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 Table of Contents  
CASE REPORT
Year : 2023  |  Volume : 12  |  Issue : 1  |  Page : 34-37

An unusual course of pneumonia in a term neonate with suspected multisystem inflammatory syndrome secondary to severe acute respiratory syndrome coronavirus 2 infection


1 Department of Neonatology, Institute of Medical Science and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of Pediatrics, Institute of Medical Science and SUM Hospital, Bhubaneswar, Odisha, India

Date of Submission16-Oct-2022
Date of Acceptance23-Oct-2022
Date of Web Publication03-Jan-2023

Correspondence Address:
Debasish Nanda
D-003, Gymkhana Palm Heights, Near Sum Hospital, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcn.jcn_96_22

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  Abstract 


Neonatal multisystem inflammatory syndrome secondary to severe acute respiratory syndrome coronavirus 2 (SARS-CoV2) infection is a relatively new entity with only a few cases being reported in the literature. We report the case of a 25-day-old term neonate who presented with severe respiratory distress, required respiratory support, and systemic antibiotics at admission. Two-dimensional echocardiography revealed the features of pulmonary arterial hypertension. Computed tomography scan of the chest was suggestive of features of organizing pneumonia. In view of multisystem involvement, raised inflammatory markers, and high anti-SARS-CoV2 antibody, intravenous immunoglobulin was administered, following which the baby improved and weaned off to room air and eventually discharged.

Keywords: Intravenous immunoglobulin, lobar consolidation, multisystem inflammatory syndrome, neonate


How to cite this article:
Ragireddy A, Das RK, Mallick B, Nanda D. An unusual course of pneumonia in a term neonate with suspected multisystem inflammatory syndrome secondary to severe acute respiratory syndrome coronavirus 2 infection. J Clin Neonatol 2023;12:34-7

How to cite this URL:
Ragireddy A, Das RK, Mallick B, Nanda D. An unusual course of pneumonia in a term neonate with suspected multisystem inflammatory syndrome secondary to severe acute respiratory syndrome coronavirus 2 infection. J Clin Neonatol [serial online] 2023 [cited 2023 Jan 27];12:34-7. Available from: https://www.jcnonweb.com/text.asp?2023/12/1/34/366897




  Introduction Top


Multisystem inflammatory syndrome (MIS), following severe acute respiratory syndrome coronavirus 2 (SARS-CoV2) infection, is increasingly reported in pediatric population in the recent times.[1] The syndrome was first described in April 2020, when a number of children with features of severe multisystem involvement similar to Kawasaki disease and toxic shock syndrome were reported from the United Kingdom.[1] Neonatal disease appears to be less common, partly due to the protective antibodies acquired from mother. The risk of vertical transmission of SARS-CoV2 to the neonate is low. In a recent study, the risk of perinatal transmission and horizontal transmission was reported to be 8% and 1.5%, respectively.[2] A virus-induced fetal inflammatory response and subsequent manifestation of MIS have been reported.[3] The syndrome occurs after the virus has been cleared by the immune system, suggesting a postinfectious etiology, and a temporal association with SARS-CoV2 infection has been demonstrated.[4],[5] A few case series have described the clinical profile and outcome of neonatal MIS recently.[6],[7] With a better understanding, new clinical manifestations of neonatal MIS are continuously being reported.


  Case Report Top


A 25-day-old female baby presented to the emergency unit with breathing difficulty and poor feeding for 2 days. The baby was delivered to a 32-year-old primigravida mother at 38 completed weeks through the vaginal route. Birth weight of the baby was 2850 g, and baby did not require any resuscitation after birth. There was no history of any maternal illness during the antenatal period except for a history of mild cough for a day 5 days prior to delivery, which resolved without any medication. The baby was discharged home on the 3rd postnatal day and was on exclusive breast feeding.

On initial evaluation, baby was normothermic (temperature 36.8°C), had tachypnea (respiratory rate of 76/min) and chest retractions, and oxygen saturation of 78% in room air. Baby was admitted to neonatal intensive care unit, and nasal continuous positive airway pressure (CPAP) was administered. Baby was commenced on full feeds, and considering the possibility of late-onset sepsis, intravenous antibiotics were started empirically. Chip-based reverse transcription-polymerase chain reaction for SARS-CoV2 was negative at admission. Initial investigations revealed a normal blood count (hemoglobin 13.6 g/dl, total leukocyte counts of 18,400/cmm, absolute neutrophil counts of 7200/cmm, and platelet count of 3.2 lakh/cu.mm); C-reactive protein was 18.6 mg/dl (reference range: <1 mg/dl). Blood gas obtained after starting CPAP was within the normal range. Cerebrospinal fluid (CSF) study was within the normal range. Blood culture was reported sterile after 48 h of incubation. Chest radiograph revealed perihilar infiltrates in the right lung [Figure 1].
Figure 1: Serial chest radiograph showing changing multifocal consolidation (arrowhead). (a-c) Serial radiographs during the course of disease, (d) radiograph after a course of IVIG. IVIG - Intravenous immunoglobulin

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On the 3rd day of admission, respiratory distress worsened and baby developed features of poor perfusion. Baby was intubated, and mechanical ventilation was started. A normal saline bolus was administered, followed by dopamine infusion (10 μg/kg/min). Two-dimensional echocardiography revealed the features of severe pulmonary arterial hypertension (significant tricuspid regurgitation with pulmonary arterial peak systolic pressure of 53 mm of Hg) with left ventricular ejection fraction of 44%, for which injection sildenafil and milrinone were started. Repeat blood and CSF cultures showed no growth. Baby was extubated on the 6th day to nasal CPAP. Repeated attempts to wean off CPAP support failed. Repeat echocardiogram showed resolution of pulmonary arterial hypertension. Serial chest radiographs revealed a changing pattern of consolidation during the course [Figure 1]. High-resolution computed tomography (CT) chest was suggestive of patchy subpleural consolidation in the apical segment of the right upper lobe and multiple areas of patchy subpleural, peribronchovascular opacities in the left lower lobe with a few ground-glass opacities (GGOs) and radial band of consolidation extending up to the pleura [Figure 2]. The radiologic picture was consistent with a diagnosis organizing pneumonia. Further evaluation showed serum ferritin 579.7 ng/ml (reference range: 50–150 ng/ml), D-dimer 7.08 μg/ml FEU (reference range: <0.5 mcg/ml FEU), and SARS-CoV2 IgG antibodies 49 U/ml (reference range: <0.8 U/ml). Considering a possibility of MIS-N, intravenous immunoglobulin (IVIG) at 2 g/kg over 48 h was initiated. Baby gradually improved and could be weaned to room air within 48 h. It was followed by IV methylprednisolone 2 mg/kg/day for 5 days and tapered in 10 days. Injection enoxaparin 1 mg/kg once daily was administered for 14 days. Antibiotics were discontinued. Baby remained hemodynamically stable and was discharged home on the day 22 of admission.
Figure 2: HRCT axial section showing multiple areas of peribronchovascular and subpleural patchy consolidations, ground-glass opacity, and radial bands of consolidation extending to the pleura in the left lower lobe. HRCT - High-resolution computed tomography

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  Discussion Top


MIS secondary to SARS-CoV2 infection is a well-described entity in adults and pediatric population.[8] The clinical presentation of neonatal MIS may mimic sepsis like illness, which necessitates a high index of suspicion for clinical diagnosis. In the present case, the neonate had a respiratory distress at admission, later developed shock and found to have pulmonary hypertension on echocardiograph, treated with antibiotics suspecting a sepsis like illness. However, a lack of clinical response to antibiotics, persistent radiologic abnormality, lack of alternate explanation for the condition, and prompt response to IVIG and steroid may explain the clinical diagnosis of MIS-N in the present case.

Organizing pneumonias, previously known as bronchiolitis obliterans with organizing pneumonia, are classified as interstitial lung diseases which can occur secondary to various underlying etiologies.[9],[10] The idiopathic form is known as cryptogenic organizing pneumonia. The radiologic appearance of organizing pneumonia is polymorphous. In the classical form, focal subpleural and/or peribronchovascular areas of consolidation are found, which are often bilateral and asymmetrical and predominantly affecting the lower lobes.[9],[10] Associated GGOs with air bronchogram may also be found. Areas of GGOs surrounded by ring of parenchymal consolidation (reverse halo sign or atoll sign) and radial bands of consolidation extending toward pleura, solitary, or multiple nodules are some of the other radiologic findings observed in CT scan in such cases.[9],[10] The radiological picture was consistent with the diagnosis of organizing pneumonia in the present case.

More et al. have reported a series of 20 neonates with MIS secondary to SARS-CoV2 infection and have divided the cases into three categories: likely MIS, possible MIS, and unlikely MIS. Respiratory distress was documented in 80% of cases (four out of five) with a diagnosis of likely MIS and 45% cases (four out of nine) with possible MIS. Nearly 30% of neonates (six out of 20) developed symptoms during the 3rd or 4th week of life.[6] Pulmonary hypertension was observed in two of the neonates. In a recent case series on neonatal MIS, cardiac manifestations were reported in 90% of cases, and 55% of neonates had respiratory distress requiring a respiratory support.[7] Almost 61% of the mothers of these neonates were asymptomatic during pregnancy. None of the mother had fever or any other symptoms attributed to SARS-CoV2 infection at the time of delivery, and none of them were tested for SARS-CoV2 prior to delivery.[7] This highlights the possibility of perinatal transmission to neonates from asymptomatic mothers. The strategy of COVID-19 testing in pregnant women during labor has recently been revised. According to the recent ICMR guideline, pregnant women in or near labor, who are hospitalized for delivery should not be tested unless warranted or develop any symptom.[11] The time of presentation of neonates with MIS varies widely. Majority of cases, as described, have reported the onset within the 1st or 2nd week. However, later presentation has also been reported.[6],[7] Onset of clinical symptoms may depend on the time of acquisition of SARS-CoV2 infection. In our case, the time of acquisition of SARS-CoV2 infection remains unclear. The presence of anti-SARS-CoV2 IgG antibodies may suggest the possibility of intrauterine transmission, but the same could not be confirmed. Mother was not tested for SARS-CoV2 infection prior to delivery. Other family members did not have any flu-like illness and not being tested for SARS-CoV2 infection during the period. More than 90% of adults or pregnant women with SARS-CoV2 infection remain asymptomatic. The neonate might have acquired the infection in utero or in the postnatal period, which subsequently manifested as MIS.


  Conclusion Top


Organizing pneumonia which has been described as a common radiologic feature in adults with MIS can also be seen in neonates. Multisystem involvement, lack of adequate response to initial treatment, and a high index of suspicion for the possibility of MIS can help in early diagnosis and prompt institution of treatment in such cases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given his consent for images and other clinical information to be reported in the journal. The guardian understands that names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Abrams JY, Godfred-Cato SE, Oster ME, Chow EJ, Koumans EH, Bryant B, et al. Multisystem inflammatory syndrome in children associated with severe acute respiratory syndrome coronavirus 2: A systematic review. J Pediatr 2020;226:45-54.e1.  Back to cited text no. 1
    
2.
More K, Chawla D, Murki S, Tandur B, Deorari AK, Kumar P, et al. Outcomes of neonates born to mothers with coronavirus disease 2019 (COVID-19) – National neonatology forum (NNF) India COVID-19 registry. Indian Pediatr 2021;58:525-31.  Back to cited text no. 2
    
3.
McCarty KL, Tucker M, Lee G, Pandey V. Fetal inflammatory response syndrome associated with maternal SARS-CoV-2 infection. Pediatrics 2021;147:e2020010132.  Back to cited text no. 3
    
4.
Alcock J, Masters A. Cytokine storms, evolution and COVID-19. Evol Med Public Health 2021;9:83-92.  Back to cited text no. 4
    
5.
Nakra NA, Blumberg DA, Herrera-Guerra A, Lakshminrusimha S. Multi-system inflammatory syndrome in children (MIS-C) following SARS-CoV-2 infection: Review of clinical presentation, hypothetical pathogenesis, and proposed management. Children (Basel) 2020;7:E69.  Back to cited text no. 5
    
6.
More K, Aiyer S, Goti A, Parikh M, Sheikh S, Patel G, et al. Multisystem inflammatory syndrome in neonates (MIS-N) associated with SARS-CoV2 infection: A case series. Eur J Pediatr 2022;181:1883-98.  Back to cited text no. 6
    
7.
Pawar R, Gavade V, Patil N, Mali V, Girwalkar A, Tarkasband V, et al. Neonatal multisystem inflammatory syndrome (MIS-N) associated with prenatal maternal SARS-CoV-2: A Case Series. Children (Basel) 2021;8:572.  Back to cited text no. 7
    
8.
Radia T, Williams N, Agrawal P, Harman K, Weale J, Cook J, et al. Multi-system inflammatory syndrome in children & adolescents (MIS-C): A systematic review of clinical features and presentation. Paediatr Respir Rev 2021;38:51-7.  Back to cited text no. 8
    
9.
Roberton BJ, Hansell DM. Organizing pneumonia: A kaleidoscope of concepts and morphologies. Eur Radiol 2011;21:2244-54.  Back to cited text no. 9
    
10.
Baque-Juston M, Pellegrin A, Leroy S, Marquette CH, Padovani B. Organizing pneumonia: What is it? A conceptual approach and pictorial review. Diagn Interv Imaging 2014;95:771-7.  Back to cited text no. 10
    
11.
Available from: https://www.icmr.gov.in/pdf/covid/strategy/Advisory_COVID_Testing_10012022.pdf. [Last accessed on 2022 Jul 22].  Back to cited text no. 11
    


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  [Figure 1], [Figure 2]



 

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