A High Risk of Missing Congenital Cytomegalovirus-Associated Hearing Loss through Newborn Hearing Screening in Japan

Minami, Shujiro; Yamanobe, Yoshiharu; Nakano, Atsuko; Sakamoto, Hirokazu; Masuda, Sawako; Takiguchi, Tetsuya; Katsunuma, Sayaka; Sugiuchi, Tomoko; Morita, Noriko; Kaga, Kimitaka; Matsunaga, Tatsuo

doi:10.3390/jcm10215056

Cited by 5 publications

(5 citation statements)

References 28 publications

(21 reference statements)

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“…In a previous study, 171 of 10 964 newborns failed the NHS test, and only 2 of them were CMV positive, while three additional newborns with congenital CMV infection were tested because of other clinical indications 14 . Other studies showed that approximately 39%–43% of infants with congenital CMV infection were not identified by hearing‐targeted screening, placing them at risk for late‐onset sensorineural hearing loss 15,16 . The detection efficiency of NHS was also low in our study, suggesting that it is insufficient to screen for congenital CMV infection only by NHS tests.…”

Section: Discussioncontrasting

confidence: 57%

“…14 Other studies showed that approximately 39%-43% of infants with congenital CMV infection were not identified by hearingtargeted screening, placing them at risk for late-onset sensorineural hearing loss. 15,16 The detection efficiency of NHS was also low in our study, suggesting that it is insufficient to screen for congenital CMV infection only by NHS tests. the incidence of congenital CMV infection to be 0.31% (66/21272) in Japan.…”

Section: Discussionmentioning

confidence: 58%

See 1 more Smart Citation

Expanded targeted screening for congenital cytomegalovirus infection

Zhang

Egashira

et al. 2023

Congenital Anomalies

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An early diagnosis and intervention for congenital cytomegalovirus infection canreduce long-term disability; however, the introduction of universal neonatal screening has been controversial worldwide. The present study clarified the outcome of a targeted screening protocol for detecting congenital cytomegalovirus infection based on suggestive perinatal conditions. In addition, the positive rate was compared to those from the reported studies and the validity of the targeted screening criteria was discussed. A total of 2121 newborn infants were admitted to our hospital between October 2018 and October 2021. Cytomegalovirus DNA was examined by the isothermal nucleic acid amplification method for urine samples from newborns with any of the following: microcephaly, abnormal ultrasound findings in the brain and visceral organs, repeated failure in neonatal hearing screening, suspicious maternal cytomegalovirus infection during pregnancy, and other abnormal findings suggestive of congenital cytomegalovirus infection. Among 2121 newborns, 102 (4.8%) were subject to the urine cytomegalovirus DNA test based on the abovementioned criteria. Of them, three were cytomegalovirus DNA-positive. According to the protocol, the cytomegalovirus DNA-positive rates were 0.14% among the total enrollment of 2121 newborns and 2.9% (3/102) among the targeted newborns. This protocol may overlook congenital cytomegalovirus infection that is asymptomatic or exhibits inapparent clinical manifestations only at birth; however, it is feasible and helps lead to the diagnosis of congenital cytomegalovirus infection that may otherwise be overlooked.

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Section: Discussioncontrasting

confidence: 57%

Section: Discussionmentioning

confidence: 58%

Expanded targeted screening for congenital cytomegalovirus infection

Zhang

Egashira

et al. 2023

Congenital Anomalies

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“…Another potential specimen type is umbilical‐cord blood, which was used in at least one cCMV screening study 30 . Multiple retrospective research studies conducted in Japan have successfully retrieved and tested dessicated umbilical cord specimens stored by families to identify children suspected of having cCMV disease 66–68 . It is unknown whether it might be acceptable to test stored umbilical cord specimens from deceased offspring for the presence of CMV.…”

Section: Future Study Designsmentioning

confidence: 99%

Estimates of congenital cytomegalovirus‐attributable infant mortality in high‐income countries: A review

Grosse,

Fleming,

Pesch

et al. 2024

Reviews in Medical Virology

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As many as 5%–10% of infants with symptomatic congenital cytomegalovirus (cCMV) disease, or 0.4%–0.8% of all liveborn infants with cCMV infection, die in early infancy in high‐income countries. However, estimates are uncertain due to several potential biases that can result from data limitations and study designs. First, infants with cCMV infections who die prior to diagnosis, which usually occurs at 1–4 weeks after birth, may be excluded from both the count of deaths and the denominator of cCMV births, resulting in left truncation and immortal time biases. These ‘biases’ are features of the data and do not reflect bias on the part of researchers, but understanding the potential existence of threats to validity can help with interpretation of findings. Left truncation of infant deaths occurring prior to diagnosis of cCMV can result in understatement of the burden of infant deaths due to cCMV. Conversely, overestimation of infant deaths associated with symptomatic cCMV may occur in clinical case series owing to greater representation of relatively severely affected infants owing to ascertainment and referral biases. In this review, we summarise the characteristics of 26 studies that reported estimates of cCMV‐associated infant deaths, including potential biases or limitations to which those estimates may have been subject. We discuss study designs whose implementation might generate improved estimates of infant deaths attributable to cCMV. More complete estimates of the overall public health impact of cCMV could inform current and future screening, prevention, and vaccine research.

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“…However, nearly 15% of children with asymptomatic cCMV infection develop SNHL later in life and, therefore, remain undetected according to the UNHS [6]. Minami et al [7] reported a high risk of missing patients with cCMV infection with SNHL in UNHS (13 of 44 patients with cCMV infection passed UNHS bilaterally).…”

Section: Introductionmentioning

confidence: 99%

Congenital Cytomegalovirus Infection and Maternal Primary Cytomegalovirus Infection in Universal Newborn Hearing Screening Referral Patients: A Prospective Cohort Study

Kitamura¹,

Toriyabe²,

Hagimoto-Akasaka³

et al. 2022

Clin. Exp. Obstet. Gynecol.

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Background: There are no detailed reports in the literature on maternal cytomegalovirus antibody screening for universal newborn hearing screening (UNHS) referral patients. We examined maternal cytomegalovirus antibody screening results and estimated the incidence of maternal primary cytomegalovirus infection among UNHS referral patients. Methods: During September 2013-March 2021, fresh urine samples were collected in the first week after birth from 98 neonates with UNHS referral results at 15 obstetrical institutions in Mie, Japan (the first hearing screening). We performed a real-time polymerase chain reaction analysis to detect cytomegalovirus DNA in the samples. Infants with ≥200 copies/mL of cytomegalovirus DNA were diagnosed with congenital cytomegalovirus (cCMV) infection. A second hearing screening was performed, and patients with positive results were sent to the otorhinolaryngologists for further examinations of congenital hearing loss. We calculated incidence rates (%) with 95% confidence intervals (CIs) for cCMV infection among patients with UNHS referral results and maternal primary cytomegalovirus infection among patients who underwent maternal cytomegalovirus antibody screening. Results: Among the 98 neonates with UNHS referral results (the first hearing screening), 5 were diagnosed with cCMV infection (incidence rate: 5.1%; 95% CI: 0.8-9.5). All five patients with cCMV had positive second hearing screening results and were sent to their otorhinolaryngologists. All five were diagnosed with congenital hearing loss, and four were diagnosed with congenital hearing loss secondary to cCMV infection. The remaining patient with cCMV infection was diagnosed with congenital hearing loss unrelated to cCMV infection. Of the 98 patients, 60 underwent maternal cytomegalovirus antibody screening. Among the 60 patients, six had maternal primary cytomegalovirus infection during pregnancy (incidence rate: 10.0%; 95% CI: 2.4-17.6). Of the six patients, four were positive for cytomegalovirus immunoglobulin (CMV Ig) G and IgM antibodies in maternal blood with low CMV IgG antibody avidity results during early pregnancy, while the remaining two had maternal CMV IgG antibody seroconversion during pregnancy. Conclusions: This is the first study to examine the maternal primary cytomegalovirus infection incidence rate in patients with UNHS referral results (the first hearing screening). We identified a 10-fold higher risk in this population (10.0%) than in the general population (0.98%).

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A High Risk of Missing Congenital Cytomegalovirus-Associated Hearing Loss through Newborn Hearing Screening in Japan

Cited by 5 publications

References 28 publications

Expanded targeted screening for congenital cytomegalovirus infection

Expanded targeted screening for congenital cytomegalovirus infection

Estimates of congenital cytomegalovirus‐attributable infant mortality in high‐income countries: A review

Congenital Cytomegalovirus Infection and Maternal Primary Cytomegalovirus Infection in Universal Newborn Hearing Screening Referral Patients: A Prospective Cohort Study

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