Congenital cytomegalovirus infection in monozygotic twins with twin-to-twin transfusion syndrome

Nakajima, Junya; Sunohara, Daisuke; Kawashima, Hisashi

doi:10.1007/s13312-015-0650-x

Cited by 5 publications

(4 citation statements)

References 9 publications

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“…We cannot evaluate chorionicity in this data set; however, the discordance of infection in monochorionic twins leads one to ponder about the epigenetics at play. Twin-twin transfusion syndrome is also noted in two other concordant cases of MZ twins labeled (Griesmaier et al, 2010;Nakajima et al, 2015). In the Nakajima study, the smaller/donor twin was more profoundly affected, while in the Griesmaier study, both twins are affected.…”

Section: Discussionmentioning

confidence: 93%

Vertical Transmission and Discordance of Cytomegalovirus in Twin Pregnancies

Hutton

Rowan

2021

Front. Cell. Infect. Microbiol.

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ObjectiveThe objectives are to estimate the vertical transmission rate in twins relative to singleton pregnancies, to evaluate whether discordance within twin pairs is rare, and to characterize concordance within monozygotic and dizygotic twin pairs in relation to hereditability.MethodsWe first sought to estimate the vertical transmission rate of congenital CMV infection in twins by gathering cohort-based studies of congenital CMV in which vertical transmission in both singleton and twin pregnancies was reported. This also allowed us to compare singleton and twin infection rates. From the above studies and other large cohorts of congenitally infected infants, the percentage of discordantly infected twin pairs determined whether this is a rare phenomenon. Theorizing discordance is not rare, we then analyzed data from cases with twin outcomes for congenital CMV infection, according to whether the twins were monozygotic or dizygotic, and calculated their corresponding concordance rates to estimate the broad-sense heritability. Lastly, we described other factors that might affect vertical transmission.ResultsFrom five articles following at-risk pregnancies, the rate of vertical transmission in twin pregnancies is 58.7% (95% CI 43.3-72.3%) whereas in singleton pregnancies it is 31.4% (95% CI: 29.0-34.0%) p = 0.0002. Of ten studies of larger cohorts of infants with congenital CMV infection, 21 of 42 twin pairs with at least one twin infected were discordant for congenital CMV (50.0%, 95% CI: 34.4–65.6%) indicating discordance of congenital CMV infection in twin pairs is not rare. Of 28 studies covering 37 twin pairs where at least one twin had congenital CMV, and zygosity was known, eleven of thirteen monozygotic twin pairs (84.6%; 95% CI: 53.7-97.3%) were concordant for CMV infection, and nine of twenty-four dizygotic twin pairs (37.5%; 95% CI: 19.6-59.2%) were concordant for infection giving an estimated hereditability of 94.2%. Within these 37 twin pairs, factors such as primary or recurrent maternal infection, prematurity, growth discordance, and sex are described; however, in many of these cases these factors are unknown.ConclusionThe rate of vertical transmission of congenital CMV is higher for twins than singletons. Discordance of congenital CMV in twins is not rare and suggests a possible genetic susceptibility to congenital CMV.

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Section: Discussionmentioning

confidence: 93%

Vertical Transmission and Discordance of Cytomegalovirus in Twin Pregnancies

Hutton

Rowan

2021

Front. Cell. Infect. Microbiol.

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“…1). Study characteristics appear in Table 2 (11–41). The systematic review included 18 single-case reports, 6 case series, 5 cohort/cross-sectional studies, and 1 pre-post intervention study.…”

Section: Resultsmentioning

confidence: 99%

“…Risk of hearing loss is correlated with CMV viral load (42). Between twins, placental histopathology and amniotic fluid assays have indicated that symptom severity may be associated with viral load in the placenta (22,26), although differential outcomes have been documented in identical twins who shared a monochorionic placenta (25,31). Our review found that each cCMV-positive patient’s odds of hearing loss are tripled if their twin is also infected, which may relate to viral load.…”

Section: Discussionmentioning

confidence: 99%

Congenital Cytomegalovirus Infection and Hearing Outcomes in Twins: A Systematic Review of Reported Cases

Cordray¹,

Liu-Lam

Tey³

et al. 2022

Otol Neurotol

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ObjectiveCongenital cytomegalovirus infection is the leading nonhereditary cause of pediatric sensorineural hearing loss. This systematic review evaluated infection concordance and comparative hearing abilities in twins/multiple births to model infection patterns.Databases ReviewedPubMed, Embase, Web of Science, and Google Scholar.MethodsStudies that reported hearing outcomes of congenital cytomegalovirus infection in at least one multiple birth were eligible. Concordant infections (both twins) and discordant infections (single twin) were included. Multiple reviewers performed data extraction and quality assessment. Analyses involved relative risk of infection concordance by zygosity and chorionicity and odds of hearing loss by infection concordance. Hearing outcomes were compared between siblings.ResultsOf 247 studies screened, 31 were included (74.2% high quality). The review captured 40 eligible multiple births. Among infected patients, 42.9% (95% confidence interval, 31.2–55.2%) demonstrated hearing loss. All uninfected twins had normal hearing. Most infections were concordant, and infected patients experienced 4.11 (1.18–14.36) times greater odds of hearing loss if their twin was also infected (p = .02). Yet siblings’ hearing outcomes diverged in over 40% of concordant cases. If either twin is infected, infection risk in the second twin is 3.25 (1.83–5.79) times greater in monozygotic than dizygotic twins and 2.50 (1.61–3.88) times greater in monochorionic than dichorionic twins (both p < .001). We describe a case from our practice.ConclusionCongenital cytomegalovirus infection patterns and hearing outcomes can vary widely even within a shared fetal and postnatal environment. Suspected infection in a twin indicates that both should receive testing and continued monitoring for late-onset sequelae.

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“…CMV infection in both monochorionic and dichorionic twins can occur in only one fetus or both twins, clinical symptoms and prognosis were quite different ( Table 3 ). In the 21 reported cases of congenital CMV infected twins [2] , [3] , [4] , [6] , [7] , [8] , [9] , [10] , [11] , [12] , [13] , [14] , 10 cases (47.6%) were single infant infection, of which 3 cases (30%) were stillborn or died after birth, 2 cases (20%) were terminated. 11 cases (52.3%) were infected with both twins, 1 case of them both twins were stillbirths, one of twins in a case died after birth, twins in a case were terminated.…”

Section: Discussionmentioning

confidence: 99%

Differential congenital cytomegalovirus infection in dichorionic diamniotic twins—A case report and literature review

Feng

Yang

et al. 2022

IDCases

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Background Cases of differential congenital cytomegalovirus (CMV) infection in twins are rarely reported. The chance of congenital infection and the clinical outcome of monochorionic diamniotic or dichorionic diamniotic twins are highly uncertain. Cases presentation We reported a case of differential congenital CMV infection in dichorionic diamniotic twins. Despite being exposed to the same maternal environment and similar genetic background, twins reacted differently to maternal infection and presented with non-concordant infection status. The potential mechanism of discordant infection from aspects of type of chorion and placenta and immune status has been discussed through literature review. Conclusion CMV infection can present as differential congenital infection in twin pregnancy, with various clinical symptoms. Fetal cellular immune function may be involved in the pathogenesis.

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Congenital cytomegalovirus infection in monozygotic twins with twin-to-twin transfusion syndrome

Abstract: Intrauterine hemodynamics may be important in pathophysiology of congenital cytomegalovirus infection.

Cited by 5 publications

References 9 publications

Vertical Transmission and Discordance of Cytomegalovirus in Twin Pregnancies

Vertical Transmission and Discordance of Cytomegalovirus in Twin Pregnancies

Congenital Cytomegalovirus Infection and Hearing Outcomes in Twins: A Systematic Review of Reported Cases

Differential congenital cytomegalovirus infection in dichorionic diamniotic twins—A case report and literature review

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