Foscarnet therapy for congenital cytomegalovirus liver fibrosis following prenatal ascites

Abstract

We report on an infant with multi-system disease including liver fibrosis, right microphthalmia with cataract, interstitial pneumonitis, and hyperechoic lesions in the basal ganglia and in the periventricular and thalamic regions. Prenatal ascites with hepatomegaly concomitantly with detection of cytomegalovirus (CMV) DNA in the amniotic fluid, following recurrent maternal CMV infection, had been shown. Although CMV culture and DNA detection were negative in the urine, the infant was given foscarnet because CMV infection was demonstrated in the liver by DNA detection and immunohistochemical staining. Favorable clinical outcome and absence of CMV in the liver were subsequently shown. Our case suggests that congenital CMV disease following maternal recurrence may not be associated with disseminated infection but only with intracellular infection. The diagnosis should therefore be based on CMV detection in the involved organs. Moreover, this is the first report on the possible efficacy and safety of foscarnet for therapy of immunocompetent infants with congenital CMV disease.

A practical guide to the management of anophthalmia and microphthalmia

“…There have also been case reports of microphthalmia associated with other intrauterine infections such as varicella, Toxoplasma gondii, herpes simplex virus, and cytomegalovirus. [8][9][10][11] In addition, early assessment of hearing is particularly important to allow prompt intervention in the case of abnormality.…”

Fetal ascites owing to congenital cytomegalovirus: response to ganciclovir

“…25 Nigro et al used foscarnet for postnatal therapy of immunocompetent infants with congenital CMV disease presenting with acsites with beneficial results. 2 Other promising anti-CMV drugs in clinical development are fomivirsen, maribavir, BAY 38-4766, GW275175X and CMX001. 25 Although ganciclovir appears to be effective in ameliorating some symptoms of congenital CMV infection such as ascites and thrombocytopenia, unfortunately, in severely affected infants, it cannot prevent developmental delay and sensorineural deafness.…”

“…However, chronic hepatitis or even lethal cirrhosis can occasionally develop. 2 Congenital CMV infection presenting with fetal ascites is uncommon. [3][4][5] We report an infant who presented with fetal and neonatal ascites along with other features of congenital CMV infection and describe the response to ganciclovir.…”

A dilemma in management of an unwell infant: Short term mortality vs. long term morbidity

“…A single case report documents its use in a neonate with congenital CMV with extensive liver pathology. 13 It is used most commonly to treat GCV resistant CMV or in combination with GCV in patients whose CMV disease is clinically not responding to standard treatment. As FSC does not require activation by viral kinase, resistance to GCV due to a mutation in the UL97 gene will not cause resistance to FSC.…”