TY - JOUR
T1 - Progressive, Long-Term Hearing Loss in Congenital CMV Disease after Ganciclovir Therapy
AU - the Congenital Cytomegalovirus Longitudinal Study Group
AU - Lanzieri, Tatiana M.
AU - Caviness, Alison Chantal
AU - Blum, Peggy
AU - Demmler-Harrison, Gail
AU - Ahmed, Shahzad
AU - Baer, Hanna
AU - Bhatt, Amit R.
AU - Blum, Peggy
AU - Brown, Frank
AU - Catlin, Francis
AU - Caviness, Alison C.
AU - Coats, David K.
AU - Edmonds, Jane C.
AU - Flores, Marily
AU - Franklin, Daniel
AU - Gandaria, Cindy
AU - Greer, Jewel
AU - Griesser, Carol
AU - Hussein, Mohamed A.
AU - Iovino, Isabella
AU - Istas, Allison
AU - Jin, Haoxing
AU - Kelinske, Mary K.
AU - Klingen, Joseph T.
AU - Laurente, Antone
AU - Littman, Thomas
AU - Murphy, Mary
AU - Miller, Jerry
AU - Nelson, Christopher
AU - Noyola, Daniel
AU - Paysse, Evelyn A.
AU - Percy, Alan
AU - Reis, Sara
AU - Reynolds, Ann
AU - Rozelle, Judith
AU - Smith, O'Brien
AU - Steinkuller, Paul
AU - Turcich, Marie
AU - Vinson, Sherry Sellers
AU - Voigt, Robert G.
AU - Walmus, Bethann
AU - Williams, Jill
AU - Williamson, Daniel
AU - Yen, Kimberly G.
AU - Yow, Martha D.
AU - Demmler-Harrison, Gail J.
N1 - Publisher Copyright:
© 2021 Authors.
PY - 2022/1/1
Y1 - 2022/1/1
N2 - Background: Long-term hearing outcomes among children with symptomatic congenital cytomegalovirus (CMV) disease who received 6-week ganciclovir therapy early in life are unknown. Methods: Longitudinal study of 76 children with symptomatic congenital CMV disease, born 1983-2005, who were categorized into three groups: group A treated with ganciclovir; group B untreated who had microcephaly, chorioretinitis, or sensorineural hearing loss (SNHL; ≥25 dB) diagnosed in the first month of life (congenital); and group C untreated who did not meet criteria for group B. Results: Patients in groups A (n = 17), B (n = 27), and C (n = 32) were followed to median age of 13, 11, and 13 years, respectively. In group A, patients received ganciclovir for median of 40 (range, 11-63) days; 7 (41%) had grade 3 or 4 neutropenia. Congenital SNHL was diagnosed in 11 (65%) patients in group A, 15 (56%) in group B, and none in group C. Early-onset SNHL was diagnosed between ages ≥1-12 months in an additional 4 (24%), 6 (22%), and 8 (25%) patients in groups A, B, and C, respectively. By the end of follow-up, 12 (71%), 16 (59%), and 7 (22%) of patients in groups A, B, and C, respectively, had severe (>70 dB) SNHL in the better-hearing ear. Conclusions: In this study, most patients with symptomatic congenital CMV disease and congenital or early-onset SNHL eventually developed hearing loss severe enough to have been potential candidates for cochlear implantation, with or without 6-week ganciclovir therapy. Understanding long-term hearing outcomes of patients treated with 6-month oral valganciclovir (current standard of care) is needed.
AB - Background: Long-term hearing outcomes among children with symptomatic congenital cytomegalovirus (CMV) disease who received 6-week ganciclovir therapy early in life are unknown. Methods: Longitudinal study of 76 children with symptomatic congenital CMV disease, born 1983-2005, who were categorized into three groups: group A treated with ganciclovir; group B untreated who had microcephaly, chorioretinitis, or sensorineural hearing loss (SNHL; ≥25 dB) diagnosed in the first month of life (congenital); and group C untreated who did not meet criteria for group B. Results: Patients in groups A (n = 17), B (n = 27), and C (n = 32) were followed to median age of 13, 11, and 13 years, respectively. In group A, patients received ganciclovir for median of 40 (range, 11-63) days; 7 (41%) had grade 3 or 4 neutropenia. Congenital SNHL was diagnosed in 11 (65%) patients in group A, 15 (56%) in group B, and none in group C. Early-onset SNHL was diagnosed between ages ≥1-12 months in an additional 4 (24%), 6 (22%), and 8 (25%) patients in groups A, B, and C, respectively. By the end of follow-up, 12 (71%), 16 (59%), and 7 (22%) of patients in groups A, B, and C, respectively, had severe (>70 dB) SNHL in the better-hearing ear. Conclusions: In this study, most patients with symptomatic congenital CMV disease and congenital or early-onset SNHL eventually developed hearing loss severe enough to have been potential candidates for cochlear implantation, with or without 6-week ganciclovir therapy. Understanding long-term hearing outcomes of patients treated with 6-month oral valganciclovir (current standard of care) is needed.
KW - congenital cytomegalovirus
KW - ganciclovir
KW - sensorineural hearing loss
UR - http://www.scopus.com/inward/record.url?scp=85123901321&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85123901321&partnerID=8YFLogxK
U2 - 10.1093/jpids/piab095
DO - 10.1093/jpids/piab095
M3 - Article
C2 - 34718680
AN - SCOPUS:85123901321
VL - 11
SP - 16
EP - 23
JO - Journal of the Pediatric Infectious Diseases Society
JF - Journal of the Pediatric Infectious Diseases Society
SN - 2048-7193
IS - 1
ER -