Cylindroma is a benign skin appendageal
tumor arising from pluripotent stem cells in the follicle. Multiple cylindroma
typically occurs as a component of Brooke-Spiegler syndrome or as the only skin
lesion of familial cylindromatosis. We herein report an unusual case of
multiple cylindroma involving the trunk without any other features of
Brooke-Spiegler syndrome and no family history supporting the possibility of
familial cylindromatosis. Immunohistochemical features of CYLD and NF-κB, which
are involved in the pathogenesis of cylindroma, in the tumors are also
presented.
References
[1]
Sellheyer, K. (2015) Spiradenoma and Cylindroma Originate from the Hair Follicle Bulge and Not from the Eccrine Sweat Gland: An Immunohistochemical Study with CD200 and Other Stem Cell Markers. Journal of Cutaneous Pathology, 42, 90-101. http://dx.doi.org/10.1111/cup.12406
[2]
Massoumi, R., Podda, M., Fassler, R. and Paus, R. (2006) Cylindroma as Tumor of Hair Follicle Origin. Journal of Investigative Dermatology, 126, 1182-1184. http://dx.doi.org/10.1038/sj.jid.5700218
[3]
Klein; W., Chan, E. and Seykora, J.T. (2005) Tumors of the Epidermal Appendages. In: Elder, D.E., Elenitsas, R., Johnson B.L. and Murphy, G.F., Eds., Lever’s Histopathology of the Skin, Lippincott Williams & Wilkins, Philadelphia, 897-898.
[4]
Calonje, E. (2010) Tumours of the Skin Appendages. In: Burns, T., Breathnach, S., Cox, N. and Griffiths, C., Eds., Rook’s Textbook of Dermatology, Wiley-Blackwell, Singapore, 28-53. http://dx.doi.org/10.1002/9781444317633.ch53
[5]
Rajan, N. and Ashworth, A. (2015) Inherited Cylindromas: Lessons from a Rare Tumour. The Lancet Oncology, 16, e460-e469. http://dx.doi.org/10.1002/9781444317633.ch53
[6]
Saga, K. (2001) Histochemical and Immunohistochemical Markers for Human Eccrine and Apocrine Sweat Glands: An Aid for Histopathologic Differentiation of Sweat Gland Tumors. Journal of Investigative Dermatology. Symposium Proceedings, 6, 49-53. http://dx.doi.org/10.1046/j.0022-202x.2001.00005.x
[7]
Blake, P.W. and Toro, J.R. (2009) Update of Cylindromatosis Gene (CYLD) Mutations in Brooke-Spiegler Syndrome: Novel Insights into the Role of Deubiquitination in Cell Signaling. Human Mutation, 30, 1025-1036.
http://dx.doi.org/10.1002/humu.21024
[8]
Bowen, S., Gill, M., Lee, D.A., Fisher, G., Geronemus, R.G., Vazquez, M.E. and Celebi, J.T. (2005) Mutations in the CYLD Gene in Brooke-Spiegler Syndrome, Familial Cylindromatosis, and Multiple Familial Trichoepithelioma: Lack of Genotype-Phenotype Correlation. Journal of Investigative Dermatology, 124, 919-920.
http://dx.doi.org/10.1111/j.0022-202X.2005.23688.x
[9]
Tauriello, D.V., Haegebarth, A., Kuper, I., Edelmann, M.J., Henraat, M., Canninga-van Dijk, M.R., Kessler, B.M., Clevers, H. and Maurice, M.M. (2010) Loss of the Tumor Suppressor CYLD Enhances Wnt/Beta-Catenin Signaling through K63-Linked Ubiquitination of Dvl. Molecular Cell, 37, 607-619.
http://dx.doi.org/10.1016/j.molcel.2010.01.035
[10]
Sun, S.C. (2010) CYLD: A Tumor Suppressor Deubiquitinase Regulating NF-kappaB Activation and Diverse Biological Processes. Cell Death & Differentiation, 17, 25-34. http://dx.doi.org/10.1038/cdd.2009.43
[11]
Woodon, D. (2010) Cylindroma. In: Weedon, D., Ed., Weedon’s Skin Pathology, Churchill Livingstone Elsevier, London, 785-786.
[12]
Durani, B.K., Kurzen, H., Jaeckel, A., Kuner, N., Naeher, H. and Hartschuh, W. (2001) Malignant Transformation of Multiple Dermal Cylindromas. British Journal of Dermatology, 145, 653-656.
http://dx.doi.org/10.1046/j.1365-2133.2001.04460.x
[13]
Weedon, D. (2010) Tumors of Cutaneous Appendages. In: Weedon, D., Ed., Weedon’s Skin Pathology, Churchill Livingstone Elsevier, China, 785-786. http://dx.doi.org/10.1016/b978-0-7020-3485-5.00034-6
[14]
Bignell, G.R., Warren, W., Seal, S., Takahashi, M., Rapley, E., Barfoot, R., Green, H., Brown, C., Biggs, P.J., Lakhani, S.R., Jones, C., Hansen, J., Blair, E., Hofmann, B., Siebert, R., Turner, G., Evans, D.G., Schrander-Stumpel, C., Beemer, F.A., van Den Ouweland, A., Halley, D., Delpech, B., Cleveland, M.G., Leigh, I., Leisti, J. and Rasmussen, S. (2000) Identification of the Familial Cylindromatosis Tumour-Suppressor Gene. Nature Genetics, 25, 160-165.
http://dx.doi.org/10.1016/b978-0-7020-3485-5.00034-6
[15]
Massoumi, R. (2011) CYLD: A Deubiquitination Enzyme with Multiple Roles in Cancer. Future Oncology, 7, 285-297. http://dx.doi.org/10.2217/fon.10.187
[16]
Lin, P.Y., Fatteh, S.M. and Lloyd, K.M. (1987) Malignant Transformation in a Solitary Dermal Cylindroma. Archives of Pathology & Laboratory Medicine, 111, 765-767.
[17]
Munger, B.L., Graham, J.H. and Helwig, E.B. (1962) Ultrastructure and Histochemical Characteristics of Dermal Eccrine Cylindroma (Turban Tumor). Journal of Investigative Dermatology, 39, 577-595.
[18]
Urbach, F., Graham, J.H., Goldstein, J. and Munger, B.L. (1963) Dermal Eccrine Cylindroma: A Histochemical, Electron Microscopic, and Therapeutic (X-Ray) Study. Archives of Dermatological, 88, 880-894.
[19]
Crain, R.C. and Helwig, E.B. (1961) Dermal Cylindroma (Dermal Eccrine Cylindroma). American Journal of Clinical Pathology, 35, 504-515.
[20]
Canedo, T., de Almeida, M.P., Cuzzi, T. and Ramos-e-Silva, M. (2010) Immunophenotypic Aspects of Cylindroma and Nodular Hidradenoma. Journal of the European Academy of Dermatology and Venereology, 24, 178-185.
http://dx.doi.org/10.1111/j.1468-3083.2009.03398.x
[21]
Cotton, D.W. and Braye, S.G. (1984) Dermal Cylindromas Originate from the Eccrine Sweat Gland. British Journal of Dermatology, 111, 53-61. http://dx.doi.org/10.1111/j.1365-2133.1984.tb04016.x
[22]
Missall, T.A., Burkemper, N.M., Jensen, S.L. and Hurley, M.Y. (2009) Immunohistochemical Differentiation of Four Benign Eccrine Tumors. Journal of Cutaneous Pathology, 36, 190-196.
http://dx.doi.org/10.1111/j.1600-0560.2008.00991.x
[23]
Penneys, N.S. and Kaiser, M. (1993) Cylindroma Expresses Immunohistochemical Markers Linking It to Eccrine Coil. Journal of Cutaneous Pathology, 20, 40-43. http://dx.doi.org/10.1111/j.1600-0560.1993.tb01247.x
[24]
Hashimoto, K. and Lever, W.F. (1969) Histogenesis of Skin Appendage Tumors. Archives of Dermatological, 100, 356-369. http://dx.doi.org/10.1001/archderm.1969.01610270098021
[25]
Lever, W.F. and Hashimoto, K. (1966) Histogenesis of Various Skin Appendage Tumors in the Ligh of Histochemical and Electron Microscopical Findings (Eccrine Poroma, Eccrine Spiradenoma, Syringoma, Cylindroma and Calcified Epithelioma). Hautarzt, 17, 161-173.
[26]
Layegh, P., Sharifi-Sistani, N., Abadian, M. and Moghiman, T. (2008) Brooke-Spiegler Syndrome. Indian Journal of Dermatology, Venereology and Leprology, 74, 632-634. http://dx.doi.org/10.4103/0378-6323.45109
[27]
Parren, L.J., Bauer, B., Hamm, H. and Frank, J. (2008) Brooke-Spiegler Syndrome Complicated by Unilateral Hearing Loss. International Journal of Dermatology, 47, 56-59.
