DOI QR코드

DOI QR Code

쌍생아에서 선천 거대세포바이러스 감염 증례

Discordant Congenital Cytomegalovirus Infection in Twins

  • 김이슬 (성균관대학교 의과대학 삼성서울병원 소아청소년과) ;
  • 강지만 (성균관대학교 의과대학 삼성서울병원 소아청소년과) ;
  • 이지훈 (성균관대학교 의과대학 삼성서울병원 소아청소년과) ;
  • 장윤실 (성균관대학교 의과대학 삼성서울병원 소아청소년과) ;
  • 박원순 (성균관대학교 의과대학 삼성서울병원 소아청소년과) ;
  • 김예진 (성균관대학교 의과대학 삼성서울병원 소아청소년과)
  • Kim, Yi-Seul (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kang, Ji-Man (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Lee, Ji-Hoon (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Chang, Yoon Sil (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Park, Won Soon (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine) ;
  • Kim, Yae-Jean (Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine)
  • 투고 : 2016.09.15
  • 심사 : 2016.10.13
  • 발행 : 2017.04.25

초록

거대세포바이러스 감염은 선천감염의 가장 흔한 원인 중의 하나이다. 본 증례는 쌍생아에서 다른 임상 양상을 보인 선천 거대세포바이러스 감염 증례에 대한 국내 첫 보고이다. 31세 초산부가 쌍생아(2 융모막, 2 양막)를 임신하였다. 임신 34주 산전 초음파에서 한쪽 태아의 크기가 작고 양측 뇌실 확장증이 발견되었으며 임신 37주경에 제왕절개로 분만하였다. 첫 번째 신생아는 남자로 출생 체중 2,410 g, 아프가 점수 8/9점으로 출생하였으며 두 번째 신생아는 여자로 출생 체중 1,380 g, 아프가점수 5/8점으로 출생하였다. 두 번째 신생아는 자궁 내 성장지연, 소두증, 소하악증, 사지 관절의 강직을 보였다. 불일치 쌍생아에 대한 원인을 찾기 위한 검사를 진행하였으며 두 번째 신생아의 혈액검사에서 거대세포바이러스 immunoglobulin M이 양성반응을 보였으며 소변과, 혈액, 뇌척수액에서 거대세포바이러스 중합효소연쇄반응이 양성소견을 보였다. 반면에 정상으로 출생한 남아의 거대세포바이러스에 대한 검사는 모두 음성이었다. 감염된 환자의 안과적 검진에서 거대세포바이러스 망막염이 관찰되었고, 후에 실시한 청력검사에서 양측의 감음 난청이 확인되었다. 환자는 ganciclovir 치료를 받았으며 현재 여아는 심한 발달지연이 있으며 침대에서 누워서 생활하는 중이다. 이와 같이 쌍생아에서의 선천 거대세포 바이러스 감염증이 한 명에게만 발생하는 경우의 원인은 현재까지 명확하지 않다. 하지만 이 증례를 통하여 심한 불일치 쌍생아의 경우에 선천 거대세포바이러스 감염의 가능성을 고려해 보아야 한다.

Cytomegalovirus (CMV) infection is one of the most common congenital infections. The first case of discordant congenital CMV infection in twins occurred in Korea. A 31-year-old woman became pregnant with twins (dichorionic-diamniotic). An elective caesarean section was performed at 37 weeks. The first baby was male, weighing 2,410 g with an Apgar score of 8/9. The second baby was female, weighing 1,380 g with an Apgar score of 5/8. She had experienced intrauterine growth retardation, and presented with microcephaly, micrognathia, and joint stiffness. During the work-up for discordant twins, the second baby's serum test was positive for CMV immunoglobulin M. Her urine, blood, and cerebrospinal fluid (CSF) were CMV polymerase chain reaction positive. The first baby's CMV tests were negative. Ophthalmologic exam and audiometry performed on the second baby showed CMV retinitis and bilateral sensorineural hearing loss. She was treated with intravenous ganciclovir. Currently, she is bed-ridden and has significant developmental delay. Although the causes of discordant congenital CMV infection in twins are unclear, this case shows that discordant congenital CMV infection should be considered in twins with significant differences in intrauterine growth or clinical symptoms after birth.

키워드

참고문헌

  1. Wu HY, Huang SC, Huang HC, Hsu TY, Lan KC. Cytomegalovirus infection and fetal death in one monozygotic twin. Taiwan J Obstet Gynecol 2011;50:230-2. https://doi.org/10.1016/j.tjog.2011.01.008
  2. Bodeus M, Hubinont C, Goubau P. Increased risk of cytomegalovirus transmission in utero during late gestation. Obstet Gynecol 1999;93(5 Pt 1):658-60. https://doi.org/10.1016/S0029-7844(98)00538-9
  3. Lazzarotto T, Gabrielli L, Foschini MP, Lanari M, Guerra B, Eusebi V, et al. Congenital cytomegalovirus infection in twin pregnancies: viral load in the amniotic fluid and pregnancy outcome. Pediatrics 2003;112:e153-7. https://doi.org/10.1542/peds.112.2.e153
  4. Simioni C, Sanchez Oliveira Rde C, Moscovi T, D' Agostini Deutsch A, Cordioli E, Santos E. Twin pregnancy and congenital cytomegalovirus: case report and review. J Matern Fetal Neonatal Med 2013;26:622-4. https://doi.org/10.3109/14767058.2012.745503
  5. Ahlfors K, Ivarsson SA, Nilsson H. On the unpredictable development of congenital cytomegalovirus infection. A study in twins. Early Hum Dev 1988;18:125-35. https://doi.org/10.1016/0378-3782(88)90049-7
  6. Egana-Ugrinovic G, Gonce A, Garcia L, Marcos MA, Lopez M, Nadal A, et al. Congenital cytomegalovirus infection among twin pairs. J Matern Fetal Neonatal Med 2016;29:3 439-44.
  7. Kawana K, Nakayama M, Yasugi T, Ishiwata M, Marumo G, Sakai M, et al. Differential clinical manifestations of congenital cytomegalovirus infection between dizygotic twins: a case report. Am J Perinatol 2004;21:383-6. https://doi.org/10.1055/s-2004-835307
  8. Samedi VM, Skappak C, Jantzie L, Trevenen C, Kamaluddeen M, Ekwalanga P, et al. Comparison of presentation, course, and outcome of congenital and acquired cytomegalovirus infection in twins. AJP Rep 2016;6:e1-5.
  9. Yinon Y, Yagel S, Tepperberg-Dikawa M, Feldman B, Schiff E, Lipitz S. Prenatal diagnosis and outcome of congenital cytomegalovirus infection in twin pregnancies. BJOG 2006; 113:295-300. https://doi.org/10.1111/j.1471-0528.2006.00854.x
  10. Griesmaier E, Neubauer V, Blum S, Trawoger R, Keller M, Kiechl-Kohlendorfer U. Neurodevelopmental outcome following congenital cytomegalovirus infection in preterm infants with twin-to-twin transfusion syndrome: a case report. Klin Padiatr 2010;222:312-4. https://doi.org/10.1055/s-0030-1263130
  11. Seguin J, Cho CT. Congenital cytomegalovirus infection in one monozygotic twin. JAMA 1988;260:3277.
  12. Nakajima J, Sunohara D, Kawashima H. Congenital cytomegalovirus infection in monozygotic twins with twin-to-twin transfusion syndrome. Indian Pediatr 2015;52:429-31. https://doi.org/10.1007/s13312-015-0650-x
  13. Nigro G, La Torre R, Anceschi MM, Mazzocco M, Cosmi EV. Hyperimmunoglobulin therapy for a twin fetus with cytomegalovirus infection and growth restriction. Am J Obstet Gynecol 1999;180:1222-6. https://doi.org/10.1016/S0002-9378(99)70620-4
  14. Azam AZ, Vial Y, Fawer CL, Zufferey J, Hohlfeld P. Prenatal diagnosis of congenital cytomegalovirus infection. Obstet Gynecol 2001;97:443-8.
  15. Manoura A, Hatzidaki E, Korakaki E, Margari KM, Galanakis E, Giannakopoulou C. Symptomatic congenital cytomegalovirus infection in one twin after recurrent maternal infection. Pediatr Int 2006;48:88-90. https://doi.org/10.1111/j.1442-200X.2006.02158.x

피인용 문헌

  1. Serratia marcescens as a cause of unfavorable outcome in the twin pregnancy vol.16, pp.1, 2017, https://doi.org/10.1515/med-2021-0205