the patient was a 41-week - gestational - age infant girl with a birth weight of 3300 g. after an uneventful pregnancy , the patient was delivered spontaneously and healthy until this episode . at 35 days of age , the patient was admitted to our hospital with a one - day history of anorexia , vomiting and recurrent diarrhea . on admission , conscious level was glasgow coma scale e2 v2 m2 , and blood pressure was 104/flat . physical examination revealed a distended and tender abdomen .
laboratory values were significant for a c- reactive protein ( crp ) of 1.7 mg / dl ( normal range was less than 0.3 mg / dl ) .
the patient was suspected intestinal obstruction due to enteritis , antibiotics were started ( abpc / mcipc and amk ) .
4 hours after admission , she was pale with a temperature of 39.2 c and 220 per minutes pulse .
blood pressure was low , and the fevers , vomiting , and diarrhea persisted and worsened despite antibiotics .
she failed to respond to medical managements and died 27 hours after admission due to multiple organ failures .
the cyst was unilocular , contained watery fluid , did not communicate with the adjacent intestinal lumen ( fig .
microscopic finding of the ileocecal duplication cyst had an undefined mucosal lining though it resembled pyrolic glands or duodenal glands .
duplications of the alimentary tract can be located anywhere between the mouth and the anus.4 they are most commonly found in the ileum ( 30% ) , ileocecal valve ( 30% ) , duodenum ( 10% ) , stomach ( 8% ) , jejunum ( 8% ) , colon ( 7% ) and rectum ( 5% ) with the half of the all lesions may contain ectopic gastric mucosa.5,6 most alimentary tract duplications are cystic ( 80%90% ) , with the remainder being tubular.7 they have three common features , hollow structure that has a wall of smooth muscle often shared with the contiguous intestinal tract , lined by mucous membrane similar to some part of the alimentary canal , and usually attached to some part of the alimentary tube.8 the clinical symptoms are highly variable from minor digestive problems to intestinal obstruction , gastrointestinal bleeding , or perforation.9 most patients present before the age of 2 years with obstructive symptoms , pain , or an abdominal mass .
the presence of gastric or pancreatic mucosa can lead to peptic ulceration , perforation , and hemorrhage .
early diagnosis and treatment reduces the risk of dangerous complications , such as bowel perforation , bleeding , obstruction , and malignant alteration .
resection of duplication alone is the treatment of choice , and in uncomplicated cases laparoscopic resection is advised .
surgically treated intestinal duplications have good prognosis , however , such duplications can have a fatal consequence if not properly treated .
possible complications like bowel perforation , bleeding , obstruction , and malignant alterations are the reason why all duplications should be surgically treated at the time of diagnosis.10 alimentary tract duplications can be easily mis - diagnosed as other disorders.5 pyloric and duodenal duplications can mimic hypertrophic pyloric stenosis or choledochal cyst.11,12 ovarian cyst should be considered in female patients , and adolescent patients can sometimes have a presumptive diagnosis of crohn s disease.5 yousefzadeh et al13 showed that duplication of the colon and rectum clinically presented as abdominal pain , vomiting and chronic constipation , a significant number of patients were thought to have hirschsprung s disease . in our fatal case ,
there are two reasons for this diagnostic difficulty : first , fevers , vomiting , and recurrent diarrhea persisted and worsened despite antibiotics .
the patient was suspected intestinal obstruction due to enteritis , however , actually the ileocecal duplication cyst led to intestinal obstruction and necrotizing ileum ; second , the patient died in an instant due to multiple organ failures .
if this patient was considered for exploratory laparotomy , she might be able to be alive .
however , the patient was getting worse very quickly due to multiple organ failures and showed intractable psvt .
exploratory laparotomy was impossible . it is demonstrated a high association of other anomalies in these patients including skeletal and urologic abnormalities,13,14 however , in this case , there were no other congenital anomalies .
sudden unexpected death of infants associated with duplication cysts is extremely rare with only 4 cases reported in medical literature to date ( table 1 ) .
puligandla ps et al5 and byard rw15 reported the patients presented with group b streptococcal sepsis and clostridium perfringens sepsis , respectively .
kibayashi et al16 reported the patient died at home after being diagnosed as having gastroenteritis .
in present case , though blood , stool and cyst cultures were negative , the patient had high fever and crp level elevated .
infection is suspected to be a risk factor for sudden and unexpected death in infants with duplication cysts . in all cases ,
this potential diagnosis should be borne in mind for a patient who complains of abdominal symptoms with an unknown cause , and duplication cyst should be recognized as a fatal cause in infant .
 duplications of the alimentary tract are very rare . a one - month - old female presented with symptoms of anorexia , vomiting and continuous watery diarrhea . 
 the plain abdominal radiograph showed thickened intestinal wall and signs of small bowel obstruction . 
 the fevers , vomiting , and continuous wartery diarrhea persisted despite antibiotics , and worsened . 
 the patient failed to respond to medical managements , 27 hours after admission , the patient died due to multiple organ failures . 
 the autopsy was performed , small bowel obstruction due to an ileocecal duplication cyst ( 3  3 cm ) was recognized . 
 the ileocecal duplication cyst was attached to the ileum which was changed edematous and necrotic . 
 this potential diagnosis should be borne in mind for a patient who complains of abdominal symptoms with an unknown cause , and duplication cyst should be recognized as a fatal cause in infant . 
Report of Case
Discussion