicable Co- Existence of Eccrine Spiradenoma and Lichen Planus in an old Case of Basal Cell Carcinoma

Carcinoma Ex Spiradenoma/Cylindroma Confirmed by Immunohistochemical and Molecular Loss-of-Heterozygosity Profiling

Tumor Mapping in 2 Large Multigenerational Families With CYLD Mutations

Contact Info

Product

Resources

About