Case Report
Copyright ©2014 Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 16, 2014; 2(9): 469-473
Published online Sep 16, 2014. doi: 10.12998/wjcc.v2.i9.469
Pneumatosis cystoides intestinalis associated with toxic epidermal necrolysis: A case report
Si-Yuan Yao, Ryutaro Seo, Tohru Nagano, Kazuo Yamazaki
Si-Yuan Yao, Departments of Surgery, Kobe City Medical Center General Hospital, Chuo-ku, Kobe 650-0047, Japan
Ryutaro Seo, Kazuo Yamazaki, Departments of Anesthesiology, Kobe City Medical Center General Hospital, Chuo-ku, Kobe 650-0047, Japan
Tohru Nagano, Departments of Dermatology, Kobe City Medical Center General Hospital, Chuo-ku, Kobe 650-0047, Japan
Author contributions: Yao SY, Seo R, Nagano T and Yamazaki K contributed to the manuscript writing and revision.
Correspondence to: Si-Yuan Yao, MD, Departments of Surgery, Kobe City Medical Center General Hospital, 2-1-1, Minatojima-Minamimachi, Chuo-ku, Kobe 650-0047, Japan. siyuan@kobe-nishishimin-hospi.jp
Telephone: +81-78-3024321 Fax: +81-78-3027537
Received: April 30, 2014
Revised: June 3, 2014
Accepted: June 27, 2014
Published online: September 16, 2014
Processing time: 148 Days and 0.2 Hours
Abstract

Toxic epidermal necrolysis (TEN) is a severe adverse drug reaction, which is characterized by erythema, blisters, and/or erosions of the mucous membranes and skin, but intestinal involvement is rare. In contrast, pneumatosis cystoides intestinalis (PCI) is a rare condition associated with a wide variety of underlying diseases, but to date no patient has presented with PCI associated with TEN. A 55-year-old man was admitted to intensive care unit for treatment of TEN caused by phenobarbital. On day 8 after admission, he presented with progressive abdominal distention and hypotension. Computed tomography (CT) showed gas in the superior mesenteric vein and air filled cysts in the walls of the small intestine. He was suspected of having septic shock due to PCI. As there were no indications of bowel ischemia or necrosis, the patient was managed conservatively with antibiotics and oxygen therapy. On day 10 after admission, he was weaned off catecholamines, with CT on day 11 showing complete resolution of gas in the superior mesenteric vein and air filled cysts. To our knowledge, this article describes the first patient presenting with PCI associated with TEN.

Keywords: Toxic epidermal necrolysis; Intestinal involvement; Pneumatosis cystoids intestinalis; Septic shock; Conservative treatment

Core tip: Toxic epidermal necrolysis is a severe adverse drug reaction, which affects skin and mucosa of whole body. However, intestinal involvement is rare documented. We report the case of a 55-year-old man with toxic epidermal necrolysis caused by phenobarbital. He was diagnosed with pneumatosis cystoides intestinalis during the clinical course. Although septic shock was accompanied, conservative treatment was effective. To our knowledge, this article describes the first patient presenting with pneumatosis cystoides intestinalis associated with toxic epidermal necrolysis.