Esophageal Leiomyomatosis in a Woman with a History of Vulvar Leiomyoma and Barrett’s Esophagus

Abstract

Background: The diagnosis and treatment of esophageal pathology remains a challenge despite advances in preoperative endoscopy, radiographic staging, and perioperative care. Case Report: In this article, we present an interesting case of esophageal leiomyomatosis in a woman with a history of vulvar leiomyoma and Barrett’s esophagus. This paper represents the first reported simultaneous occurrence of these three pathologic entities in the English literature. Conclusions: The clinical presentation and characteristic pathologic findings in patients with esophageal leiomyomatosis are reviewed. Diagnostic and therapeutic approaches to esophageal masses are discussed including the indications for esophageal resection.

Alport syndrome with diffuse leiomyomatosis

“…Additionally, esophagoscopy can only show acute esophagitis and barett esophagus [Nahmad and Clatworthy, 1973;Leborgne et al, 1989;Compagnoni et al, 2000], again also documented in our patient. Muscular hypertrophy always affects the esophagus but may as well involve extraesophageal organs like the tracheobronchial [Leborgne et al, 1989;Antignac and Heidet, 1996] and female genital tracts [Compagnoni et al, 2000;Guillem et al, 2001]. Guthrie [1945] described a 9-year-old girl with involvement of the esophagus, pylorus, duodenum, and jejunum.…”

Vulvar leiomyoma in association with gastrointestinal leiomyoma

“…Combined leiomyoma of the vulva and oesophagus is even rarer, with only 21 cases reported 3,4 . Some cases were leiomyomatosis where there was diffuse smooth muscle hypertrophy 3,4 or multiple nodules 5 rather than a discrete tumour. Our tumour was macroscopically discrete but had nodules histopathologically.…”

“…Our tumour was macroscopically discrete but had nodules histopathologically. It has been suggested that these cases represented an ‘esophago‐vulvar’ syndrome 3 …”

18F-Fluorodeoxyglucose PET/CT in a Patient with Esophageal and Genital Leiomyomatosis

“…Rarely, thickened layers of the esophageal wall can also be caused by diffuse leiomyomatosis, a benign muscular abnormality (1). Diffuse leiomyomatosis is also known as giant muscular hypertrophy with diffuse proliferation of the esophageal smooth muscle.…”

“…Diffuse esophageal leiomyomatosis is a rare benign disease of proliferating smooth muscle that can be accompanied by leiomyoma of extraesophageal organs including the tracheobronchial and genital tracts (1-3). Moreover, diffuse leiomyomatosis can be associated with Alport syndrome in familial cases (2, 4).…”