Int J Clin Exp Pathol 2013;6(12):3026-3028 www.ijcep.com /ISSN:1936-2625/IJCEP1309034
Letter to Editor Esophageal squamous cell carcinoma in situ overlying leiomyoma: a case report with review of the literature Mitsuaki Ishida1, Yosuke Mochizuki2, Muneo Iwai1, Keiko Yoshida1, Akiko Kagotani1, Hidetoshi Okabe1 Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, 2Division of Digestive Endoscopy, Shiga University of Medical Science, Shiga, Japan 1
Received September 12, 2013; Accepted September 29, 2013; Epub November 15, 2013; Published December 1, 2013
Mesenchymal tumor of the esophagus is rare, and the most common mesenchymal tumor of the esophagus is leiomyoma [1-3]. The incidence of esophageal leiomyoma from autopsy cases ranges from 0.005 to 5% [2, 3]. Although detailed histopathological examination of autopsy and surgically resected case series demonstrated that its incidence is higher at 7.9% . This disease is typically found in patients between 20 and 50 years of age, and more often in men by ratio of 2:1 . Esophageal leiomyoma represents 10% of all gastrointestinal leiomyomas . Moreover, it usually appears as a solitary tumor (97%) , and multiple leiomyomas of the esophagus have been extremely rarely documented [4-6]. Albeit extremely rare, coexistence of squamous cell carcinoma (SCC) of the esophagus overlying benign mesenchymal tumor has been reported . Herein, we describe the fifth documented case of SCC in situ (high-grade intraepithelial neoplasia) of the esophagus overlying leiomyoma with review of the literature. A 75-year-old Japanese female with past history of hyperglycemia and osteoporosis was detected with an esophageal tumor at health checkup, and was subsequently referred to our hospital. Endoscopic examination revealed a submucosal tumor, measuring 5 x 4 mm in diameter, 20 cm from the incision (Figure 1). Endoscopic mucosal resection of the esophageal tumor was performed. Histopathological examination of the endoscopically-resected specimen revealed a wellcircumscribed submucosal nodular lesion with continuity with the muscularis mucosae (Figure
2A). The submucosal nodule was composed of proliferation of interlacing bundles of spindle cells with eosinophilic cytoplasm and bland cigar-shaped nuclei without conspicuous nucleoli (Figure 2B). Neither mitotic figures nor necrosis was noted. A tiny intraepithelial depressed lesion, measuring 1 x 0.8 mm in diameter, was present overlying the submucosal tumor (Figure 2A). Proliferation of atypical squamous cells with large nuclei in the entire layer of the squamous epithelium was observed (Figure 2C). Mitotic figures were present in the upper region of the squamous epithelium. No invasive growth was noted (Figure 2A, 2C). Immunohistochemical studies were performed using an autostainer (Ventana) by the same method as previously reported [7-10]. The spindle cells were diffusely positive for desmin, alpha-smooth muscle actin (Figure 3A), but negative for CD34, c-kit, and S-100 protein. Ki-67 labeling index was less than 1%. Ki-67positive atypical squamous cells were observed in the entire layer of squamous epithelium (Figure 3B), but overexpression of p53 protein was not noted. According to these results, an ultimate diagnosis of coexistence of esophageal SCC in situ (high-grade intraepithelial neoplasia) overlying leiomyoma was made. There have been several reports of esophageal SCC overlying benign submucosal tumor . Iwaya et al. have summarized the previously reported 13 cases of esophageal SCC overlying benign submucosal tumor . The most common histopathological subtype of submucosal
Esophageal SCC overlying leiomyoma
Figure 1. Endoscopic examination showing a wellcircumscribed submucosal tumor in the esophagus.
Figure 2. Histopathological features. A: Squamous cell carcinoma in situ (arrows) overlying submucosal tumor. HE, x 40. B: Fascicular growth of the spindle cells with eosinophilic cytoplasm and bland cigarshaped nuclei. HE, x 200. C: Proliferation of atypical squamous cells with large round nuclei in the entire layer of the squamous epithelium. Mitotic figures are scattered. HE, x 200.
Figure 3. Immunohistochemical features. A: Desmin is expressed in the spindle cells. x 40. B: Ki-67-positive atypical squamous cells are present in the entire layer of the squamous mucosa. x 200.
tumor was leiomyoma (10 cases), and that of the remaining cases was lipoma . Moreover, 3027
most of the SCC had invaded into the lamina propria . The present case is the fifth documented case of esophageal SCC in situ overlying leiomyoma. Table 1 summarizes the clinicopathological features of the four previously reported cases as well as the present one . All cases were middle-aged to elderly Japanese patients, and males were preferentially affected (male: female 4:1). Leiomyoma Int J Clin Exp Pathol 2013;6(12):3026-3028
Esophageal SCC overlying leiomyoma Table 1. Clinicopathological features of esophageal squamous cell carcinoma in situ overlying leiomyoma Case No. 1 2 3 4 Present Case
Age/Gender 53/Male 52/Male 61/Male 63/Male 75/Female
Size of leiomyoma (mm) 8 x 6 x 2.5 15 x 11 x 7 Not available Not available 5x4
arose from the muscularis mucosae in 4 cases and from the muscularis propria in the remaining 1 case. The relationship between the occurrence of SCC and leiomyoma has been discussed. Some authors speculated that the occurrence of SCC overlying submucosal tumor may be induced by chronic irritation of the esophageal mucosa due to intraluminal protrusion of the submucosal tumor [11, 12]. Moreover, curative endoscopic resection of esophageal SCC overlying submucosal tumor has been applied recently . Therefore, it is important to recognize that albeit extremely rare, SCC can be present overlying esophageal submucosal tumor, and accurate diagnosis of the presence and depth of SCC is required. Disclosure of conflict of interest None. Address correspondence to: Dr. Mitsuaki Ishida, Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga University of Medical Science, Tsukinowa-cho, Seta, Otsu, Shiga, 520-2192, Japan. Tel: +81-77-548-2603; Fax: +8177-548-2407; E-mail: [email protected]
  
Takubo K, Nakagawa H, Tsuchiya S, Mitomo Y, Sasajima K, Shirota A. Seeding leiomyoma of the esophagus and esophagogastric junction zone. Hum Pathol 1981; 12: 1006-1010. Kramer MD, Gibb SP, Ellis FH Jr. Giant leiomyoma of esophagus. J Surg Oncol 1986; 33: 166169. Postlethwait RW, Musser AW. Changes in the esophagus in 1,000 autopsy specimens. J Thorac Cardiovasc Surg 1974; 68: 953-956. Mutrie CJ, Donahue DM, Wain JC, Wright CD, Gaissert HA, Grillo HC, Mathisen DJ, Allan JS. Esophageal leiomyoma: a 40-year experience. Ann Thorac Surg 2005; 79: 1122-1125.
Origin of leiomyoma Muscularis mucosae Muscularis mucosae Muscularis mucosae Muscularis propria Muscularis mucosae
Reference    
Iwaya T, Maesawa C, Uesugi N, Kimura T, Ikeda K, Kimura Y, Mitomo S, Ishida K, Sato N, Wakabayashi G. Coexistence of esophageal superficial carcinoma and multiple leiomyomas: A case report. World J Gastroenterol 2006; 28: 4588-4592. Seremetis MG, Lyons WS, deGuzman VC, Peabody JW Jr. Leiomyomata of the esophagus. An analysis of 838 cases. Cancer 1976; 38: 21662177. Ishida M, Iwai M, Yoshida K, Kagotani A, Okabe H. Sarcomatoid carcinoma with small cell carcinoma component of the urinary bladder: A case report with review of the literature. Int J Clin Exp Pathol 2013; 6: 1671-1676. Ishida M, Mori T, Umeda T, Kawai Y, Kubota Y, Abe H, Iwai M, Yoshida K, Kagotani A, Tani T, Okabe H. Pleomorphic lobular carcinoma in a male breast: case report with review of the literature. Int J Clin Exp Pathol 2013; 6: 14411444. Nakanishi R, Ishida M, Hodohara K, Yoshida T, Yoshii M, Okuno H, Horinouchi A, Iwai M, Yoshida K, Kagotani A, Okabe H. Prominent gelatinous bone marrow transformation presenting prior to myelodysplastic syndrome: a case report with review of the literature. Int J Clin Exp Pathol 2013; 6: 1677-1682. Ishida M, Mochizuki Y, Iwai M, Yoshida K, Kagotani A, Okabe H. Pigmented squamous intraepithelial neoplasia of the esophagus. Int J Clin Exp Pathol 2013; 6: 1868-1873. Watanabe M, Baba T, Hotchi M. A case of leiomyoma of the lamina muscularis mucosae of the esophagus with a complication of carcinoma in situ of the overlying mucosa. Acta Pathol Jpn 1987; 37: 1845-51. Yoshikane H, Tsukamoto Y, Niwa Y, Goto H, Hase S, Maruta S, Shimodaira M, Miyata A. The coexistence of esophageal submucosal tumor and carcinoma. Endoscopy 1995; 27: 119-23. Ishihara R, Yamamoto S, Yamamoto S, Iishi H, Takeuchi Y, Kato M, Masuda E, Tatsumi K, Higashino K, Uedo N, Tatsuta M. Endoscopic resection of the esophageal squamous cell carcinoma overlying leiomyoma. Gastrointest Endosc 2008; 67: 745-7.
Int J Clin Exp Pathol 2013;6(12):3026-3028