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Efficacy of sildenafil and high-dose anakinra in an MIS-C patient with pulmonary vasculitis: A case report.
La Torre, Francesco; Calabrese, Gerolmina; Signorile, Katia; Bizzoco, Francesca; Mastrorilli, Carla; Strippoli, Antonella; Amato, Doriana; Carella, Francesco; Vairo, Ugo; Giordano, Paola; Milella, Leonardo; Cardinale, Fabio.
  • La Torre F; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Calabrese G; Pediatric Intensive Care Unit, Giovanni XXIII Pediatric Hospital, Bari, Italy.
  • Signorile K; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Bizzoco F; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Mastrorilli C; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Strippoli A; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Amato D; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Carella F; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
  • Vairo U; Pediatric Cardiology Unit, Giovanni XXIII Pediatric Hospital, Bari, Italy.
  • Giordano P; Department of Interdisciplinary Medicine, Pediatric Section, University of Bari, Bari, Italy.
  • Milella L; Pediatric Intensive Care Unit, Giovanni XXIII Pediatric Hospital, Bari, Italy.
  • Cardinale F; Department of Pediatrics, Giovanni XXIII Pediatric Hospital, University of Bari, Bari, Italy.
Front Pediatr ; 10: 1015617, 2022.
Article in English | MEDLINE | ID: covidwho-2232219
ABSTRACT
Multisystem inflammatory syndrome in children (MIS-C) is a newly identified clinical entity still not very well known in terms of epidemiology, pathogenesis, and long-term outcome. Pulmonary involvement with acute respiratory failure is an unusual life-threatening complication of MIS-C, often a reason for admission to the pediatric intensive care unit (PICU) and the use of mechanical ventilation. We present a case of a 7-year-old male patient, previously healthy, hospitalized for MIS-C, treated with intravenous immunoglobulins (IVIG), high dose methylprednisolone, and anakinra. After 2 days of the aforementioned therapy, the patient presented with hypoxia (SatO2 85% in ambient air room) and breathing difficulties. A chest computed tomography (CT) scan showed the presence of multiple bilateral basal parenchymal thickening and small basal pleural effusion and an arterial blood gas analysis revealed severe hypoxia (PaO2/FiO2 ratio, 170 mmHg). Because of a worsening of respiratory distress, the patient was transferred to the PICU, where invasive mechanical ventilation and a continuous infusion of anakinra (12 mg/kg/day) were started. An echocardiogram was performed, which showed an increase in pulmonary pressure (40 mmHg) with normal heart ejection fraction (55%), and the hypothesis of pulmonary vasculitis involving the pulmonary arterioles was made. Therefore, therapy with sildenafil (0.15 mg/kg/day) was promptly set up, with an immediate improvement of the clinical picture of respiratory failure, reduction of pulmonary pressure (23 mmHg), and subsequent extubation at 36 h with a regular clinical course until discharge. As far as we know, our case represents the first report of pulmonary vasculitis in an MIS-C patient. The use of sildenafil and high-dose continuous anakinra may represent a rescue therapy in cases of MIS-C with pulmonary vasculitis or with difficulty in extubation, allowing a short-term hospitalization in intensive care and improving the long-term outcome in these patients.
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Full text: Available Collection: International databases Database: MEDLINE Type of study: Case report / Prognostic study Language: English Journal: Front Pediatr Year: 2022 Document Type: Article Affiliation country: Fped.2022.1015617

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Full text: Available Collection: International databases Database: MEDLINE Type of study: Case report / Prognostic study Language: English Journal: Front Pediatr Year: 2022 Document Type: Article Affiliation country: Fped.2022.1015617