Journal of gastrointestinal surgery : official journal of the Society for Surgery of the Alimentary Tract · 2012
Management of squamous cell cancer of the oesophagus in a patient with a polyglandular endocrinopathy (APECED) and achalasia.
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Mc Cormack Orla, Timlin Marie, Mc Gowan Anne, Healy Marie-Louise, Ravi Narayanasamy, Reynolds John V
Аннотация
INTRODUCTION: We report a case of a 37-year-old man, with a background of a rare polyglandular autoimmune syndrome and achalasia, who developed an oesophageal tumour. Both autoimmune polyendocrinopathy candidiasis ectodermal dystrophy (APECED) or type I polyglandular syndrome and achalasia confer increased risk of development of oesophageal squamous cell carcinoma.
METHODS: Despite having had multiple endoscopic examinations and dilatations in the recent past, this patient presented with dysphagia, and on endoscopy, he was found to have a mid-oesophageal tumour. A multidisciplinary team approach was vital in his management as careful monitoring of underlying disorders including Addison's disease and hypoparathyroidism were challenging during neoadjuvant chemoradiotherapy and in the perioperative period.
RESULTS: He made an uneventful recovery after a three-stage oesophagectomy, and histologically, he had a complete pathological response.
CONCLUSION: To our knowledge, this is the first successful outcome of a patient with APECED and oesophageal carcinoma in the literature.
METHODS: Despite having had multiple endoscopic examinations and dilatations in the recent past, this patient presented with dysphagia, and on endoscopy, he was found to have a mid-oesophageal tumour. A multidisciplinary team approach was vital in his management as careful monitoring of underlying disorders including Addison's disease and hypoparathyroidism were challenging during neoadjuvant chemoradiotherapy and in the perioperative period.
RESULTS: He made an uneventful recovery after a three-stage oesophagectomy, and histologically, he had a complete pathological response.
CONCLUSION: To our knowledge, this is the first successful outcome of a patient with APECED and oesophageal carcinoma in the literature.