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Basic Characteristics of Mutations
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Mutation Site
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A594V |
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Mutation Site Sentence
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We present our clinical experience with the off-label use of letermovir for SP in a severely immunocompromised 2-year-old toddler with refractory pre-B-cell ALL and bilateral retinitis caused by resistant CMV (A594V UL97 mutation) following a haploidentical TCRalphabeta+ /CD19+ depleted HCT. |
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Mutation Level
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Amino acid level |
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Mutation Type
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Nonsynonymous substitution |
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Gene/Protein/Region
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UL97 |
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Standardized Encoding Gene
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UL97
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Genotype/Subtype
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- |
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Viral Reference
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-
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Functional Impact and Mechanisms
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Disease
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Cytomegalovirus infections
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Immune
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- |
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Target Gene
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-
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Clinical and Epidemiological Correlations
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Clinical Information
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- |
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Treatment
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- |
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Location
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- |
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Literature Information
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PMID
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37466088
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Title
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Pharmacokinetic analysis driven letermovir dosing in pediatric hematopoietic cell transplantation patients with resistant CMV disease
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Author
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Pai VB,Tansmore J,Song E
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Journal
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Pediatric transplantation
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Journal Info
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2024 Feb;28(1):e14580
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Abstract
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BACKGROUND: Reactivation of cytomegalovirus (CMV) in CMV-seropositive patients after haploidentical T-cell receptor alphabeta(+) /CD19(+) depleted hematopoietic cell transplant (HCT) is common. Due to delayed CMV-specific immune reconstitution, patients may require prolonged antiviral therapy, including secondary prophylaxis (SP). We present our clinical experience with the off-label use of letermovir for SP in a severely immunocompromised 2-year-old toddler with refractory pre-B-cell ALL and bilateral retinitis caused by resistant CMV (A594V UL97 mutation) following a haploidentical TCRalphabeta(+) /CD19(+) depleted HCT. METHODS: The patient underwent measurement of two separate sets of letermovir serum concentrations, drawn at pre-dose, 1 and 4 h (and 8 h during the second therapeutic drug monitoring) post-dose. Pharmacokinetic parameters, including AUC(0-24) were calculated, and dose adjustment was performed based on the drug level. RESULTS: While receiving oral letermovir 240 mg once daily without cyclosporine, the observed AUC(0-24) was high (75 815 ng h/mL) with a C(min) of 209 ng/mL. The dose was reduced by 25% to 180 mg once daily. Despite the dose reduction, both AUC(0-24) and C(min) values further increased to 119 095 ng h/L and 959 ng/mL, respectively. The patient continued oral letermovir 180 mg once daily for about 3 months, with adequate viral suppression (CMV viral load in plasma <150 IU/mL) and no recurrent CMV end-organ disease or adverse events. CONCLUSIONS: Given limited options for anti-CMV therapy in young children with resistant CMV, letermovir could be considered as an alternative antiviral for SP. Further studies are warranted to evaluate the pharmacokinetics of letermovir in pediatric allogeneic HCT recipients.
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Sequence Data
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-
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