Discordant Congenital Cytomegalovirus Infection in Twins

Yi-Seul Kim; Ji-Man Kang; Ji-Hoon Lee; Yoon Sil Chang; Won Soon Park; Yae-Jean Kim

doi:10.14776/piv.2017.24.1.65

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Kim, Kang, Lee, Chang, Park, and Kim: Discordant Congenital Cytomegalovirus Infection in Twins

Case Report

Pediatr Infect Vaccine 2017;24(1):65-70.

Published online: 21 January 2017

DOI: https://doi.org/10.14776/piv.2017.24.1.65

Discordant Congenital Cytomegalovirus Infection in Twins

Yi-Seul Kim, Ji-Man Kang, Ji-Hoon Lee, Yoon Sil Chang, Won Soon Park, Yae-Jean Kim

Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, the Republic of Korea

Correspondence: Yae—Jean Kim Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, the Republic of Korea Tel: +82—2—3410—3539, Fax: +82—2—3410—0043 E—mail: yaejeankim@skku.edu

Received 15 September 201613 October 2016 Accepted 13 October 2016

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

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.

Keywords: Congenital, Infection, Cytomegalovirus, Twins

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Fig. 1.

The results of urine cytomegalovirus (CMV) polymerase chain reaction (PCR).

Fig. 2.

(A, B) Brain ultrasonography performed 2 days after birth shows ventriculomegaly with increased periventricular echogenicity. (C, D) Brain magnetic resonance imaging at postnatal age 4 months shows cortical malformation and extensive white matter volume loss with circumventricular calcifications (arrow) and cerebellar hypoplasia.

Table 1.

Case Reports of Congenital CMV Infection in Twin Pregnancies

Study	Case	Chorionicitv	Type of maternal infection	Conqenital CMV infection	Clinical outcome
Seguin etal. (1988)¹¹⁾	1	MC	Unknown	Uninfected/infected	Asymptomatic/symptomatic
Nigro etal. (1999)¹³⁾	1	DC	Primary	Infected/nfected	Asymptomatic/asymptomatic
Azam etal.(2001)¹⁴⁾	1	UL	Unknown	Infected/unifected	Asmptomatic/symptomatic
lazzaroto et al (2003)³⁾	3	DC	Primary	Infected/uninfected Infected/infected/infected Infected/infected	Symptomatic (fetal death)/asymptomatic Symptomatic/symptomatic/symptomatic Asymptomatic/symptomatic
Kawana et al. (2004)⁷⁾	1	DC	Primary	Infected/infected	Symptomatic (severe)/symptomatic
Yinon et al.(2006)⁹	6	DC	Primary Recurrent	Uinfected/infected Infected/infected Infected/infected Infected/infected Infected/infected Infected/infected Infected/infected	Asymptomatic/symptomatic (TOP) Asymptomatic/asymptomatic Asymptomatic/asymptomatic Asymptomatic/asymptomatic (death) Symptomatic (OIFD)/symptomatic (IUFD) TOP∗/TOP∗
Manoura et al. (2006)¹⁵⁾	1	DC	Recurrent	Infected/uninfected	Symptomatic/asymptomatic
Griesmaier et al. (2010)¹⁰⁾	1	MC	Primary	Infected/infected	Symptomatic/symptomatic
Wu et al. (2011)¹⁾	1	MC	Unknown	Infected/infected	Asymptomatic/symptomatic (fetal death)
Simioni et al. (2013)⁴⁾	1	DC	Primary	Infected/Uninfected	Asymptomatic/asymptomatic
Nakajima et al. (2015)¹²⁾	1	MC	Unknown	Infected/infected	Symptomatic (severe)/symptomatic
Samedi et al. (2016)⁸⁾	1	DC	Primary	Infected/infected	Symotomatic/asymotomatic
Euana–Uarinovic et al. (2016)⁶⁾	6	MC DC	Unknown	Infected/infected Uninfected/infected Uninfected/infected Uninfected/infected Uninfected/infected Uninfected/infected	Symotomatic/symoptmatic Asymotomatic/symoptmatic TOP/asymptomatic TOP/asymptomatic TOP (due to NTD)/asymptomatic IUFD/asymptomatic Asymptomatic/asymptomatic
Present study	1	DC	Unknown	Infected/uninfected	Symptomatic/asymptomatic

^∗ Amniotic fluid results were positive and TOP was performed due to parents' request. Abbreviations: CMV, cvtomeqalovirus; MC, monochorinonic; DC, dichorionic; TOP, termination of preqnancv; IUFD, intrauterine fetal death; NTD, neural tube defect.

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