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Received : July 16, 2014, Revised: August 10, 2014 Accepted : September 18, 2014
Correspondence : Myo-Jing Kim, M.D.
Department of Pediatrics, Dong-A University College of Medicine, 1,3-ga, Dongdaesin-dong, Seo-gu, Busan 602-714, Korea
Tel : +82-51-240-2589, Fax : +82-51-242-2765 E-mail : [email protected]
Copyrightⓒ By The Korean Society of Perinatology
대 한 주 산 회 지 제25권 제3호, 2014 Korean J Perinatol Vol.25, No.3, Sep., 2014 http://dx.doi.org/10.14734/kjp.2014.25.3.202
증 례
old mother, who had presented with 33 hours of pre
mature rupture of membranes and confirmed histologic chorioamnionitis. The infant’s Apgar score was 1 at one minute and 4 at five minutes. She had many pro
blems associated with prematurity, including respi
ratory distress syndrome requiring high frequency ventilator and surfactant replacement therapy, refrac
tory hypotension requiring inotropics and hydrocor
tisone, thrombocytopenia, suspected sepsis, and patent ductus arteriosus requiring surgical ligation after failed ibuprofen treatment at 8 days of life. At 12 days of life, she exhibited bilious gastric aspirates, and plain ra
diography showed a gasless abdomen for two days.
Abdominal ultrasonography was not performed. Enteral feeding had not started until that time and there was no hematochezia. Laboratory results showed leuko
cytosis: white blood cell count (WBC) 25,050 (seg
mented neutrophil 74%)/mm3, hemoglobin 11 g/dL, platelet 161,000/mm3, ne gative Creactive protein, pH 7.363, base excess 3.2 mEq/L, bicarbonate 20.9 mEq/L, negative blood culture. NEC was initially suspected, but abdominal distension was minimal, and no pneumatosis intestinalis was found on subsequent Intussusception is highly uncommon during the
neonatal period and is exceedingly rare in premature infants. The rarity of the condition, and the difficulty differentiating it from a common neonatal disease such as necrotizing enterocolitis (NEC), often delays diagnosis and surgical intervention, which contributes to increased morbidity and mortality.1, 2 In this study, we present a case of multiple intussusceptions in an extremely premature infant, born in gestational week 23+1, who was misdiagnosed with NEC. To our know
ledge, this is the most premature case that has survived with multiple intussusceptions.
Case report
A female infant weighing 640 g was born at gesta
tional week 23+1 by cesarean section to a 33year
Multiple Intussusceptions in an Extremely Premature Infant
Ha-Su Kim, M.D.
1, Hyun-A Kim, M.D.
1, Sung-Heun Kim, M.D.
2, Shin-Yun Byun, M.D.
3, and Myo-Jing Kim, M.D.
1Departments of Pediatrics1 and Surgery2, Dong-A University, College of Medicine, Busan, Korea Department of Pediatrics3, Pusan National University School of Medicine, Yangsan, Korea
Intussusception in premature infants is very rare. Here, we report a case of multiple intussusceptions in an extremely preterm infant, born at 23+1 weeks gestation, who underwent an explolaparotomy, for bowel perforation and misdiagnosed necrotizing enterocolitis, at 20 days of life. To our knowledge, this is the most prematurely born baby that has survived with multiple intussusceptions.
Key Words : Intussusception, Extremely premature infant
김하수 등 : - 초극소미숙아에서 발생한 다발성 장중첩증 -
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radiography. Vancomycin and meropenem were treatment. On day 20, we per formed an abdominal radiograph due to marked abdo minal distension and desaturation, which demonstrated free abdominal air (Fig. 1). Laboratory results were not specific: WBC 19,670 (segmented neutrophil 69%)/mm3, hemoglobin 9.6 g/dL, platelet 144,000/mm3, negative Creactive protein, pH 7.234, base excess 1.5 mEq/L, bicarbonate 26.3 mEq/L. Our diagnosis was NEC with perforation. A laparotomy was per formed imme
diately. The intraoperative findings were ileoileal intussusception with small bowel perforation, requiring ileostomy, approximately 20 cm proximal to the ileocecal valve. The intestinal wall displayed
moderate congestion, and two additional intussus
ceptions, which were successfully reduced manually, at 20 cm distal from Treitz ligament (Fig. 2). No lead point was detected. Upon examination of the gastro
intestinal tube, no abnormality was observed. Patho
logic evaluation of the specimen revealed inflammation with congestion at the site of intussusceptions. The postoperative course was uneventful. The enteral feeding was started 7 days after the operation, and full enteral feeding was performed at 42 days of life.
Repair of the ileostomy was performed successfully with primary endtoend anastomosis at 5 months of life. The child was discharged in excellent general condition.
Discussion
Intussusception occurs very infrequently in the neonatal period, with a reported incidence ranging from 0.3% to 2.7% in the first month of life, and results in less than 3% of all neonatal bowel obstructions.3, 4 Small bowel intussusceptions occur in less than 10%
of all age groups, yet they are more common up to 68
% in premature neonates, particularly in the ileum.3, 5 In Korea, there were three postnatal intussuscep
tions in prematurity were reported previously.68 All
Fig. 1. Abdominal radiography that shows small intestine dilated loops and free abdominal air (arrow).
Perforation Ileo-ileal intussusception
F ig. 2. Gross finding of intussusceptions.
Ha-Su Kim, et al. : - Intussusceptions in Premature Infant -
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of the patients (25 weeks, 27 weeks, and 28 weeks) were diagnosed to NEC with perforation at first time, but after laparotomy (at 39 days, at 25 days, and at 33 days), they were confirmed to intussusception same as this case.68 Diagnosis time was slight late compared to this case (at 20 days). Just one case6 was performed abdominal ultrasonography, intussusception was not detected by sonography. Two of them6, 8 were ileoileocolic intussusceotions and one case had no infor
mation about that. The etiology of neonatal intussus
ceptions in premature infants remains unclear. As in the present case, intestinal hypoperfusion or ischemia would result in dysmotility and stasis, but ac celerated peristalsis would occur in the early or re covering phase of the event, potentially acting as a func tional leading point.9
These clinicopathological characteristics may be responsible for diagnostic confusion with other condi tions, particularly with NEC, which is relatively more common in these patients. The mean time from onset of signs and symptoms to surgical intervention was 9.9±11.8 days in reported cases of intussusceptions in neonates.9, 10 Indeed, Wang et al.10 reported that in
tussusceptions must be highly suspected in a neonate who is diagnosed with NEC, but who has a more stable course than would be expected. Avansino et al.11 men
tioned that the most common imaging finding in pre
mature neonates with intussusceptions is dilated bowel loops. Similarly, the abdominal radiograph of the patient presented in this study revealed small intestine dilated loops. Pneumatosis intestinalis or portal venous gas was not found. Recent studies reported that abdominal ultrasound was helpful for rapid and accurate diagnosis of intussusceptions, as the clinical picture was not explained by the initially suspected NEC.2, 9 But this case, we did not performed abdominal ultrasonography due to no doubt to intussusception. The underlying
condition was too severe to allow surgery in the patient, but prompt laparotomy following diagnosis is crucial for achieving better outcomes.1
Intussusception in neonates is an extremely rare clinical entity and can often be confused with other causes of intestinal obstruction and intestinal disten
sion. There are neither definite risk factors nor clinical or diagnostic characteristics to definitively differentiate between intussusceptions and NEC in preterm infants.
A high degree of suspicion is needed to avoid misdiag
nosis.
Conflicts of interest
The authors have no conflicts of interest relevant to this article
References
1) Shima Y, Kumasaka S, Yashiro K, Nakajima M, Migita M.
Intussusception in an extremely premature infant following bacterial sepsis. Eur J Pediatr 2012;171:725-7.
2) Loukas I, Baltogiannis N, Plataras C, Skiathitou AV, Siaha- nidou S, Geroulanos G. Intussusception in a premature neo- nate: a rare often misdiagnosed cause of intestinal obstruction.
Case Rep Med 2009;2009:607989.
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6) Kim SW, Lee JJ, Yoo BH, Cha SJ, Lee JB, Kwon GY. Double intussusception in a preterm infant. Korean J Perinatol 2010;
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8) Cho HJ, Kim JY, Kim MJ, Kim YH, Jung JA, Yang S, et al.
A case of intussusception diagnosed by exploratory laparo- tomy in a very low birth weight infant. J Korean Soc Neonatol
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10) Wang NL, Yeh ML, Chang PY, Sheu JC, Chen CC, Lee HC, et al. Prenatal and neonatal intussusception. Pediatr Surg Int 1998;13:232-6.
11) Avansino JR, Bjerke S, Hendrickson M, Stelzner M, Sawin R.
Clinical features and treatment outcome of intussusception in premature neonates. J Pediatr Surg 2003;38:1818-21.
2007;14:98-102.
9) Martinez Biarge M, Garcia-Alix A, Luisa del Hoyo M, Alarcon A, Saenz de Pipaon M, Hernandez F, et al. Intussusception in a preterm neonate; a very rare, major intestinal problem-- systematic review of cases. J Perinat Med 2004;32:190-4.
= 국 문 초 록 =
초극소 미숙아에서 발생한 다발성 장중첩증
동아대학교 의과대학 소아과학교실
1, 동아대학교 의과대학 외과학교실
2, 부산대학교 어린이병원 소아청소년과
3김하수
1・김현아
1・김성흔
2・변신연
3・김묘징
1장중첩증은 미숙아에서 매우 드문 질환이다. 본 저자들은 임신 나이 23주 1일로 출생한 초극소 미숙아에서 생후 20일 에 괴사성 장염과 관련된 장천공이 의심되어 시험적 개복술을 시행하였으나 장중첩증으로 최종 진단된 증례를 경험하 였기에 보고하고자 한다. 본 증례는 다발성 장중첩증으로 진단되어 수술 후 생존한 가장 어린 미숙아로 증례 보고의 의 의가 있다.
중심 단어 : 장중첩증, 초극소 미숙아
