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Radiol Case Rep
Radiol Case Rep
Radiology Case Reports
1930-0433
Elsevier

S1930-0433(24)00746-5
10.1016/j.radcr.2024.07.160
Case Report
Multisystem inflammatory syndrome in neonates associated with pneumothorax: Case report
Naous Amal MD a.naous@bau.edu.lb
ab⁎
Ghannoum Walaa MD a
Abbas Aya MBBCh b
Darwish Hussein MBBCh b
Naja Zeina MD a
Rajab Mariam MD a
a Faculty of Medicine and Surgery, Beirut Arab University, Beirut, Lebanon
b Department of Pediatrics, Faculty of Medicine at Beirut Arab University, Makassed General Hospital, Beirut, Lebanon
⁎ Corresponding author. a.naous@bau.edu.lb
22 8 2024
11 2024
22 8 2024
19 11 51245127
23 1 2024
26 7 2024
28 7 2024
© 2024 The Authors. Published by Elsevier Inc. on behalf of University of Washington.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
The coronavirus disease 2019 (COVID-19) pandemic that was spread worldwide since 2019 and showed a highly contagious character affecting the lifestyle of people worldwide causing symptoms that are not limited to the respiratory system only but had multi-systemic effects that may progress to severe complications that roughly affect people's health. A newly recognized SARS-CoV-2-associated syndrome called pediatric multisystem inflammatory syndrome has been described worldwide. Initially, it was reported as hyper-inflammatory shock and “Kawasaki-like” symptoms with fever and conjunctivitis, a similar syndrome is also reported in neonates and called Multisystem Inflammatory Syndrome of neonates (MIS-N). In this paper, we presented a case of a newborn baby girl born by caesarian section affected by multisystem inflammatory syndrome of neonates presenting with respiratory distress on her third day of life, then she developed bilateral pneumothorax and pneumomediastinum respectively that required intubation, which highlights that the recognition of pneumothorax and pneumomediastinum as potential presentations of immunoglobulin G (IgG) positive MIS-N in newborns remains crucial.

Keywords

The coronavirus disease 2019
Multisystem inflammatory syndrome of children
Multisystem inflammatory syndrome of neonates
Pneumothorax
Kawasaki-like disease, Neonate
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pmcIntroduction

The coronavirus disease 2019 (COVID-19) is a pandemic that struck in 2019 and had systemic complications that affected all of the age groups and had a severe impact on people's life style and a remarkable impact to note on high-risk group pregnant women. Multisystem inflammatory syndrome in neonates (MIS-N) is a rare postinfectious hyperinflammatory disorder associated with SARS-CoV-2 in neonates, and is characterized by overwhelming systemic inflammation, fever, hypotension, and cardiac dysfunction. Table 1 demonstrates the proposed inclusion criteria for the neonatal multisystem inflammatory syndrome (MIS-N) secondary to maternal SARS CoV-2 exposure or infection [1].Table 1 Proposed inclusion criteria for neonatal multisystem inflammatory syndrome (MIS-N) secondary to maternal SARS CoV-2 exposure or infection.

Table 1(1) A neonate aged <28 days at the time of presentation.

(2) Laboratory or epidemiologic evidence of SARS-CoV-2 infection in the mother.• Positive SARS-CoV-2 testing by RT-PCR, serology (IgG or IgM), or antigen during pregnancy.

• Symptoms consistent with SARS CoV-2 infection during pregnancy.

• COVID-19 exposure with confirmed SARS CoV-2 infection during pregnancy.

• Serological evidence (positive IgG specific to SARS CoV-2 but not IgM) in the neonate.

(3) Clinical criteria:• Severe illness necessitating hospitalization AND

• Two or more organ systems affected [i.e., cardiac, renal, respiratory, hematologic, gastrointestinal, dermatologic, neurological, temperature instability (fever or hypothermia)] OR

• Cardiac AV conduction abnormalities OR coronary dilation or aneurysms (without involvement of a second organ system).

(4) Laboratory evidence of inflammation• One or more of the following: an elevated CRP, ESR, fibrinogen, procalcitonin, D-dimer, ferritin, LDH, or IL-6; elevated neutrophils or reduced lymphocytes; low albumin.

(5) No alternative diagnosis (such as birth asphyxia–cord pH ≤ 7.0 and Apgar score ≤ 3 at 5 min; viral or bacterial sepsis–confirmed blood culture; maternal lupus resulting in neonatal AV conduction abnormalities; presence of these findings indicating an alternate diagnosis excludes MIS-N).

	
CRP, C-reactive protein; ESR, erythrocyte sedimentation rate; LDH, lactate dehydrogenase; IL-6, interleukin 6.

This case report aimed to describe a case of MIS-N presented a month after the WHO declaration of the end of COVID-19 as a global pandemic and with unusually reported clinical picture and complications.

Case report

A case of a 3-day-old female patient born at 38th week of gestation by scheduled secondary Caesarean section to a 24-year-old G2P2A0 mother. The baby was born on June 10, 2023, at 11:00 am, 1 month after the WHO declaration of the end of COVID pandemic.

Maternal prenatal care

During pregnancy, the mother was followed regularly every month. Screening ultrasound was done at the fifth month of gestation showed that the fetus had ectopic right kidney which was present in the pelvis, and then a morphological ultrasound done at the seventh month of gestation showed same the finding.

The obstetric history was negative for gestational diabetes mellitus, gestational hypertension, urinary tract infections, and vaginal infections and the mother had a smooth course of pregnancy.

The mother reported an upper respiratory tract infection and cough at the beginning of 5 months of gestation, so COVID infection was suspected, but no further investigations were done.

The mother was a nonsmoker, and nonworker and she gained 5 kilograms during her pregnancy.

Birth details

The patient was born with a birth weight of 2785 g, and length of 49 cm, and a head circumference of 35 cm, with Apgar scores of 7 and 9, at 1 and 5 minutes respectively, 10 minutes later she developed respiratory distress with tachypnea without desaturation and the patient was placed on nasal cannula.

Neonatal course, diagnostic workup, and management

Upon admission to the neonatal intensive care unit, the arterial blood gas (ABG) showed respiratory acidosis, (PH = 7.23, PCO2 = 76.2 and PO2 = 74.8), which was corrected by putting the baby on high flow nasal cannula, and the rest of the laboratory tests were normal. On the third day of life, the baby developed desaturation; chest X-ray was done to show right-sided pneumothorax as shown in Fig. 1. The patient was intubated and was put on synchronized intermittent mandatory ventilation (SIMV) mode, antibiotic regimen was started and right chest tube was inserted. The baby's condition didn't improve, so CT scan of the chest was done to show bilateral ground glass opacity and interlobular septal thickening with pneumomediastinum and left pneumothorax as seen in Fig. 2, so left chest tube was inserted. Blood serology for COVID-19 immunoglobulin M (IgM) and immunoglobulin G (IgG) was sent to show high levels of serum COVID-19 IgG. The baby was diagnosed to have MIS-N and IV steroids was added to the treatment. Kidney Ureter Bladder (KUB) X-ray showed a distended bowel and prominent meconium impaction, and pelvic ultrasound showed pelvic displacement of the right kidney.Fig. 1 Three-day- old newborn with MISN presenting with distress. Finding: right pneumothorax (the whole right lung) Technique: Chest X-ray.

Fig 1

Fig. 2 Three-day- old newborn with MISN presenting with distress Finding: diffuse ground glass opacity with interlobular septal thickening, bilateral pneumothorax. Technique: CT scan of the chest.

Fig 2

The baby's condition improved gradually and he was discharged home after 10 days.

Discussion

It is known that maternal history of exposure to COVID-19 may potentially be associated with multisystem inflammation, thrombosis, and atrioventricular conduction abnormalities in the early neonatal period, however, neonatal MIS-N is relatively rare [1]. To our knowledge, there is an increasing literature describing neonates born to mothers with SARS-CoV-2 infection presenting with multisystem inflammatory syndrome in neonates. Initially, some studies refer it to a horizontal transmission due to familial clusters [2], but further studies showed evidence of transplacental transmission of SARS-CoV-2 infection [3] and transplacental transfer of Immunoglobulin G (IgG) antibodies in seropositive mothers [4].

Despite the increased hospitalization incidence of the multi-inflammatory syndrome of neonates, the diagnosis is still challenging due to the variety of clinical presentations, according to the case series [1], 18 patients out of 20 (90%) presented with cardiac involvement, 55% with respiratory involvement, requiring ventilator/CPAP and 30% with gastrointestinal involvement and other systems were less involved. Another case report reported a multisystem involvement (shock and respiratory failure) along with acute kidney injury [5]. Also, another study showed both mediastinal emphysema and pneumothorax post-COVID-19 in an adult patient [6], and another reported a case of severe spontaneous pneumothorax along with pulmonary fibrosis post-COVID-19 infection [7].

Our case presented with respiratory symptoms manifested by respiratory distress and pneumothorax along with pneumomediastinum, gastrointestinal manifestations are presented by a picture of meconium impaction.

This reported case was a full-term baby unlike another study [5], but similar to the case series mentioned before where 15% of the reported cases were full-term babies, 38.8% of them were born by Caesarean section from a symptomatic mother similarly to our case [1]. In addition, serum Immunoglobulin G (IgG) was positive similar to 18 patients out of 20 (90%) of the reported cases [1].

A recent study suggests that extensive alveolar destruction caused by COVID-19 infection may lead to bullae formation resulting in subsequent pneumothorax [8]. We also suggest that the same effect may be caused by the alveolar destruction caused by the immune system dysregulation, not the infection itself, and leads to the development of pneumothorax.

In our case, the patient was desaturated so the management focused on the respiratory symptoms by inserting chest tubes on both sides and intubating the patient on synchronized intermittent mandatory ventilation (SIMV) mode along with steroids and antibiotic regimen. Similarly, 100% of the patients reported case series who were treated with steroids along with additional treatments and discharged maximum of 38 days except 2 cases that were dead [1].

Conclusion

In conclusion, although it's a rare complication of the multisystem inflammatory syndrome of newborns, pneumothorax and pneumomediastinum are some of the clinical presentations of an Immunoglobulin G (IgG)-positive MIS-N, and should be still considered as one of the differentials even after the end of covid-19 as a global emergency.

Patient consent

Ethical clearance was obtained and patient consent was obtained from the parents.

Acknowledgments: The authors have no acknowledgements. No funding was received for conducting this study. The authors declare they have no financial interests.

This manuscript has not been previously published and is not being concurrently submitted elsewhere.

Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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