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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
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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.1 Departments 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
Intussusception is highly uncommon during the
old mother, who had presented with 33 hours of pre
neonatal period and is exceedingly rare in premature
mature rupture of membranes and confirmed histologic
infants. The rarity of the condition, and the difficulty
chorioamnionitis. The infant’s Apgar score was 1 at
differentiating it from a common neonatal disease
one minute and 4 at five minutes. She had many pro
such as necrotizing enterocolitis (NEC), often delays
blems associated with prematurity, including respi
diagnosis and surgical intervention, which contributes
ratory distress syndrome requiring high frequency
1, 2
to increased morbidity and mortality.
In this study,
ventilator and surfactant replacement therapy, refrac
we present a case of multiple intussusceptions in an
tory hypotension requiring inotropics and hydrocor
extremely premature infant, born in gestational week
tisone, thrombocytopenia, suspected sepsis, and patent
+1
23 , who was misdiagnosed with NEC. To our know
ductus arteriosus requiring surgical ligation after failed
ledge, this is the most premature case that has survived
ibuprofen treatment at 8 days of life. At 12 days of life,
with multiple intussusceptions.
she exhibited bilious gastric aspirates, and plain ra diography showed a gasless abdomen for two days.
Case report
Abdominal ultrasonography was not performed. Enteral feeding had not started until that time and there was
A female infant weighing 640 g was born at gesta tional week 23+1 by cesarean section to a 33-year-
no hematochezia. Laboratory results showed leuko cytosis: white blood cell count (WBC) 25,050 (seg mented neutrophil 74%)/mm3, hemoglobin 11 g/dL,
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
platelet 161,000/mm3, negative C-reactive 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
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김하수 등 : -
초극소미숙아에서 발생한 다발성 장중첩증 -
radiography. Vancomycin and meropenem were
moderate congestion, and two additional intussus
treatment. On day 20, we performed an abdominal
ceptions, which were successfully reduced manually,
radiograph due to marked abdominal distension and
at 20 cm distal from Treitz ligament (Fig. 2). No lead
desaturation, which demonstrated free abdominal air
point was detected. Upon examination of the gastro
(Fig. 1). Laboratory results were not specific: WBC
intestinal tube, no abnormality was observed. Patho
3
19,670 (segmented neutrophil 69%)/mm , hemoglobin 3
logic evaluation of the specimen revealed inflammation
9.6 g/dL, platelet 144,000/mm , negative C-reactive
with congestion at the site of intussusceptions. The
protein, pH 7.234, base excess -1.5 mEq/L, bicarbo
postoperative course was uneventful. The enteral
nate 26.3 mEq/L. Our diagnosis was NEC with
feeding was started 7 days after the operation, and
perforation. A laparotomy was performed imme
full enteral feeding was performed at 42 days of life.
diately. The intraoperative findings were ileo-ileal
Repair of the ileostomy was performed successfully
intussusception with small bowel perforation,
with primary end-to-end anastomosis at 5 months
requiring ileostomy, approximately 20 cm proximal
of life. The child was discharged in excellent general
to the ileo-cecal valve. The intestinal wall displayed
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.6-8 All
Fig. 1. Abdominal radiography that shows small intestine dilated loops and free abdominal air (arrow).
Ileo-ileal intussusception Fig. 2. Gross finding of intussusceptions.
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Perforation
Ha-Su Kim, et al. : - Intussusceptions in Premature Infant -
of the patients (25 weeks, 27 weeks, and 28 weeks)
condition was too severe to allow surgery in the patient,
were diagnosed to NEC with perforation at first time,
but prompt laparotomy following diagnosis is crucial
but after laparotomy (at 39 days, at 25 days, and at 33
for achieving better outcomes.1
days), they were confirmed to intussusception same 6-8
as this case.
Diagnosis time was slight late compared 6
Intussusception in neonates is an extremely rare clinical entity and can often be confused with other
to this case (at 20 days). Just one case was performed
causes of intestinal obstruction and intestinal disten
abdominal ultrasonography, intussusception was not
sion. There are neither definite risk factors nor clinical
6, 8
detected by sonography. Two of them
were ileo-
or diagnostic characteristics to definitively differentiate
ileo-colic intussusceotions and one case had no infor
between intussusceptions and NEC in preterm infants.
mation about that. The etiology of neonatal intussus
A high degree of suspicion is needed to avoid misdiag
ceptions in premature infants remains unclear. As in
nosis.
the present case, intestinal hypoperfusion or ischemia
Conflicts of interest
would result in dysmotility and stasis, but accelerated peristalsis would occur in the early or recovering
The authors have no conflicts of interest relevant
phase of the event, potentially acting as a functional
to this article
leading point.9 These clinicopathological characteristics may be
References
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
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. 3) Slam KD, Teitelbaum DH. Multiple sequential intussuscep tions causing bowel obstruction in a preterm neonate. J Pediatr Surg 2007;42:1279-81. 4) Margenthaler JA, Vogler C, Guerra OM, Limpert JN, Weber TR, Keller MS. Pediatric surgical images: Small bowel intus susception in a preterm infant. J Pediatr Surg 2002;37:1515-7. 5) Al-Salem AH, Habash BM. Ileoileal intussusception: a report of four cases. Ann Saudi Med 2000;20:310-2. 6) Kim SW, Lee JJ, Yoo BH, Cha SJ, Lee JB, Kwon GY. Double intussusception in a preterm infant. Korean J Perinatol 2010; 21:408-11. 7) Kim DY, Kim SC, Kim AR, Kim KS, Pi SY, Kim IK. Intes tinal perforations in very low birth weight infants. J Korean Assoc Pediatr Surg 2001;7:112-7. 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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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.
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.
=국문초록=
초극소 미숙아에서 발생한 다발성 장중첩증 동아대학교 의과대학 소아과학교실1, 동아대학교 의과대학 외과학교실2, 부산대학교 어린이병원 소아청소년과3
김하수1・김현아1・김성흔2・변신연3・김묘징1 장중첩증은 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
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