Case study Squamous cell carcinoma arising in a communicating bronchopulmonary-foregut malformation Keisuke Matsusaka MD a,b, ⁎ , Yoshihiro Kinoshita MD c , Harushi Udagawa MD c , Masashi Fukayama MD b , Kenichi Ohashi MD a a Department of Pathology, Toranomon Hospital, Tokyo 105-8470, Japan b Department of Pathology, Graduate School of Medicine, The University of Tokyo, Tokyo 113-0033, Japan c Department of Surgery, Toranomon Hospital, Tokyo 105-8470, Japan Received 19 September 2009; revised 5 December 2009; accepted 22 January 2010 Keywords: Bronchopulmonary sequestration; Communicating bronchopulmonary- foregut malformation; Squamous cell carcinoma Summary Communicating bronchopulmonary-foregut malformation, a variant of bronchopulmonary sequestration, is a rare anomaly characterized by communication between an isolated portion of the respiratory tree and the gastrointestinal tract. We report herein a unique case involving a 43-year-old man with squamous cell carcinoma arising in communicating bronchopulmonary-foregut malformation. This patient had a workup for a chief complaint of exacerbation of constitutional dysphagia, resulting in detection of squamous cell carcinoma involving the lower esophagus. Under the clinical diagnosis of esophageal carcinoma, esophagectomy was performed after neoadjuvant chemoradiotherapy. Pathologic findings showed that squamous cell carcinoma had arisen in malformed bronchopulmonary tissue constituting part of the distal esophagus segmentally. This case was unique in that squamous cell carcinoma developed in an extremely rare type of congenital abnormality that had functioned as a passageway for food from birth, as a result of chronic irritation for more than 4 decades. © 2010 Elsevier Inc. All rights reserved. 1. Introduction Bronchopulmonary sequestration (BPS) can be described as a malformation in which nonfunctioning lung tissue is separated from the normal bronchopulmonary tree and vascularized by an aberrant systemic artery. BPS is a rare abnormality with an estimated incidence of 0.15% to 1.8% of all pulmonary malformations [1]. With such a rare anomaly as BPS, an extremely unusual variant has reportedly been characterized by a patent congenital communication between the esophagus or stomach and an isolated malformed portion of the respiratory system [2,3]. We report herein a unique case involving a 43-year-old man with squamous cell carcinoma (SCC) arising in a bronchopulmonary malforma- tion constituting part of the lower esophagus segmentally and having functioned as a passageway for food from birth. 2. Case report This patient was a man in his 40s who noticed a constitutional difficulty in swallowing without any signifi- cant symptoms such as dyspnea or recurrent respiratory tract infection. Starting from the age of 41 years, however, ⁎ Corresponding author. Genome Science Division, Research Center for Advanced Science and Technology, The University of Tokyo, Tokyo 153- 8904, Japan. E-mail addresses: ksk.matsusaka@gmail.com, matsusaka-hma@umin.ac.jp (K. Matsusaka). www.elsevier.com/locate/humpath 0046-8177/$ – see front matter © 2010 Elsevier Inc. All rights reserved. doi:10.1016/j.humpath.2010.01.024 Human Pathology (2010) 41, 1650–1654