|Year : 2019 | Volume
| Issue : 3 | Page : 272-274
Unusual association of the neonatal herpes simplex virus hepatitis with congenital hydrocephalus
Aditya Pratap Singh, Ramesh Tanger, Arun Kumar Gupta, Dileep Garg
Department of Pediatric Surgery, SMS Medical College, Jaipur, Rajasthan, India
|Date of Submission||26-Apr-2018|
|Date of Acceptance||24-Jul-2018|
|Date of Web Publication||15-May-2019|
Aditya Pratap Singh
Near The Mali Hostel, Main Bali Road, Falna, Pali, Rajasthan
Source of Support: None, Conflict of Interest: None
We report a 2-month-old male baby who presented with yellow discoloration of skin, high-colored urine, and intermittent clay-colored stools since 15 days of life. Head size was apparently looking large with dilated veins over the scalp. The baby was diagnosed and treated as neonatal herpes simplex virus hepatitis that led to cholestatic jaundice and cerebral aqueductal stenosis with hydrocephalus. It is a very rare association and less reported earlier in the literature.
Keywords: Cerebral aqueductal stenosis, cholestatic jaundice, hydrocephalus, neonatal hepatitis
|How to cite this article:|
Singh AP, Tanger R, Gupta AK, Garg D. Unusual association of the neonatal herpes simplex virus hepatitis with congenital hydrocephalus. Med J DY Patil Vidyapeeth 2019;12:272-4
|How to cite this URL:|
Singh AP, Tanger R, Gupta AK, Garg D. Unusual association of the neonatal herpes simplex virus hepatitis with congenital hydrocephalus. Med J DY Patil Vidyapeeth [serial online] 2019 [cited 2019 May 25];12:272-4. Available from: http://www.mjdrdypv.org/text.asp?2019/12/3/272/258212
| Introduction|| |
Neonatal jaundice has been reported many times in medical literature and has varied etiology ranging from infections to congenital malformations. Neonatal herpes simplex virus (HSV) infection is rare, but associated with severe morbidity and mortality rates. The infection most commonly occurs in the intrapartum period, but it may also occur in the intrauterine or postpartum period.
| Case Report|| |
A 2-month-old male infant was brought by his parents at the age of 1 month because of yellow discoloration of the skin and sclera. The infant was born at term with birth weight of 2.5 kg. there was no family history of neonatal jaundice. baby was the product of nonconsanguious marriage. the pregnancy was uneventful. The mother did not have a history or signs of HSV infection. The infant was fully breastfed and was thriving well, he was deeply jaundiced, his urine had deep yellow color, and his stool was of intermittent clay color. The baby was not dysmorphic. Abdominal examination revealed hepatomegaly of 2 cm with liver span of 6 cm. Head size was large with dilated veins. Anterior fontanel was wide open, pulsatile with marked sutural separation.
His full blood count was normal. Liver function tests showed total serum bilirubin 16 mg/dl, direct bilirubin 10 mg/dl, aspartate aminotransferase (AST) 395 U/L, alanine aminotransferase (ALT) 298 U/L, total protein 5.7 g/dl, albumin 3.8 g/dl, alkaline phosphatase 970 U/L, LDH 1018 U/L, prothrombin time with international normalized ratio 1.55, AFP 826 IU/ml, and serum GGT 1173.9 u/l; stool is positive for fecal fat; and renal function tests, activated partial thromboplastin time, and serum electrolytes were normal. Urine for reducing substances was negative. Hepatitis B and C screening was negative; TORCH screening showed negative cytomegalovirus [CMV] IgM and rubella IgM and positive CMV IgG, HSV IgM, IgG, and rubella IgG. Abdominal ultrasound (USG) showed enlarged liver with normal texture and echogenicity, no focal lesion, contracted gallbladder, and mild intrahepatic biliary duct dilatation without ascites. USG brain showed bilateral ventricular dilatation with Ventriculo-hemispheric ratio (VHR)=0.7, suggestive of hydrocephalus (? aqueductal stenosis) [Figure 1]a. Noncontrast computed tomography head showed gross hydrocephalus due to aqueductal stenosis. As the above tests were inconclusive, a hydroxy iminodiacetic acid (HIDA) scan was obtained which showed intense homogeneous tracer uptake in the liver with no excretion into the duodenum and bowel in the dynamic images as well as the 2 h and 24 h images, which was suggestive of extrahepatic biliary atresia [Figure 1]b. Magnetic resonance cholangiopancreatography showed nonvisualization of the gallbladder and liver was normal with intrahepatic biliary radicals not dilated (? biliary atresia) [Figure 2]a. A percutaneous liver biopsy showed neonatal hepatitis. The baby was diagnosed as cholestatic jaundice due to neonatal hepatitis with hydrocephalus due to aqueductal stenosis.
|Figure 1: (a) Noncontrast computerized tomography head image (arrow shows normal size forth ventricle). (b) Hydroxy iminodiacetic acid scan image (arrow shows liver uptake but not any excretion)|
Click here to view
|Figure 2: (a) Magnetic resonance cholangiopancreatography image (arrow shows no intrahepatic biliary radical dilatation with no visualization of gallbladder). (b) Postoperative image|
Click here to view
We put ventriculoperitoneal shunt (medium pressure) for the hydrocephalus. Postoperatively, fontanels become shrunken. The baby was commenced on fat-soluble vitamins (A, D, E, and K), ursodeoxycholic acid, zinc sulfate, multivitamin syrup, phenobarbitone, Vitamin E, and Vitamin K. The baby started to show gradual improvement in his liver profile, remained stable, and maintained normal synthetic liver function tests. He was discharged home after 4 weeks with liver function that showed total serum bilirubin of 7 mg/dl, direct bilirubin 5 mg/dl, AST 197 U/L, ALT 116 U/L, alkaline phosphatase 1037 U/L, total protein 5 g/dl, and albumin 3 g/dl. The patient is under follow-up regularly with a follow-up of 4 months. The baby had follow-up without any shunt complications. The bilirubin levels came down to 2 mg/dl [Figure 2]b.
| Discussion|| |
Neonatal HSV infection, although relatively rare, has a high mortality and morbidity. It is estimated that 1 neonate in 3200 live births has HSV infection. Neonatal herpes can be localized to skin, eye, mouth or central nervous system or can cause disseminated infection involving multiple organs. Disseminated infection is the most severe form of neonatal herpes, with a mortality rate of 85% for untreated neonates. Intravenous acyclovir given in a high dose and early in the course of the disease significantly improves prognosis.
Viral hepatitis may be due to CMV, rubella virus, HSV, human herpesvirus 6, varicella, coxsackievirus, echovirus, reovirus 3, parvovirus B19, HIV, enteroviruses, paramyxovirus, and hepatitis A, B, or C.
The absence of neurologic signs and symptoms and normal neuroimaging with the excellent developmental outcome also suggest absence of encephalitis. We treated the patient with only choleretic agents and multivitamins. We had a good response with decreased levels of the serum bilirubin and liver enzymes. Similar results were seen by Sakaria et al.
| Conclusion|| |
Our patient had neonatal HSV hepatitis, associated with CNS anomaly in the form of obstructive hydrocephalus. It needs further research and study to have a better management and cause of such association.
We would like to thank Dr. Neelam Dogra, Anaesthesiologist, Senior professor, SMS Medical College Jaipur, Rajasthan, India.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Shorter RG, Baggenstoss AH, Logan GB. Neonatal hepatitis. AMA J Dis Child 1959;98:359-69.
Abuhasna SD, Shihab ZM, Al Niyadi SM, Tatari HM, Al Jundi AH, Atwa KH. Neonatal herpes simplex fulminant hepatitis successfully treated with acyclovir. J Clin Neonatol 2012;1:87-90.
] [Full text]
White JC, Magee SR. Neonatal herpes infection: Case report and discussion. J Am Board Fam Med 2011;24:758-62.
Kimberlin D. Herpes simplex virus, meningitis and encephalitis in neonates. Herpes 2004;11 Suppl 2:65A-76A.
Kimberlin DW, Lin CY, Jacobs RF, Powell DA, Corey L, Gruber WC. Safety and efficacy of high-dose intravenous acyclovir in the management of neonatal herpes simplex virus infections. Pediatrics 2001;108:230-8.
Hicks J, Barrish J, Zhu SH. Neonatal syncytial giant cell hepatitis with paramyxoviral-like inclusions. Ultrastruct Pathol 2001;25:65-71.
Sakaria R, Shah I, Bhatnagar S. Neonatal hepatitis with herpes simplex. Med J DY Patil Univ 2016;9:397-9. [Full text]
[Figure 1], [Figure 2]