Surg Today DOI 10.1007/s00595-014-1055-5
CASE REPORT
Gigantic lymphangioma with marked extraluminal progression of the ascending colon: report of a case Hidetoshi Katsuno • Koutarou Maeda • Tsunekazu Hanai • Masahiro Mizuno • Takamitsu Kurashita • Tetsuya Tsukamoto
Received: 22 January 2014 / Accepted: 30 April 2014 Ó Springer Japan 2014
Abstract We report a case of gigantic cystic lymphangioma of the ascending colon excised through an open laparotomy. A 34-year-old woman consulted a gynecologist for treatment of infertility. Transvaginal ultrasonography revealed a cystic mass in the pelvis, and she was transferred to our hospital for further investigation. Abdominal enhanced computed tomography (CT) showed a bulky cystic mass, 25 cm or larger, in the abdominal and pelvic cavity. Colonoscopy revealed a cystic submucosal tumor with a cushion sign. Cystic lymphangioma was diagnosed and excised via open surgery as we could not exclude its malignant potential. Pathological examination confirmed lymphangioma. To our knowledge, this is the most gigantic lymphangioma of the colon documented in the literature. About 3 months after surgery, the patient discovered that she was pregnant and her first baby was delivered at term, uneventfully. Keywords
Colon Lymphangioma Fertility
H. Katsuno (&) K. Maeda T. Hanai M. Mizuno T. Kurashita Department of Surgery, School of Medicine, Fujita Health University, Dengakugakubo 1-98, Kutsukake, Toyoake, Aichi 470-1192, Japan e-mail: [email protected] T. Tsukamoto Diagnostic Pathology, School of Medicine, Fujita Health University, Dengakugakubo 1-98, Kutsukake, Toyoake, Aichi 470-1192, Japan
Introduction Lymphangiomas are benign lesions, seen most commonly in the head and neck of children. They are thought to be derived from a congenital lymphatic malformation or anomaly rather than a neoplasm [1, 2]. Colonic lymphangioma is relatively rare, accounting for lower than 5 % of all lymphangiomas and 0.3 % of colorectal tumors, excluding adenoma or carcinoma [1, 3]. In general, lymphangiomas are diagnosed coincidentally during colonoscopy. Although many lymphangiomas are asymptomatic, they may cause complications such as intussusception or protein-losing enteropathy and require surgical treatment [4, 5]. We report a case of a gigantic lymphangioma of the ascending colon in a woman in her mid-30 s and review the relevant literature.
Case report A 34-year-old woman without any abdominal symptoms was referred by her gynecologist to our department for investigation of an abdominal cystic tumor detected by screening ultrasonography at a private fertility clinic. Her abdomen was soft and nearly flat without tenderness. She had no history of surgery and her blood test results, including the levels of tumor markers (CEA, CA19-9, CA125) and albumin, were all within normal limits. Abdominal computed tomography (CT) and magnetic resonance imaging (MRI) showed a multi-locular cystlike bulky mass occupying the abdomen and pelvis (Fig. 1a, b). Barium enema study showed a smooth filling defect in the ascending colon (data not shown), and colonoscopy revealed a submucosal tumor, 2 cm 9 2 cm, with a bridging fold and a positive cushion sign
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Surg Today Fig. 1 a Enhanced coronal computed tomography showed a bulky multi-lobular cystic lesion occupying the lower abdomen and pelvis. b T2-weighted magnetic resonance coronal imaging showed well-defined high signal intensity adjacent to the ascending colon
Fig. 2 a Colonoscopic findings. A round smooth surfaced pinkish mass covered by normal mucosa in the ascending colon with a positive cushion sign. b Endoscopic ultrasonography showed a multi-locular anechoic area in the submucosa and a normal layer of muscularis propria
Fig. 3 a Intraoperative appearance of the huge cystic lesion connecting to the ascending colon. b Macroscopically, the specimen was a 2.0 9 2.0 9 2.0 cm submucosal cystic tumor (black arrow) with marked extraluminal progression
(Fig. 2a). Endoscopic ultrasonography demonstrated a non-echoic multilocular structure in the third-layer of the bowel wall (Fig. 2b). We chose open laparotomy, rather
123
than endoscopic resection or laparoscopic surgery, because of the size of the tumor. After placing the patient in a supine position under general anesthesia, a
Surg Today
lumen m mm sm cyst
a
mp
b
mp
ss
mp
ss
mp c
d
Fig. 4 Pathological findings. Macroscopic and microscopic view of the cyst. a Cut surface showing a mucosal polypoid lesion (white arrow) and serosal large cysts (white arrow heads). b Histology of the mucosal lesion. Cysts are localized in the submucosa (sm), some containing serous fluid with clusters of lymphocytes (inset).
m mucosa, mm muscularis mucosae. (c and d) A cyst penetrates desmin positive muscularis propriae (mp) continuing to the subserosa (ss) (black arrows). HE staining (b and c). Immunohistochemistry against desmin (d). Original magnification 912.5 (b–d)
median skin incision was made. A bulky cystic mass occupied a substantial part of the abdominal cavity without adhering to other organs. Intra-abdominal exploration confirmed that the bilateral ovaries and uterus were grossly normal. A lesion considered as the tumor origin in the ascending colon (Fig. 3a) was resected, followed by a hand-sewn end-to end anastomosis. Her postoperative course was uneventful. A pregnancy was confirmed just 3 months after surgery and she gave birth to a healthy baby at term.
25 9 16 9 9 cm (Fig. 4a). Tissues were embedded in paraffin and sectioned for subsequent histopathological analyses. Serosal fluid containing lymphocyte clusters were found in the submucosal lesion (Fig. 4b). The cysts continued to subserosal areas penetrating the muscularis propriae as visualized with desmin immumostaining (Fig. 4c, d). Histology confirmed gigantic lymphangioma in the ascending colon. Histopathological examination revealed that the cystic lesions were lined with thin monolayer cells, which were immunopositive for CD31 but negative for other endothelial markers, including Von Willebrand factor (data not shown), CD34, and D2-40 (Podoplanin) (data not shown). Mesothelial markers, calretinin and WT-1, were not detected (data not shown), which excluded the possibility of cystic mesothelioma [6]. The final pathological diagnosis was lymphangioma, based mainly on the presence of lymphocyte clusters in a cyst and immunopositivity of CD31 to the lining of the cells.
Pathology The specimen consisted of several huge cysts filled with serous fluid and weighed approximately 930 g (Fig. 3b). It was fixed in 10 % formalin and cut longitudinally along the colonic duct. The cysts formed submucosal polypoid lesions as big as 2.5 9 2.5 9 2 cm in the lumen, which connected to the subserosal large cysts measuring
123
Surg Today
9
11
100 36
86
51
14
20
Fig. 5 Distribution of the numbers of colorectal lymphangiomas reported in the literature
Discussion Colorectal lymphangioma was initially reported by Chisholm et al. [7], and the first Japanese case was described by Yoshitoshi et al. in [8]. It has been reported more frequently in recent years since the widespread application of colonoscopy. We reviewed 346 cases of lymphangioma of the colon and rectum published in the English (Medline and Pubmed) and Japanese (Japan medical abstracts society) literature. Huguet et al. [1] reported that approximately 95 % of colorectal lymphangiomas were in patients from Eastern countries, with 85 % of the total cases being from Japan. The most common sites of colorectal lymphangioma seem to be the transverse colon, ascending colon, and cecum, in decreasing order of frequency (Fig. 5). In a series reported by Matsuda et al. [9], the tumor size was identified in 210 cases, with the maximum diameter ranging from 0.5 to 23.0 cm (mean 2.8 ± 2.1 cm). Only 3 of 250 colorectal lymphangiomas were over 10 cm in diameter, including this case, which represents the largest to be documented in the literature. Wegener et al. [10] classified lymphangioma into three types: simple, cavernous, and cystic. The classification of this entity is based on the size of the lymphatic cavity and the nature of the lymphatic wall. Cystic lymphangioma, as in this case, is the most common type and characterized by flat epithelial endothelium and the wall containing alternatively lymphoid tissue, small lymphatic spaces, smooth muscle, and foam cells [11]. Therefore, CT and MRI generally show the lymphangioma as a multi-loculated cyst-like structure [12].
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While many of the reported cases were diagnosed incidentally in patients without any symptoms, like our patient, several cases of acute abdominal pain caused by intussusception [4, 13] and protein-losing enteropathy were also reported [5]. Colonoscopy is presumably the most useful test for diagnosing lymphangioma of the large bowel, whereas in the past, barium enema study used to be the most diagnostic procedure [14]. The features that suggest lymphangioma of the colon are a pinkish, translucent, tense and lustrous surface, and change in shape caused by peristalsis, compression, or the patient’s position [15]. The differential diagnosis should include other submucosal lesions such as lipoma, leiomyoma, cyst, and cavernous hemangioma, as well as carcinoid, carcinoma, and mucocele [4]. We chose to perform open laparotomy rather than laparoscopic surgery or endoscopic resection for complete excision, because the lymphangioma was so large. Although the patient had no symptoms directly related to the colonic lymphangioma, there was potential for the development of symptoms if she became pregnant. Moreover, excision is required to prevent recurrence, rupture, torsion, and bleeding [16]. Chang reported a similar case of bulky mesenteric cystic lymphangioma in the transverse colon, dominating the pelvis of a woman in her late 20 s, who also consulted a gynecologist for primary sterility [17]. However, there is no solid evidence of a causal correlation between colonic lymphangioma and infertility. Although the indication for surgical intervention for colonic lymphangioma is still controversial, surgery should be performed for such patients, especially in their fertile years. Conflict of interest
We declare no conflict of interest.
References 1. Huguet KL, Metzger PP, Menke DM. Colorectal lymphangioma. Am Surg. 2007;73:414–6. 2. Vargas-Serrano B, Alegre-Bernal N, Cortina-Moreno B, Rodriguez-Romero R, Sanchez-Ortega F. Abdominal cystic lymphangiomas: US and CT findings. Eur J Radiol. 1995;19:183–7. 3. Stout AP. Tumors of the colon and rectum (excluding carcinoma and adenoma). In: Turell R, editor. Diseases of the colon and anorectum, 2nd ed. Philadelphia: Saunders; 1969. p. 295–311. 4. Matsuba Y, Mizuiri H, Murata T, Niimi K. Adult intussusception due to lymphangioma of the colon. J Gastroenterol. 2003;38:181–5. 5. Kim J, Han D, Hong CH, Lee HL, Kim JP, Sohn JH, et al. Colonic lymphangiomatosis associated with protein-losing enteropathy. Dig Dis Sci. 2005;50:1747–53. 6. Nagata H, Yonemura Y, Canbay E, Ishibashi H, Narita M, Mike M, et al. Differentiating a large abdominal cystic lymphangioma from multicystic mesothelioma: report of a case. Surg Today. 2013;44:1367–70. 7. Chisholm A, Hillkowitz P. Lymphangioma of the rectum. Am J Surg. 1932;17:281–2.
Surg Today 8. Yoshitoshi Y, Oda T, Utsumi Y, Kaneko E, Yamashita K. Case of lymphangioma of the ascending colon. Nihon Rinsho Japanese J Clin Med. 1965;23:2264–7. 9. Matsuda T, Matsutani T, Tsuchiya Y, Okihama Y, Egami K, Yoshioka M, et al. A clinical evaluation of lymphangioma of the large intestine: a case presentation of lymphangioma of the descending colon and a review of 279 Japanese cases. J Nihon Med Sch. 2001;68:262–5. 10. Wegner G. Lymphangiome. Arch Klin Chir. 1877;20:641. 11. Vara-Thorbeck C, Toscano Mendez R, Herrainz Hidalgo R, Mata Martin JM, Vara-Thorbeck R. Laparoscopic resection of a giant mesenteric cystic lymphangioma. Europ J Surg Acta Chirurgica. 1997;163:395–6. 12. Ros PR, Olmsted WW, Moser RP Jr, Dachman AH, Hjermstad BH, Sobin LH. Mesenteric and omental cysts: histologic classification with imaging correlation. Radiology. 1987;164:327–32.
13. Kim TO, Lee JH, Kim GH, Heo J, Kang DH, Song GA, et al. Adult intussusception caused by cystic lymphangioma of the colon: a rare case report. World J Gastroenterol. 2006;12:2130–2. 14. Berardi RS. Lymphangioma of the large intestine: report of a case and review of the literature. Dis Colon Rectum. 1974;17:265–72. 15. Kuramoto S, Sakai S, Tsuda K, Kaminishi M, Ihara O, Oohara T, et al. Lymphangioma of the large intestine. Report of a case. Dis Colon Rectum. 1988;31:900–5. 16. Chung SH, Park YS, Jo YJ, Kim SH, Jun DW, Son BK, et al. Asymptomatic lymphangioma involving the spleen and retroperitoneum in adults. World J Gastroenterol. 2009;15:5620–3. 17. Chang MS, Miyasaka Y, Mitsui T. A case of mesenteric cystic lymphangioma in an adult patient. J Jpn Surg Assoc. 2008;69:2114–8.
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CASE REPORT
Gigantic lymphangioma with marked extraluminal progression of the ascending colon: report of a case Hidetoshi Katsuno • Koutarou Maeda • Tsunekazu Hanai • Masahiro Mizuno • Takamitsu Kurashita • Tetsuya Tsukamoto
Received: 22 January 2014 / Accepted: 30 April 2014 Ó Springer Japan 2014
Abstract We report a case of gigantic cystic lymphangioma of the ascending colon excised through an open laparotomy. A 34-year-old woman consulted a gynecologist for treatment of infertility. Transvaginal ultrasonography revealed a cystic mass in the pelvis, and she was transferred to our hospital for further investigation. Abdominal enhanced computed tomography (CT) showed a bulky cystic mass, 25 cm or larger, in the abdominal and pelvic cavity. Colonoscopy revealed a cystic submucosal tumor with a cushion sign. Cystic lymphangioma was diagnosed and excised via open surgery as we could not exclude its malignant potential. Pathological examination confirmed lymphangioma. To our knowledge, this is the most gigantic lymphangioma of the colon documented in the literature. About 3 months after surgery, the patient discovered that she was pregnant and her first baby was delivered at term, uneventfully. Keywords
Colon Lymphangioma Fertility
H. Katsuno (&) K. Maeda T. Hanai M. Mizuno T. Kurashita Department of Surgery, School of Medicine, Fujita Health University, Dengakugakubo 1-98, Kutsukake, Toyoake, Aichi 470-1192, Japan e-mail: [email protected] T. Tsukamoto Diagnostic Pathology, School of Medicine, Fujita Health University, Dengakugakubo 1-98, Kutsukake, Toyoake, Aichi 470-1192, Japan
Introduction Lymphangiomas are benign lesions, seen most commonly in the head and neck of children. They are thought to be derived from a congenital lymphatic malformation or anomaly rather than a neoplasm [1, 2]. Colonic lymphangioma is relatively rare, accounting for lower than 5 % of all lymphangiomas and 0.3 % of colorectal tumors, excluding adenoma or carcinoma [1, 3]. In general, lymphangiomas are diagnosed coincidentally during colonoscopy. Although many lymphangiomas are asymptomatic, they may cause complications such as intussusception or protein-losing enteropathy and require surgical treatment [4, 5]. We report a case of a gigantic lymphangioma of the ascending colon in a woman in her mid-30 s and review the relevant literature.
Case report A 34-year-old woman without any abdominal symptoms was referred by her gynecologist to our department for investigation of an abdominal cystic tumor detected by screening ultrasonography at a private fertility clinic. Her abdomen was soft and nearly flat without tenderness. She had no history of surgery and her blood test results, including the levels of tumor markers (CEA, CA19-9, CA125) and albumin, were all within normal limits. Abdominal computed tomography (CT) and magnetic resonance imaging (MRI) showed a multi-locular cystlike bulky mass occupying the abdomen and pelvis (Fig. 1a, b). Barium enema study showed a smooth filling defect in the ascending colon (data not shown), and colonoscopy revealed a submucosal tumor, 2 cm 9 2 cm, with a bridging fold and a positive cushion sign
123
Surg Today Fig. 1 a Enhanced coronal computed tomography showed a bulky multi-lobular cystic lesion occupying the lower abdomen and pelvis. b T2-weighted magnetic resonance coronal imaging showed well-defined high signal intensity adjacent to the ascending colon
Fig. 2 a Colonoscopic findings. A round smooth surfaced pinkish mass covered by normal mucosa in the ascending colon with a positive cushion sign. b Endoscopic ultrasonography showed a multi-locular anechoic area in the submucosa and a normal layer of muscularis propria
Fig. 3 a Intraoperative appearance of the huge cystic lesion connecting to the ascending colon. b Macroscopically, the specimen was a 2.0 9 2.0 9 2.0 cm submucosal cystic tumor (black arrow) with marked extraluminal progression
(Fig. 2a). Endoscopic ultrasonography demonstrated a non-echoic multilocular structure in the third-layer of the bowel wall (Fig. 2b). We chose open laparotomy, rather
123
than endoscopic resection or laparoscopic surgery, because of the size of the tumor. After placing the patient in a supine position under general anesthesia, a
Surg Today
lumen m mm sm cyst
a
mp
b
mp
ss
mp
ss
mp c
d
Fig. 4 Pathological findings. Macroscopic and microscopic view of the cyst. a Cut surface showing a mucosal polypoid lesion (white arrow) and serosal large cysts (white arrow heads). b Histology of the mucosal lesion. Cysts are localized in the submucosa (sm), some containing serous fluid with clusters of lymphocytes (inset).
m mucosa, mm muscularis mucosae. (c and d) A cyst penetrates desmin positive muscularis propriae (mp) continuing to the subserosa (ss) (black arrows). HE staining (b and c). Immunohistochemistry against desmin (d). Original magnification 912.5 (b–d)
median skin incision was made. A bulky cystic mass occupied a substantial part of the abdominal cavity without adhering to other organs. Intra-abdominal exploration confirmed that the bilateral ovaries and uterus were grossly normal. A lesion considered as the tumor origin in the ascending colon (Fig. 3a) was resected, followed by a hand-sewn end-to end anastomosis. Her postoperative course was uneventful. A pregnancy was confirmed just 3 months after surgery and she gave birth to a healthy baby at term.
25 9 16 9 9 cm (Fig. 4a). Tissues were embedded in paraffin and sectioned for subsequent histopathological analyses. Serosal fluid containing lymphocyte clusters were found in the submucosal lesion (Fig. 4b). The cysts continued to subserosal areas penetrating the muscularis propriae as visualized with desmin immumostaining (Fig. 4c, d). Histology confirmed gigantic lymphangioma in the ascending colon. Histopathological examination revealed that the cystic lesions were lined with thin monolayer cells, which were immunopositive for CD31 but negative for other endothelial markers, including Von Willebrand factor (data not shown), CD34, and D2-40 (Podoplanin) (data not shown). Mesothelial markers, calretinin and WT-1, were not detected (data not shown), which excluded the possibility of cystic mesothelioma [6]. The final pathological diagnosis was lymphangioma, based mainly on the presence of lymphocyte clusters in a cyst and immunopositivity of CD31 to the lining of the cells.
Pathology The specimen consisted of several huge cysts filled with serous fluid and weighed approximately 930 g (Fig. 3b). It was fixed in 10 % formalin and cut longitudinally along the colonic duct. The cysts formed submucosal polypoid lesions as big as 2.5 9 2.5 9 2 cm in the lumen, which connected to the subserosal large cysts measuring
123
Surg Today
9
11
100 36
86
51
14
20
Fig. 5 Distribution of the numbers of colorectal lymphangiomas reported in the literature
Discussion Colorectal lymphangioma was initially reported by Chisholm et al. [7], and the first Japanese case was described by Yoshitoshi et al. in [8]. It has been reported more frequently in recent years since the widespread application of colonoscopy. We reviewed 346 cases of lymphangioma of the colon and rectum published in the English (Medline and Pubmed) and Japanese (Japan medical abstracts society) literature. Huguet et al. [1] reported that approximately 95 % of colorectal lymphangiomas were in patients from Eastern countries, with 85 % of the total cases being from Japan. The most common sites of colorectal lymphangioma seem to be the transverse colon, ascending colon, and cecum, in decreasing order of frequency (Fig. 5). In a series reported by Matsuda et al. [9], the tumor size was identified in 210 cases, with the maximum diameter ranging from 0.5 to 23.0 cm (mean 2.8 ± 2.1 cm). Only 3 of 250 colorectal lymphangiomas were over 10 cm in diameter, including this case, which represents the largest to be documented in the literature. Wegener et al. [10] classified lymphangioma into three types: simple, cavernous, and cystic. The classification of this entity is based on the size of the lymphatic cavity and the nature of the lymphatic wall. Cystic lymphangioma, as in this case, is the most common type and characterized by flat epithelial endothelium and the wall containing alternatively lymphoid tissue, small lymphatic spaces, smooth muscle, and foam cells [11]. Therefore, CT and MRI generally show the lymphangioma as a multi-loculated cyst-like structure [12].
123
While many of the reported cases were diagnosed incidentally in patients without any symptoms, like our patient, several cases of acute abdominal pain caused by intussusception [4, 13] and protein-losing enteropathy were also reported [5]. Colonoscopy is presumably the most useful test for diagnosing lymphangioma of the large bowel, whereas in the past, barium enema study used to be the most diagnostic procedure [14]. The features that suggest lymphangioma of the colon are a pinkish, translucent, tense and lustrous surface, and change in shape caused by peristalsis, compression, or the patient’s position [15]. The differential diagnosis should include other submucosal lesions such as lipoma, leiomyoma, cyst, and cavernous hemangioma, as well as carcinoid, carcinoma, and mucocele [4]. We chose to perform open laparotomy rather than laparoscopic surgery or endoscopic resection for complete excision, because the lymphangioma was so large. Although the patient had no symptoms directly related to the colonic lymphangioma, there was potential for the development of symptoms if she became pregnant. Moreover, excision is required to prevent recurrence, rupture, torsion, and bleeding [16]. Chang reported a similar case of bulky mesenteric cystic lymphangioma in the transverse colon, dominating the pelvis of a woman in her late 20 s, who also consulted a gynecologist for primary sterility [17]. However, there is no solid evidence of a causal correlation between colonic lymphangioma and infertility. Although the indication for surgical intervention for colonic lymphangioma is still controversial, surgery should be performed for such patients, especially in their fertile years. Conflict of interest
We declare no conflict of interest.
References 1. Huguet KL, Metzger PP, Menke DM. Colorectal lymphangioma. Am Surg. 2007;73:414–6. 2. Vargas-Serrano B, Alegre-Bernal N, Cortina-Moreno B, Rodriguez-Romero R, Sanchez-Ortega F. Abdominal cystic lymphangiomas: US and CT findings. Eur J Radiol. 1995;19:183–7. 3. Stout AP. Tumors of the colon and rectum (excluding carcinoma and adenoma). In: Turell R, editor. Diseases of the colon and anorectum, 2nd ed. Philadelphia: Saunders; 1969. p. 295–311. 4. Matsuba Y, Mizuiri H, Murata T, Niimi K. Adult intussusception due to lymphangioma of the colon. J Gastroenterol. 2003;38:181–5. 5. Kim J, Han D, Hong CH, Lee HL, Kim JP, Sohn JH, et al. Colonic lymphangiomatosis associated with protein-losing enteropathy. Dig Dis Sci. 2005;50:1747–53. 6. Nagata H, Yonemura Y, Canbay E, Ishibashi H, Narita M, Mike M, et al. Differentiating a large abdominal cystic lymphangioma from multicystic mesothelioma: report of a case. Surg Today. 2013;44:1367–70. 7. Chisholm A, Hillkowitz P. Lymphangioma of the rectum. Am J Surg. 1932;17:281–2.
Surg Today 8. Yoshitoshi Y, Oda T, Utsumi Y, Kaneko E, Yamashita K. Case of lymphangioma of the ascending colon. Nihon Rinsho Japanese J Clin Med. 1965;23:2264–7. 9. Matsuda T, Matsutani T, Tsuchiya Y, Okihama Y, Egami K, Yoshioka M, et al. A clinical evaluation of lymphangioma of the large intestine: a case presentation of lymphangioma of the descending colon and a review of 279 Japanese cases. J Nihon Med Sch. 2001;68:262–5. 10. Wegner G. Lymphangiome. Arch Klin Chir. 1877;20:641. 11. Vara-Thorbeck C, Toscano Mendez R, Herrainz Hidalgo R, Mata Martin JM, Vara-Thorbeck R. Laparoscopic resection of a giant mesenteric cystic lymphangioma. Europ J Surg Acta Chirurgica. 1997;163:395–6. 12. Ros PR, Olmsted WW, Moser RP Jr, Dachman AH, Hjermstad BH, Sobin LH. Mesenteric and omental cysts: histologic classification with imaging correlation. Radiology. 1987;164:327–32.
13. Kim TO, Lee JH, Kim GH, Heo J, Kang DH, Song GA, et al. Adult intussusception caused by cystic lymphangioma of the colon: a rare case report. World J Gastroenterol. 2006;12:2130–2. 14. Berardi RS. Lymphangioma of the large intestine: report of a case and review of the literature. Dis Colon Rectum. 1974;17:265–72. 15. Kuramoto S, Sakai S, Tsuda K, Kaminishi M, Ihara O, Oohara T, et al. Lymphangioma of the large intestine. Report of a case. Dis Colon Rectum. 1988;31:900–5. 16. Chung SH, Park YS, Jo YJ, Kim SH, Jun DW, Son BK, et al. Asymptomatic lymphangioma involving the spleen and retroperitoneum in adults. World J Gastroenterol. 2009;15:5620–3. 17. Chang MS, Miyasaka Y, Mitsui T. A case of mesenteric cystic lymphangioma in an adult patient. J Jpn Surg Assoc. 2008;69:2114–8.
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