1 Brief Communications Migrainous Infarction in a Patient With Sporadic Hemiplegic Migraine and Cystic Fibrosis: A 99mTc-HMPAO Brain SPECT Study Valentina Mancini, MD ; Giulio Mastria, MD; Viviana Frantellizzi, MD; Patrizia Troiani, MD, PhD; Stefania Zampatti, MD; Stefania Carboni, MSc; Emiliano Giardina, MSc, PhD; Rosa Campopiano, MSc, PhD; Stefano Gambardella, MD, PhD; Federica Turchi, MD; Barbara Petolicchio, MD, PhD; Massimiliano Toscano, MD, PhD; Mauro Liberatore, MD; Alessandro Viganò, MD, PhD; Vittorio Di Piero, MD, PhD Genetic mutations of sporadic hemiplegic migraine (SHM) are mostly unknown. SHM pathophysiology relies on cortical spreading depression (CSD), which might be responsible for ischemic brain infarction. Cystic fibrosis (CF) is caused by a monogenic mutation of the chlorine transmembrane conductance regulator ( CFTR), possibly altering brain excitability. We describe the case of a patient with CF, who had a migrainous stroke during an SHM attack. A 32-year-old Caucasian male was diagnosed with CF, with heterozygotic delta F508/unknown CFTR mutation. The patient experiences bouts of coughing sometimes triggering SHM attacks with visual phosphenes, aphasia, right-sided paresthesia, and hemiparesis. He had a 48-hour hemiparesis triggered by a bout of coughing with hemoptysis, loss of consciousness, and severe hypoxia-hypercapnia. MRI demonstrated transient diffusion hyperintensity in the left frontal-parietal-occipital regions resulting in a permanent infarction in the primary motor area. Later, a brain perfusion SPECT showed persistent diffuse hypoperfusion in the territories involved in diffusion-weighted imaging alteration. Migrainous infarction, depending on the co-occurrence of 2 strictly related phenomena, CSD and hypoxia, appears to be the most plausible explanation. Brain SPECT hypoperfusion suggests a more extensive per- manent neuronal loss in territories affected by aura. CF may be then a risk factor for hemiplegic migraine and stroke since bouts of coughing can facilitate brain hypoxia, triggering auras. Key words: cortical spreading depression, cystic fibrosis, aura, SHM, CFTR Abbreviations: CF cystic fibrosis, CSD cortical spreading depression, DWI diffusion-weighted imaging, ED emergency depart- ment, FHM familial hemiplegic migraine, HM hemiplegic migraine, NSAID nonsteroidal antiinflammatory drugs, PFO patent foramen ovale, RCVS reversible cerebral vasoconstriction syndrome, SHM sporadic hemiplegic migraine (Headache 2019;0:1-6) Headache doi: 10.1111/head.13472 © 2019 American Headache Society Published by Wiley Periodicals, Inc. ISSN 0017-8748 From the Department of Human Neurosciences, Sapienza University of Rome, Rome, Italy (V. Mancini, G. Mastria, F. Turchi, B. Petolicchio, M. Toscano, A. Viganò, and V. Di Piero); Department of Radiological, Oncological and Anatomopathological Sciences, Sapienza University of Rome, Rome, Italy (V. Frantellizzi and M. Liberatore); Department of Pediatrics, Sapienza University of Rome, Rome, Italy (P. Troiani); Genomic Medicine Laboratory UILDM, IRCCS Santa Lucia Foundation, Rome, Italy (S. Zampatti, S. Carboni and E. Giardina); Molecular Genetics Center, IRCCS Neuromed, Pozzilli, Italy (S. Zampatti, R. Campopiano, and S. Gambardella); Department of Biomedicine and Prevention, University of Rome “Tor Vergata,” Rome, Italy (E. Giardina). Address all correspondence to V. Mancini, Department of Human Neurosciences, Sapienza University of Rome, Viale dell’Università 30 Rome 00185, Italy, email: v.mancini92@gmail.com Accepted for publication October 29, 2018.