Esophageal cancer in an adult with congenital esophageal stenosis: a case report.

Adult Congenital esophageal stenosis Esophageal cancer Fibromuscular thickening

Journal

Surgical case reports
ISSN: 2198-7793
Titre abrégé: Surg Case Rep
Pays: Germany
ID NLM: 101662125

Informations de publication

Date de publication:
12 Mar 2024
Historique:
received: 25 01 2024
accepted: 04 03 2024
medline: 12 3 2024
pubmed: 12 3 2024
entrez: 12 3 2024
Statut: epublish

Résumé

Congenital esophageal stenosis (CES) is a rare condition. We encountered a case of esophageal cancer that developed in an adult with persistent CES. Although many studies have investigated the therapeutic outcomes and performed surveillance for symptoms after treatment for CES, few have performed long-term surveillance or reported on the development of esophageal cancer. We report this case because it is extremely rare and has important implications. A 45-year-old woman with worsening dysphagia was transferred to our hospital. The patient was diagnosed with CES at 5 years of age and underwent surgery at another hospital. The patient underwent esophageal dilatation for stenosis at 36 years of age. Esophagoscopy performed at our hospital revealed a circumferential ulcerated lesion and stenosis 15-29 cm from the incisors. Histological examination of the biopsy specimen revealed squamous cell carcinoma. Computed tomography (CT) revealed abnormal circumferential wall thickening in parts of the cervical and almost the entire thoracic esophagus. Chronic mechanical and chemical irritations are believed to cause cancer of the upper esophagus oral to a persistent CES, suggesting the need for long-term surveillance that focuses on residual stenosis and cancer development in patients with CES.

Sections du résumé

BACKGROUND BACKGROUND
Congenital esophageal stenosis (CES) is a rare condition. We encountered a case of esophageal cancer that developed in an adult with persistent CES. Although many studies have investigated the therapeutic outcomes and performed surveillance for symptoms after treatment for CES, few have performed long-term surveillance or reported on the development of esophageal cancer. We report this case because it is extremely rare and has important implications.
CASE PRESENTATION METHODS
A 45-year-old woman with worsening dysphagia was transferred to our hospital. The patient was diagnosed with CES at 5 years of age and underwent surgery at another hospital. The patient underwent esophageal dilatation for stenosis at 36 years of age. Esophagoscopy performed at our hospital revealed a circumferential ulcerated lesion and stenosis 15-29 cm from the incisors. Histological examination of the biopsy specimen revealed squamous cell carcinoma. Computed tomography (CT) revealed abnormal circumferential wall thickening in parts of the cervical and almost the entire thoracic esophagus.
CONCLUSIONS CONCLUSIONS
Chronic mechanical and chemical irritations are believed to cause cancer of the upper esophagus oral to a persistent CES, suggesting the need for long-term surveillance that focuses on residual stenosis and cancer development in patients with CES.

Identifiants

pubmed: 38467897
doi: 10.1186/s40792-024-01858-1
pii: 10.1186/s40792-024-01858-1
doi:

Types de publication

Journal Article

Langues

eng

Pagination

58

Informations de copyright

© 2024. The Author(s).

Références

Mochizuki K, Yokoi A, Urushihara N, Yabe K, Nakashima H, Kitagawa N, et al. Characteristics and treatment of congenital esophageal stenosis: a retrospective collaborative study from three Japanese children’s hospitals. J Pediatr Surg. 2021;56:1771–5. https://doi.org/10.1016/j.jpedsurg.2020.12.029 .
doi: 10.1016/j.jpedsurg.2020.12.029 pubmed: 33455805
Michaud L, Coutenier F, Podevin G, Bonnard A, Becmeur F, Khen-Dunlop N, et al. Characteristics and management of congenital esophageal stenosis: findings from a multicenter study. Orphanet J Rare Dis. 2013;8:186. https://doi.org/10.1186/1750-1172-8-186 .
doi: 10.1186/1750-1172-8-186 pubmed: 24289834 pmcid: 4222067
Terui K, Saito T, Mitsunaga T, Nakata M, Yoshida H. Endoscopic management for congenital esophageal stenosis: a systematic review. World J Gastrointest Endosc. 2015;7:183–91. https://doi.org/10.4253/wjge.v7.i3.183 .
doi: 10.4253/wjge.v7.i3.183 pubmed: 25789088 pmcid: 4360436
Brzački V, Mladenović B, Jeremić L, Živanović D, Govedarović N, Dimić D, et al. Congenital esophageal stenosis: a rare malformation of the foregut. Nagoya J Med Sci. 2019;81:535–47. https://doi.org/10.18999/nagjms.81.4.535 .
doi: 10.18999/nagjms.81.4.535 pubmed: 31849372 pmcid: 6892676
Tabira Y, Yasunaga M, Sakaguchi T, Okuma T, Yamaguchi Y, Kuhara H, et al. Adult case of squamous cell carcinoma arising on congenital esophageal stenosis due to fibromuscular hypertrophy. Dis Esophagus. 2002;15:336–9. https://doi.org/10.1046/j.1442-2050.2002.00270.x .
doi: 10.1046/j.1442-2050.2002.00270.x pubmed: 12472484
Oh CH, Levine MS, Katzka DA, Rubesin SE, Pinheiro LW, Amygdalos MA, et al. Congenital esophageal stenosis in adults: clinical and radiographic findings in seven patients. AJR Am J Roentgenol. 2001;176:1179–82. https://doi.org/10.2214/ajr.176.5.1761179 .
doi: 10.2214/ajr.176.5.1761179 pubmed: 11312178
Ikeda H, Inoue H, Abad MRA, Fujiyoshi Y, Nishikawa Y, Toshimori A, et al. Diagnosis of congenital esophageal stenosis in adults and treatment with peroral endoscopic myotomy. Ann Gastroenterol. 2021;34:493–500. https://doi.org/10.20524/aog.2021.0618 .
doi: 10.20524/aog.2021.0618 pubmed: 34276187 pmcid: 8276369
Suzuhigashi M, Kaji T, Noguchi H, Muto M, Goto M, Mukai M, et al. Current characteristics and management of congenital esophageal stenosis: 40 consecutive cases from a multicenter study in the Kyushu area of Japan. Pediatr Surg Int. 2017;33:1035–40. https://doi.org/10.1007/s00383-017-4133-0 .
doi: 10.1007/s00383-017-4133-0 pubmed: 28801834
Amin MB, Edge SB, Greene FL, Byrd DR, Brookland RK, Washington MK, et al. AJCC cancer staging manual. Springer International Publishing; 2017.
Tustumi F, Arienzo VP, Sunye IR, Lucas PFS, Colonno BB, Quintas JG, et al. Esophageal dysbiosis in achalasia and cancer development: a critical review. Genes. 2023;14:1521. https://doi.org/10.3390/genes14081521 .
doi: 10.3390/genes14081521 pubmed: 37628573 pmcid: 10454429
Pomenti S, Blackett JW, Jodorkovsky D. Achalasia: diagnosis, management and surveillance. Gastroenterol Clin North Am. 2021;50:721–36. https://doi.org/10.1016/j.gtc.2021.07.001 .
doi: 10.1016/j.gtc.2021.07.001 pubmed: 34717867

Auteurs

Yushi Fujiwara (Y)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan. fujiwaray@keiyukaisapporo.or.jp.

Hidehiko Kitagami (H)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan.

Tomohiro Kikkawa (T)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan.

Keita Sakashita (K)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan.

Takaya Kusumi (T)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan.

Yasunori Nishida (Y)

Department of Gastroenterological Surgery, Keiyukai Sapporo Hospital, Minami1-1, Hondori 9-Chome, Shiroishi-Ku, Sapporo, 003-0026, Japan.

Classifications MeSH