Case Report Volume 9 Issue 1
1Dermatology Department, Faculty of Medicine and Health Sciences, Sana’a University, Sana’a, Yemen
2Department of Conservative Dentistry, Faculty of Dentistry, Sana’a University, Sana’a, Yemen
Correspondence: Mohammad Ali Alshami, Dermatology Department, Faculty of Medicine and Health Sciences, Sana’a University, Sana’a, Yemen, Tel +967-733760082
Received: January 05, 2025 | Published: January 27, 2025
Citation: Alshami MA, Alshami AM, Alshami HM, et al. Bullous neonatal varicella, acquired postpartal from a household member, treated successfully with i.v acyclovir: a case report. J Dermat Cosmetol. 2025;9(1):5-9. DOI: 10.15406/jdc.2025.09.00282
Neonatal varicella (NV) is extremely rare, with an incidence of 2–6 per 100,000 live births per year, which is often severe and fatal. Bullous NV (BNV) is a rarer variant of varicella. Only 61 cases of NV and 16 cases of BV have been reported; most of them were due to streptococcal infections, also not true bullous varicella; we were not able to find one case of BNV. Herein, we report a case of a 13-day-old male patient presented with severe extensive bullous varicella. On the 9th day postpartum, the mother developed symptoms of a primary varicella zoster virus (VZV) infection while the infant was breastfeeding. Two days later, the infant developed a fever, followed by extensive bullous exanthema involving the entire skin and mucous membranes. He was admitted to the intensive care unit and administered intravenous acyclovir 10 mg/kg/8 h for 7 days and topical fusidic acid/betamethasone cream three times daily. The patient showed prompt improvement and was discharged after 1 week without any sequelae. This is the first case of BNV, treated successfully with only i.v. acyclovir, without VZV IgG or IVIG, which highlights the significance of early intervention with antiviral therapy in neonatal VZV infections.
Keywords: varicella-zoster virus, newborn, neonatal varicella, acyclovir, VZV IgG
Neonatal varicella (NV) is extremely rare, with an incidence of 2–6 per 100,000 live births per year, which is often severe and fatal.1 It is caused by the varicella zoster virus (VZV), also known as human herpesvirus 3, an exclusively human neurotropic virus belonging to the family Alpha-herpesvirinae.1 VZV does not have an animal reservoir and primarily infects T lymphocytes, epithelial cells, and ganglia. Neonates get infected either transplacental from the infected mother or postpartum via the respiratory system, which can be from the infected mother or other infected households.2 Symptoms appear 10–21 days after exposure and typically last 5–7 days. Two factors determine the course and severity of NV, namely the viral load and the maturity of the neonate immune system. If the neonate gets infected transplacentally, the high virus load from the infected mother and the absence of protective maternal antibodies will lead to a severe and eventually fatal course.2 In contrast, if the neonate got infected postnatally through the respiratory system, then the virus load is low, and the relatively immature immune system of the neonate can master the infection, leading to a much milder course. Primary infections usually confer lifelong immunity against the disease. The initial infection, mainly acquired via inhalation or less commonly by direct contact with vesicles, results in varicella (chickenpox), most commonly affecting children. In contrast, herpes zoster results from the reactivation of the latent VZV inside the sensory ganglion cells. Varicella is a childhood disease that occurs primarily during the first decade of life. Varicella infection in the mother during the last week of pregnancy increases the possibility of transplacental infection. To the best of our knowledge, this is the first case of bullous NV (BNV), acquired postnatally, simultaneously with his mother, from a household member.
A 13-day-old infant, weighing 2800 g and delivered vaginally, presented to our clinic with a disseminated confluent vesiculobullous rash covering most of the skin surface, including the scalp, palms, and soles (Figures 1(a-f) & 2(a-d). His mother developed varicella on the 9th day postpartum, which suggested that she acquired varicella during the last five days of pregnancy, considering the typical incubation period of 10–28 days. The infant started to develop typical skin lesions 2 days after his mother, which ruled out the possibility of postnatal infection from his mother. Upon further questioning, the mother confirmed postnatal contact with an infected household member. She explained that it is a tradition for women to congratulate the new mother, and she distinctly remember a woman with VZV infection among the visitors, one day postpartum. Based on the typical history and clinical presentation, a diagnosis of BNV was made, and the baby was administered intravenous acyclovir 10 mg/kg/8 h for 7 days, followed by supportive therapy, to which he responded well and recovered within 1 week. He was followed up at 3, 12, 24, and 36 months after discharge; no complications were found, and the shallow scars disappeared completely by 12 months. The infection was severe in this infant, as he was born to a seronegative mother, who presented with a rash only nine days after delivery, thus having no possibility for the production and transfer of protective IgM and IgG antibodies to the baby. He was treated successfully with 10 mg/kg intravenous acyclovir for 7 days, without VZV-specific hyperimmunoglobulin (VZIG) or IVIG, due to their unavailability. The present case highlights the importance of rapid diagnosis and intravenous acyclovir administration, even in the absence of varicella-zoster immune globulin, to protect the neonate, from severe and eventually fatal complications like, encephalitis, pneumonia, hepatitis, and secondary bacterial infections.
Figure 1a Back of the neonate at presentation, almost monomorphic vesiculobullous exanthem, covering most of the back. Note that some of the bullae are flat due to pressure from laying on the back.
Figure 1b Back of the child 9 days after treatment. Most of the lesions have healed, with only a few hemorrhagic crusts visible.
Figure 1c Face, scalp, and chest of the patient before treatment, covered with yellow crusts, vesicles, and tense bullae mainly on the scalp and upper chest.
Figure 1d Face of the patient 9 days after treatment. Most of the lesions have healed, with only a few hemorrhagic crusts visible.
Figure 1e Chest and abdomen at presentation, almost monomorphic vesiculobullous eruption. Note large bullae over upper chest, presternal, and paraumbilical.
Figure 1f Chest and abdomen 9 days after treatment. Most of the lesions have healed, with only a few hemorrhagic crusts visible.
Figure 2b Right palm 9 days after treatment. Most of the lesions have healed, with only a few hemorrhagic crusts visible.
VZV infection during pregnancy is associated with an increased risk for both the mother and fetus. The outcome of such an infection in infants depends on the timing of the maternal infection.3 Near-term maternal infections are associated with a high risk of neonatal varicella. Serious disseminated infections with visceral involvement may occur in the affected infants.4 NV can be expected if a mother contracts chickenpox during the final 3 weeks of pregnancy. Maternal chickenpox near-term or soon after delivery can cause severe or fatal illness in newborns. Neonatal chickenpox within the first 10–12 days of life could be attributed to the intrauterine transmission of VZV because of the incubation period of varicella. Chickenpox occurring 10–12 days after birth can be attributed to postnatal VZV infection and has a low morbidity rate because maternally derived antibodies protect most neonates.1 The severity of intrauterine-acquired neonatal chickenpox is closely associated with the time of onset of maternal infection because transplacentally transmitted antibodies can reduce the severity of symptoms in newborns. Generalized NV, which can be fatal, is highly likely if mothers develop a varicella rash 4 days before to 2 days after delivery. Some studies have reported an extended risk period for maternal rash onset from 5 days antepartum to 2 days postpartum. Only 61 cases of NV and 16 cases of BV have been reported; we were not able to find one case of BNV (Table 1 & 2).
Pub. year |
1st Author |
Title |
Journal |
Country |
Age 8Y 11Y ? |
Sex M F ? |
Number 1 1 1 |
1963 |
Canby JP |
Bullous Chickenpox (Varicella Bullosa) |
Clin Pediatr (Phila) |
USA |
? 11M 12M |
? M M |
1 1 1 |
1960 |
Saslaw S |
Varicella bullosa |
JAMA |
USA |
17M |
F |
1 |
1963 |
Seigerman H |
Varicella bullosa, a case report |
J Pediatr |
USA |
9M |
M |
1 |
1970 |
Glenn MP |
Varicella bullosa associated with measles vaccine |
Br J Dermatol |
3Y |
F |
1 |
|
1973 |
Melish ME |
Bullous varicella. its association with the staphylococcal scalded skin syndrome |
J Pediatr |
USA |
2YM |
M |
1 |
2008 |
Sulik A |
Bullous varicella in a 5-month-old infant |
Clin Exp Dermatol |
Poland |
5MF |
F |
1 |
2018 |
Suvirya S |
Monomorphic Bullous Hemorrhagic Varicella in a Patient on Methotrexate |
Indian J Paediatr Dermatol |
India |
14YF |
F |
1 |
2019 |
Mansouri S |
Bullous varicella in an immunocompetent infant |
BMJ Case Rep |
Morocco |
6M |
M |
1 |
1994 |
White GM |
Vesicles and bulla in an infant. Bullous varicella (chicken pox complicated by bullous impetigo) |
Arch Dermatol |
USA |
9M |
F |
1 |
2003 |
Sathynarayana BD |
Varicella bullosa |
Indian J Dermatol Venerol Leprol |
India |
3Y |
M |
1 |
2013 |
Sharma CM |
A Classical Case of Neonatal Varicella |
J Clin Neonatol |
India |
5D |
M |
1 |
Total |
16 |
Table 1 Reported cases of bullous varicella. Out of 16 reported cases of bullous varicella, none involved bullous neonatal varicella
Pub. year |
1st Author |
Title |
Journal |
Country |
Age |
Sex |
Nr. |
1878 |
Hubbard TW |
Varicella occurring in an infant twenty-four hours after birth |
Br Med J |
UK |
1D |
M |
1 |
1947 |
Lucchesi PF |
Varicella neonatorum |
Am J Dis Child |
USA |
10D |
F |
1 |
1958 |
Ehrlich RM |
Neonatal varicella. A case report with isolation of the virus |
J Pediatr |
Canada |
12D |
F |
1 |
1967 |
Hyatt HW |
Neonatal varicella. Report of a case in a five-day-old infant and review of the literature |
J Natl Med Assoc |
USA |
5D |
M |
1 |
1968 |
Cutter BG |
Neonatal varicella |
Med J Aust |
Australia |
3D |
M |
1 |
1986 |
Carter PE |
Neonatal varicella infection |
Lancet |
UK |
10 |
||
1986 |
Holland P |
Fatal neonatal varicella infection |
Lancet |
UK |
7D |
F |
1 |
1999 |
Singalavanija S |
Neonatal varicella. a report of 26 cases |
J Med Assoc Thai |
Thailand |
F M |
13 13 |
|
2009 |
Click JW |
Picture of the month. neonatal varicella infection |
Arch Pediatr Adolesc Med |
USA |
12D |
F |
1 |
2010 |
Thakur AK |
Neonatal varicella |
Indian Pediatr |
India |
6D |
M |
1 |
2011 |
Bhardwaj AK |
Neonatal varicella. A case report |
Australas Med J |
India |
5D |
M |
1 |
2011 |
Jackson C |
Rash in a neonate |
J Clin Virol |
UK |
17D |
F |
1 |
2012 |
Kluthe M |
Neonatal vaccine-strain varicella-zoster virus infection 22 days after maternal postpartum vaccination |
Pediatr Infect Dis J |
USA |
25D |
F |
1 |
2012 |
Singh SN |
Varicella infection in a neonate with subsequent staphylococcal scalded skin syndrome and fatal shock |
BMJ Case Rep |
India |
23D |
M |
1 |
2012 |
Hon KL |
Neonatal herpes. what lessons to learn |
Hong Kong Med J |
12DM |
1 |
||
2013 |
Sharma CM |
A Classical Case of Neonatal Varicella |
J Clin Neonatol |
India |
5D |
M |
1 |
2014 |
Marwah P |
Fatal newborn varicella despite varicella zoster immunoglobulin prophylaxis |
Indian J Dermatol Venereol Leprol |
India |
9D |
M |
1 |
2017 |
Choudhary P |
Late neonatal varicella |
Indian J Paediatr Dermatol |
India |
14D |
F |
1 |
2018 |
Machi H |
Neonatal varicella. Probable transmission from a vaccinated mother |
Pediatr Int |
Japan |
10D |
M |
1 |
2020 |
Piyanonpong W |
Effects of Intravenous Immunoglobulin and Acyclovir in Preventing Neonatal Varicella |
Case Rep Inf Dis |
Thailand |
3D |
M |
1 |
2020 |
Reddy R |
Neonatal Varicella |
J Clin Diag Res |
India |
15D |
F |
1 |
2021 |
Lai JW |
Case report. neonatal varicella acquired from maternal zoster |
Front Pediatr |
Australia |
21D |
M |
1 |
2022 |
Earlia N |
Neonatal varicella. a rare case |
Bali MedJ |
Indonesia |
14D |
F |
1 |
2023 |
Frantzis I |
Varicella in the neonatal ICU due to the Varicella vaccine Oka strain |
J Neonatal Perinatal Med |
USA |
7D |
M |
1 |
2024 |
Chap C |
Neonatal varicella, treated with oral acyclovir. A Rare and Challenging Case Report in a Limited-Resource Country like Cambodia |
Clin Case Rep |
Cambodia |
15D |
M |
1 |
2024 |
Li H |
Case Report. Taking action or standing by. managing a preterm neonate at the risk of neonatal varicella by metagenomic next-generation sequencing |
Front Pediatr |
China |
1D |
M |
1 |
2024 |
Alhwayan AA |
Intravenous Immunoglobulin and Intravenous Acyclovir as an Alternative Therapy to Varicella Zoster Immunoglobulin in the Prevention of Serious Complications of Neonatal Varicella |
Cureus |
Jordan |
2D |
M |
1 |
Total |
61 |
Table 2 Reported cases of neonatal varicella. Out of 61 reported cases of neonatal varicella, none involved bullous varicella
After maternal varicella infection during this period, the risk of severe neonatal chickenpox is generally estimated at 20–50%, with a fatal outcome reported in approximately 20% of cases.5 Unfortunately, these infants are exposed to maternal viremia without acquiring protective antibodies. Additionally, the cell-mediated immune response of the neonate is likely insufficient to prevent hematogenous dissemination of VZV after transplacental spread.2 As a result, a fatal outcome is more likely if neonatal disease occurs 5–10 days after delivery. According to available literature, 23% of infants with disseminated and fulminant infections have died.6 NV within the first 4 days after birth is typically milder. Fetuses exposed to VZV between 20 and 6 days before delivery may develop non-fatal neonatal chickenpox due to the presence of maternal antibodies, which reduce the risk of complications. Because of neonatal chickenpox, zoster may occur during infancy, usually with an uncomplicated course. The relatively short viral latency period may be explained by the immature cell-mediated immune response in young children. Maternal zoster during the perinatal period does not generally affect newborn infants because the infants possess specific maternal IgG class antibodies and do not experience the viremic spread of VZV. Treatment options include:(1) IVZIG, (2) IVIG, (3) oral, and (4) intravenous acyclovir, where the best outcomes are achieved by combination of option 1, and 4, followed by option 2, and 4, while the less favorable outcomes are achieved by option 3 alone followed by option 4 alone, which depends on the availability of the drugs.
To conclude, the presented case is the first BNV that is treated successfully with only i.v. acyclovir for 7 days, without VZIG or IVIG.
None.
The authors declare that there are no conflicts of interest.
None.
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