PMID- 33235734
OWN - NLM
STAT- PubMed-not-MEDLINE
LR  - 20220418
IS  - 2050-0068 (Print)
IS  - 2050-0068 (Electronic)
IS  - 2050-0068 (Linking)
VI  - 9
IP  - 11
DP  - 2020
TI  - Standard treatment-refractory cytomegalovirus encephalitis unmasked by immune 
      reconstitution inflammatory syndrome and successfully treated with virus-specific 
      hyperimmune globulin.
PG  - e1201
LID - 10.1002/cti2.1201 [doi]
LID - e1201
AB  - OBJECTIVES: Cytomegalovirus (CMV)-related encephalitis is a rare but potentially 
      life-threatening complication of CMV infection in immunocompromised patients. The 
      high mortality rate is associated with deficient immune system reconstitution 
      after hematopoietic stem cell transplant (HSCT) and poor bioavailability of 
      antiviral drugs in cerebrospinal fluid (CSF). CMV-related central nervous system 
      (CNS) infection may occur with aspecific symptoms, without evidence of either 
      blood viral load or magnetic resonance imaging (MRI) signs of encephalitis. 
      METHODS: Here, we describe a 10-year-old girl who underwent an allogeneic HSCT 
      and subsequently developed CMV encephalitis. Because of the absence of CMV 
      antigen in the blood, the diagnosis of encephalitis was proposed only after a 
      delay, following the onset of immune reconstitution inflammatory syndrome (IRIS). 
      Two months of combined dual antiviral therapy with ganciclovir and foscarnet 
      proved ineffective against CMV and caused significant bone marrow and renal 
      toxicity. To avoid further toxicity, the girl was given daily treatment with 
      CMV-hyperimmune globulins alone. RESULTS: After three weeks, the CSF viral load 
      dropped significantly and was undetectable within three more weeks. In the 
      meantime, the renal impairment resolved, and there was a complete bone marrow 
      recovery. CONCLUSION: We suggest that this patient succeeded in achieving CMV CSF 
      clearance with high dose of CMV-hyperimmune globulin, given alone, because of the 
      ability of immunoglobulins to penetrate the blood-brain barrier (BBB).
CI  - (c) 2020 The Authors. Clinical & Translational Immunology published by John Wiley & 
      Sons Australia, Ltd on behalf of Australian and New Zealand Society for 
      Immunology, Inc.
FAU - Maximova, Natalia
AU  - Maximova N
AUID- ORCID: 0000-0003-0934-1875
AD  - Institute for Maternal and Child Health - IRCCS Burlo Garofolo Trieste Italy.
FAU - Marcuzzi, Annalisa
AU  - Marcuzzi A
AD  - University of Ferrara Ferrara Italy.
FAU - Del Rizzo, Irene
AU  - Del Rizzo I
AD  - University of Trieste Trieste Italy.
FAU - Zanon, Davide
AU  - Zanon D
AD  - Institute for Maternal and Child Health - IRCCS Burlo Garofolo Trieste Italy.
FAU - Maestro, Alessandra
AU  - Maestro A
AD  - Institute for Maternal and Child Health - IRCCS Burlo Garofolo Trieste Italy.
FAU - Barbi, Egidio
AU  - Barbi E
AD  - Institute for Maternal and Child Health - IRCCS Burlo Garofolo Trieste Italy.
AD  - University of Trieste Trieste Italy.
FAU - Sala, Roberto
AU  - Sala R
AD  - University of Parma Parma Italy.
LA  - eng
PT  - Case Reports
DEP - 20201117
PL  - Australia
TA  - Clin Transl Immunology
JT  - Clinical & translational immunology
JID - 101638268
PMC - PMC7670254
OTO - NOTNLM
OT  - CMV-hyperimmune globulin
OT  - IL-6
OT  - cerebrospinal fluid CMV antibodies
OT  - cytomegalovirus (CMV)-related encephalitis
OT  - immune reconstitution inflammatory syndrome (IRIS)
COIS- The authors have declared that no conflict of interest exists.
EDAT- 2020/11/26 06:00
MHDA- 2020/11/26 06:01
PMCR- 2020/11/17
CRDT- 2020/11/25 05:46
PHST- 2020/05/18 00:00 [received]
PHST- 2020/09/23 00:00 [revised]
PHST- 2020/08/09 00:00 [revised]
PHST- 2020/10/02 00:00 [accepted]
PHST- 2020/11/25 05:46 [entrez]
PHST- 2020/11/26 06:00 [pubmed]
PHST- 2020/11/26 06:01 [medline]
PHST- 2020/11/17 00:00 [pmc-release]
AID - CTI21201 [pii]
AID - 10.1002/cti2.1201 [doi]
PST - epublish
SO  - Clin Transl Immunology. 2020 Nov 17;9(11):e1201. doi: 10.1002/cti2.1201. 
      eCollection 2020.