A 14 year old common variable immunodeficiency patient developed severe protein-losing enteropathy. A chronic enteral infection with human parechovirus type 1 and norovirus was diagnosed. Treatment strategies aimed at virus eradication and providing supportive care were ineffective. The antipicornavirus agent pleconaril did not have any effect on viral replication. Symptoms improved on immunosuppressive therapy, suggesting infection- related immune dysregulation in an immuno-compromised host.
References
1.
European Society for Immunodeficiencies.Diagnostic criteria for PID: Common variable immunodeficiency diagnostic criteria; 2005. (Updated September 2005. Accessed 5 March 2009). Available from: http://www.esid.org/workingparty.php?party=3&sub=2&id=73#Q3.
2.
RudgeP., WebsterA.D., ReveszT.Encephalomyelitis in primary hypogammaglobulinaemia. Brain1996; 119: 1–15.
HallidayE., WinkelsteinJ., WebsterA.D.Enteroviral infections in primary immunodeficiency (PID): a survey of morbidity and mortality. J Infect2003; 46: 1–8.
5.
HyypiäT., HorsnellC., MaaronenM.A distinct picornavirus group identified by sequence analysis. Proc Natl Acad Sci U S A1992; 89: 8847–8851.
6.
Verboon-MaciolekM.A., GroenendaalF., HahnC.D.Human parechovirus causes encephalitis with white matter injury in neonates. Ann Neurol2008; 64: 266–273.
7.
LegayV., ChomelJ.J., LinaB.Specific RT-PCR procedure for the detection of human parechovirus type 1 genome in clinical samples. J Virol Methods2002; 102: 157–160.
8.
Abdel-RahmanS.M., KearnsG.L.Single-dose pharmacokinetics of a pleconaril (VP63843) oral solution and effect of food. Antimicrob Agents Chemother1998; 42: 2706–2709.
9.
HaydenF.G., CoatsT., KimK.Oral pleconaril treatment of picornavirus-associated viral respiratory illness in adults: efficacy and tolerability in Phase II clinical trials. Antivir Ther2002; 7: 53–65.
10.
HaydenF.G., HerringtonD.T., CoatsT.L.Efficacy and safety of oral pleconaril for treatment of colds due to picornaviruses in adults: results of 2 double-blind, randomized, placebo-controlled trials. Clin Infect Dis2003; 36: 1523–1532.
RotbartH.A., WebsterA.D.Treatment of potentially life-threatening enterovirus infections with pleconaril. Clin Infect Dis2001; 32: 228–235.
13.
StarlinR., ReedN., LeemanB.Acute flaccid paralysis syndrome associated with echovirus 19, managed with pleconaril and intravenous immunoglobulin. Clin Infect Dis2001; 33: 730–732.
14.
SegantiL., Di BiaseA.M., MarchettiM.Antiviral activity of lactoferrin towards naked viruses. Biometals2004; 17: 295–299.
15.
Pleconaril. Investigators brochure1999. ViroPharma Inc.Exton, PA, USA.
16.
BellmuntA., MayG., ZellR.Evolution of poliovirus type I during 5.5 years of prolonged enteral replication in an immunodeficient patient. Virology1999; 265: 178–184.
17.
KatzS.L.Polio – new challenges in 2006. J Clin Virol2006; 36: 163–165.
18.
MacLennanC., DunnG., HuissoonA.P.Failure to clear persistent vaccine-derived neurovirulent poliovirus infection in an immunodeficient man. Lancet2004; 363: 1509–1513.