Reversible Cerebral Vasoconstriction Syndrome and Multisystem Inflammatory Syndrome in Children With COVID-19 - 18/03/22

Doi : 10.1016/j.pediatrneurol.2021.12.013

Atefeh Sadeghizadeh, MD ^a, Zahra Pourmoghaddas, MD ^b, Alireza Zandifar, MD ^c, Seyedeh Zahra Tara, MD ^d, Hamid Rahimi, MD ^b, Rana Saleh, MD ^e, Saba Ramezani, MD ^e, Mohammadreza Ghazavi, MD ^f, Omid Yaghini, MD ^f, Neda Hosseini, MD ^f, Nahid Aslani, MD ^g, Mohammad Saadatnia, MD ^h,^⁎ , Arastoo Vossough, MD, PhD ^c
^a Department of Pediatric Intensive Care, Isfahan University of Medical Sciences, Isfahan, Iran
^b Department of Pediatric Infectious, Isfahan University of Medical Sciences, Isfahan, Iran
^c Division of Neuroradiology, Department of Radiology, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania
^d Department of Pediatrics, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania
^e Department of Pediatrics, Isfahan University of Medical Sciences, Isfahan, Iran
^f Department of Pediatric Neurology, Isfahan University of Medical Sciences, Isfahan, Iran
^g Department of Pediatric Rheumatology, Isfahan University of Medical Sciences, Isfahan, Iran
^h Department of Neurology, Isfahan Neurosciences Research Center, Isfahan University of Medical Sciences, Isfahan, Iran

^∗Communications should be addressed to: Dr. Saadatnia; Department of Neurology; Isfahan Neurosciences Research Center; Isfahan University of Medical Sciences; Isfahan, Iran.Department of NeurologyIsfahan Neurosciences Research CenterIsfahan University of Medical SciencesIsfahanIran

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Abstract

Background

Multisystem inflammatory syndrome in children (MIS-C) involves multiple organs and shows increased inflammatory markers. Since the onset of the coronavirus disease 2019 (COVID-19) pandemic, several studies have reported the association between severe COVID-19 and MIS-C. Reversible cerebral vasoconstriction syndrome (RCVS) presents with thunderclap headaches and multifocal reversible vasoconstriction on imaging. RCVS is very rare in children. This article reports two cases of pediatric COVID-19 with severe MIS-C and clinical and imaging features indicative of RCVS.

Methods

Clinical, laboratory, and imaging data of the patients were reviewed. The diagnosis of RCVS was confirmed based on clinical symptomatology and brain magnetic resonance imaging findings.

Results

Two pediatric patients with clinical findings compatible with severe MIS-C and hemodynamic compromise presented to the hospital. During their hospitalization course, they developed thunderclap headaches and neurological deficits. Both were receiving vasoactive agents, intravenous immunoglobulin, and immunosuppressants. Imaging studies showed marked multifocal cerebral vasoconstriction in both cases and infarcts in one. The course and management of the patients will be presented. After controlling inflammation and elimination of triggers, both patients were ultimately symptom free upon discharge. Cerebral vasoconstriction had completely resolved on follow-up imaging.

Conclusions

Although a variety of symptoms including headaches may be seen in pediatric COVID-19 patients with MIS-C, RCVS should be considered as a differential diagnosis in cases of thunderclap headache accompanied by neurological signs in these patients. Imaging findings and follow-up are also key in establishing the diagnosis.

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Keywords : Multisystem inflammatory syndrome in children, Reversible cerebral vasoconstriction syndrome, COVID-19, Thunderclap headache

Plan

	A.S. and Z.P. have contributed equally to this article.
	Conflict of Interest: None.
	Declaration of 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.