J Korean Med Sci.  1997 Dec;12(6):570-575. 10.3346/jkms.1997.12.6.570.

Cowden's disease--a report on the first case in Korea and literature review

• Lee HR
• Moon YS
• Yeom CH
• Kim KW
• Chun JY
• Kim HK
• Choi HS
• Kim DK
• Chung TS

• KMID: 645122
• DOI: http://doi.org/10.3346/jkms.1997.12.6.570

Abstract

Cowden's disease, or multiple hamartoma syndrome, is an uncommon condition with characteristic mucocutaneous lesions associated with abnormalities of the breast, thyroid, and gastrointestinal tract. We describe a 32-year-old man with oral mucosal papillomatosis and plantar hyperkeratosis as a definite case of Cowden's disease according to the criteria proposed by Salem and Steck. The patient also had a thyroid mass and numerous gastrointestinal polyps endoscopically. Histologically the polyps were hamartomatous or hyperplastic polyps. The oral papillary lesions were fibroepithelial polyps and the thyroid mass was a follicular adenoma. We review the literature on this entity and summarize the pertinent findings. To the best of our knowledge, this is the first documented case of Cowden's disease in a Korean.

MeSH Terms

Adult
Case Report
Hamartoma Syndrome, Multiple/epidemiology
Hamartoma Syndrome, Multiple/diagnosis*
Human
Incidence
Intestinal Polyps/pathology
Keratoderma, Palmoplantar/pathology
Korea/epidemiology
Male
Papilloma/pathology
Thyroid Neoplasms/pathology
Tongue Neoplasms/pathology