Case Report
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World J Gastrointest Oncol. Feb 15, 2013; 5(2): 34-37
Published online Feb 15, 2013. doi: 10.4251/wjgo.v5.i2.34
Diffuse intestinal ganglioneuromatosis an uncommon manifestation of Cowden syndrome
Maria Teresa Herranz Bachiller, Jesus Barrio Andrés, Fernando Pons, Noelia Alcaide Suárez, Rafael Ruiz-Zorrilla, Lorena Sancho del Val, Sara Lorenzo Pelayo, Carlos De La Serna Higuera, Ramon Atienza Sánchez, Manuel Perez Miranda
Maria Teresa Herranz Bachiller, Jesus Barrio Andrés, Fernando Pons, Noelia Alcaide Suárez, Rafael Ruiz-Zorrilla, Lorena Sancho del Val, Sara Lorenzo Pelayo, Carlos De La Serna Higuera, Ramon Atienza Sánchez, Manuel Perez Miranda, Department of Gastroenterology, Rio Hortega Hospital, 47012 Valladolid, Spain
Author contributions: All authors contributed equally to this case report.
Correspondence to: Maria Teresa Herranz Bachiller, MD, Department of Gastroenterology, Rio Hortega Hospital, Dulzaina Street No. 2, 47012 Valladolid, Spain. maiteheba@hotmail.com
Telephone: +34-983-420400 Fax: +34-983-420432
Received: March 2, 2012
Revised: September 26, 2012
Accepted: December 1, 2012
Published online: February 15, 2013
Abstract

Diffuse intestinal ganglioneuromatosis is a hamartomatous polyposis characterized by a disseminated, intramural or transmural proliferation of neural elements involving the enteric plexuses. It has been associated with MEN II, neurofibromatosis type 1 and hamartomatous polyposis associated with phosphatase and tensin homolog mutation. We report the case of a female patient with a history of a breast and endometrial tumor who presented in a colonoscopy performed for rectal bleeding diffuse ganglioneuromatosis, which oriented the search for other characteristic findings of Cowden syndrome given the personal history of the patient. The presence of an esophagogastric polyposis was also noted. Cowden syndrome is characterized by skin lesions, but it is rarely diagnosed by these lesions, because they are usually overlooked. Intestinal polyposis is not a major diagnostic criterion but it is very useful for early diagnosis. The combination of colonic polyposis and glucogenic acanthosis should orient the diagnosis to Cowden syndrome.

Keywords: Ganglioneuromatosis; Gastrointestinal poliposis; Phosphatase and tensin homolog; Cowden síndrome; Hamartoma