lymphangioma is a developmental malformation of lymphatic tissue , characterized by submucosal tumors covered with normal mucosa.1 these benign tumors are commonly found in children , and although they may develop anywhere , the tumors are usually found in the head , neck , and axillary regions , and rarely in the gastrointestinal tract.2 most reported cases of lymphangioma of the colon are solitary ; few cases involve multiple lymphangioma , also known as colonic lymphangiomatosis.1 colonic lymphangiomas are typically asymptomatic , are discovered only incidentally , and usually do not require treatment.3,4,5 however , in some cases , they may cause abdominal pain , bleeding , intussusception , or protein - losing enteropathy , requiring surgical or endoscopic resection.1,6
a 38-year - old man visited our comprehensive medical center for a general check - up .
he had no previous medical problems other than diabetes mellitus and essential hypertension , which were diagnosed 1 year prior at another hospital .
laboratory test results , including the levels of tumor markers carcinoembryonic antigen and carbohydrate antigen , were all within normal ranges , except the levels of triglycerides at 206 mg / dl ( normal , 10 to 180 ) and hemoglobin a1c at 9.3% ( normal , 4.0 to 6.0 ) .
esophagogastroduodenoscopy showed hyperemic mucosal change in the antrum and body of the stomach , with multiple shallow erosions in the se - cond portion of the duodenum .
an initial colonoscopy revealed multiple , variably sized cystic mass lesions in the mid - portion of the ascending colon up to the proximal portion of the transverse colon ( fig .
computed tomography ( ct ) revealed no abnormalities other than a fatty liver ( fig .
a solution composed of hypertonic saline , epinephrine , and methylene blue was injected around some of the cystic masses and biopsy specimens were taken with snare electrocautery ( fig .
incision on one of the masses using a dual knife caused clear yellow fluid to flow out from the mass , after which the mass began to shrink .
the biopsied tissue revealed normal colonic mucosa and markedly dilated lymphatic vessels in the submucosa .
immunohistochemical staining for cd34 and d2 - 40 showed a positive reaction along the lining cells of the dilated lymphatic channels ( fig .
cd34 is an endothelial cell marker in different vascular beds in human tissues,7 and the monoclonal antibody d2 - 40 is a marker of lymphatic endothelium.8 d2 - 40 does not stain vascular endothelium .
eight months later , the patient underwent a third colonoscopic examination that showed normal colonic mucosa without the protruded lymphangiomatosis previously been observed in the ascending and transverse colon . no scarring or trace of the former lesions
lymphangioma is regarded as a developmental malformation of lymphatic tissue ; obstruction or agenesis of lymphatic tissue causes abnormal dilatation and proliferation of the lymphatic channel , leading to the formation of a cystic mass.3,4 secondary lymphangioma formation can be caused by abdominal trauma , lymphatic obstruction , inflammatory processes , cancer , surgery , or radiation . in the present case , colonic lymphangiomatosis was discovered in adulthood , suggesting that the lesion developed secondary to a local disturbance of the lymphatic circulation . however , the patient had no specific history related to colonic lymphangiomatosis , and abdominal ct showed no comorbid disease ; thus , it would be more rational to conclude that lymphangioma developed primarily in the fetal period in this case , and was silent until detection on medical examination
. lymphangiomas rarely occur in the abdomen , and the mesentery , omentum , mesocolon , and retroperitoneum are the most common sites.9 the incidence of lymphangioma in the intestinal wall is very low , and few cases of multiple colonic lymphangioma have been reported.1 in the present case , multiple lymphangiomas of the colon , known as colonic lymphangiomatosis , were observed , and had resolved by the time of the third colonoscopic examination .
it is considered benign ; no cases of malignant transformation have been reported.1 trials have been conducted to explain how lymphangiomas may be resolved .
several investigators have suggested that increased pressure in the lymphatic system may overcome incomplete obstructions .
others have hypothesized that resolution may occur through the establishment of alternative routes of lymphatic drainage.10 the latter theory seems to fit well with the present case .
it is possible that in the course of making incisions and taking excisional biopsies of the colonic lymphangiomatosis , conduits that drain lymphatic outflow toward the colonic lumen were formed , leading to the complete resolution of the lymphangiomatosis .
there is no standard approach to the treatment of intra - abdominal or intraluminal lymphangiomas .
historically , both were treated by surgical resection due to uncertainty regarding the nature of the lesion . however , since lymphangioma is a benign tumor , and most intra - abdominal or intestinal lymphangiomas are asymptomatic , they usually do not require treatment.3,4,5 kochman et al.5 suggested that asymptomatic lymphangiomas should be observed rather than treated .
large lymphangiomas may produce bowel obstruction , intussusception , bleeding , anemia , and protein - losing enteropathy , in addition to symptoms such as abdominal pain , vomiting , and diarrhea.6 complete surgical excision is considered the treatment of choice for these large , symptomatic lymphangiomas.1,6 for pedunculated tumors < 2 cm , recent reports indicate a trend towards the use of endoscopic polypectomy.11 in the present case , no treatment other than several incisions and excisional biopsies was attempted because the observed tumors were all small , sessile , and did not exhibit symptoms or cause complications . in a follow - up study after the incisions and excisional biopsies , we found that such tumors were completely resolved .
our assumption is that conduits were made at some sites of incisions or excisions , and lymphatic drainage through those conduits from the lymphatic channel to the colonic lumen brought about the resolution of the colonic lymphangiomatosis . during 8 months of surveillance after the procedure , no complications such as bleeding or protein - losing enteropathy were noticed .
this is a case report of a patient in whom dozens of lymphangiomas were found in the ascending colon up to the transverse colon , which was observed and confirmed through colonoscopy and biopsy .
the colonic lymphangiomatosis was observed to be completely resolved in follow - up studies , without any treatment .
 lymphangioma is an uncommon malformation of the lymphatic system that involves a benign proliferation of the lymphatics , with no established treatment method . 
 multiple colonic lymphangioma , or colonic lymphangiomatosis , is an extremely rare condition . 
 we report a case of colonic lymphangiomatosis that was detected during a colonoscopic examination conducted as part of a general health check - up . 
 the lesion completely resolved after excisional biopsy . 
INTRODUCTION
CASE REPORT
DISCUSSION