Coexistence of esophageal superficial carcinoma and multiple leiomyomas: A case report

Abstract

Leiomyomas are the most common benign tumors of the esophagus. They usually occur as a single lesion or as two or three nodules. Only two cases of esophageal multiple leiomyomas comprising more than 10 nodules have been reported previously. Moreover, there have been few reports of esophageal squamous cell carcinoma overlying submucosal tumors. We describe a 71-year-old man who was diagnosed as having a superficial esophageal cancer coexisting with two or three leiomyoma nodules. During surgery, 10 or more nodules that had not been evident preoperatively were palpable in the submucosal and muscular layers throughout the esophagus. As intramural metastasis of the esophageal cancer was suspected, we considered additional lymphadenectomy, but had to rule out this option because of the patient's severe anoxemia. Microscopic examination revealed that all the nodules were leiomyomas (20 lesions, up to 3 cm in diameter), and that invasion of the carcinoma cells was limited to the submucosal layer overlying a relatively large leiomyoma. This is the first report of superficial esophageal cancer coexisting with numerous solitary leiomyomas. Multiple minute leiomyomas are often misdiagnosed as intramural metastasis, and a leiomyoma at the base of a carcinoma lesion can also be misdiagnosed as tumor invasion. The present case shows that accurate diagnosis is required for the management of patients with coexisting superficial esophageal cancer and multiple leiomyomas.

Figure 1

Barium swallow esophagograms. Lack of any marked change in two smooth round defects over the course of a year. A: On admission; B: One year later.

Figure 2

A: Endoscopic examination demonstrates a polypoid lesion with a partially irregular surface; B: Chromoendoscopy with Lugol’s iodine solution demonstrates a polypoid lesion located within the non-staining area and normal mucosa covering part of the surface.

Figure 3

Resected specimen. Multiple polypoid tumors of various sizes are covered with normal mucosa (circles). A polypoid lesion in the lower third of the esophagus exhibits an irregular surface. The broken line indicates the area of SCC.

Figure 4

Photomicrograph of cross-sections (squares) of the resected specimen. Leiomyoma tissue is located in all sections (arrows). S-1: CIS overlies the leiomyoma. The region of CIS extends to the oral side of the leiomyoma (bar). S-2: SCC invades the submucosal layer in a very narrow region. S-3: A section without abnormal macroscopic findings. Multiple small leiomyomas are observed only by microscopic examination.

Figure 5

High-power view of the regions indicated by small letters in Figure 4. A: Intraepithelial SCC; B: The SMT has low cellularity, appears strongly eosinophilic on hematoxylin-eosin staining, and is composed of interlaced smooth-muscle cells with hypovascularity and no mitosis; C: The SCC nest has invaded beyond the muscularis mucosae (MM); D: Two tiny leiomyomas, both originating from the muscular layer.