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Severe headache in a middle-aged woman after device closure of a ventricular septal defect

Published online by Cambridge University Press:  09 November 2022

Jungbin Lee Open the ORCID record for Jungbin Lee [Opens in a new window]  and
Jinyoung Song Open the ORCID record for Jinyoung Song [Opens in a new window]
Jungbin Lee
Affiliation:
Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea
Jinyoung Song*
Affiliation:
Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, South Korea
*
Author for correspondence: J. Song, MD, PhD, Department of Pediatrics, Samsung Medical Center, Heart Vascular Stroke Institute, Grown-up Congenital Heart Clinic, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 06351, South Korea. Tel.: +82-2-3410-3539; Fax: +82-2-3410-0043. E-mail: amyjys@naver.com
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Abstract

Reversible cerebral vasoconstriction syndrome presents with thunderclap headache and represents a group of conditions that show reversible multifocal narrowing of cerebral arteries. Some patients who undergo device closure of an atrial septal defect complain of headache, which are posited as a migraine. Here we report a case of severe headache due not to migraine but reversible cerebral vasoconstriction syndrome after device closure of a ventricular septal defect.

Keywords

Ventricular septal defectdevice closureheadachereversible cerebral vasoconstriction syndrome
Type
Brief Report
Information
Cardiology in the Young , Volume 33 , Issue 6 , June 2023 , pp. 1023 - 1024
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Creative Commons
Creative Common License - CCCreative Common License - BY
This is an Open Access article, distributed under the terms of the Creative Commons Attribution licence (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution and reproduction, provided the original article is properly cited.
Copyright
© The Author(s), 2022. Published by Cambridge University Press

Approximately 15% of patients complain of headache after device closure of an atrial septal defect, and most of them are believed to be migraine attacks and improved spontaneously. Reference Rodes-Cabau, Horlick and Ibrahim1 Reversible cerebral vasoconstriction syndrome presents with thunderclap headache and represents a group of conditions that show reversible multifocal narrowing of cerebral arteries. Reference Sorensen2 We present a case of a 48-year-old women who presented with severe headache after device closure of a ventricular septal defect. For this patient, we diagnosed reversible cerebral vasoconstriction syndrome that improved on follow-up angiography.

Case description

A 48-year-old woman came to our hospital for device closure of a ventricular septal defect. She had no specific medical history other than intermittent dizziness and palpitation. Chest X-ray showed no cardiomegaly, but echocardiography revealed a restrictive perimembranous ventricular septal defect and left atrial enlargement. We performed transcatheter ventricular septal defect closure using a Cocoon ventricular septal defect aneurysmal-type occluder 12–8 mm (Fig 1). Reference Morray3 The next day, a junctional rhythm change was detected. After methylprednisolone pulse therapy for 3 days, sinus conversion was confirmed. She was discharged with oral prednisolone and aspirin.

Figure 1. Transcatheter VSD closure using a cocoon VSD aneurysmal-type occluder 12–8 mm (white arrow).

On the fifth day after device closure, a severe tightening headache of the whole head was reported. The headache worsened when she laid down and was relieved when she stood. Due to a constant headache even with clopidogrel, she visited the emergency department on the seventh day after device closure.

Her vital signs were stable, and there were no abnormal findings on neurologic examination. No evidence of intracranial haemorrhage nor infarction was found on CT. Cerebrospinal tapping was performed and showed normal pressure but 10,000/μL red blood cell on three-bottle test. We suspected a chronic subarachnoid haemorrhage. CT angiography and transfemoral cerebral angiography were carried out and showed multi-segmental stenosis on internal carotid artery, anterior carotid artery, and posterior carotid artery but no aneurysm or structural abnormality (Fig 2a). Finally, we confirmed chronic subarachnoid haemorrhage due to reversible cerebral vasoconstriction syndrome. After administration of nimodipine, her headache resolved, and multi-segmental stenosis was much improved on follow-up CT angiography and brain MRI one month after diagnosis (Fig 2b). We got the informed consent for publication of this case from the patient.

Figure 2. ( a ) CT angiography showed multifocal stenosis in multiple intracranial arteries (red arrows) and ( b ) follow-up CT angiography one month later showed great improvement (white arrows).

Discussion

After intra-cardiac device closure, headache supposed to be a migraine is well known and reported to be improved with clopidogrel. Reference Wilmshurst, Nightingale, Walsh and Morrison4 However, the pathophysiology of such pain remains unclear. Reference Spencer, Qureshi and Sommer5 Since it tends to improve spontaneously within 6–12 months in most patients, the headache often resolves without sufficient evaluation. Reference Spencer, Qureshi and Sommer5 To my knowledge, this is the first report of reversible cerebral vasoconstriction syndrome after ventricular septal defect device closure through sufficient evaluation for headache.

Since the headache reported by the patient was different from a migraine, a thorough evaluation was conducted. The headache mimicked a tightening of the entire head and worsened when she laid down. She even woke up with a severe headache. In contrast, migraine mainly shows a unilateral location, a pulsating pattern, and is accompanied by an aura in about 30% of cases.Reference Spencer, Qureshi and Sommer5

Reversible cerebral vasoconstriction syndrome is characterised by thunderclap headache and shows relatively good prognosis. Although it is most common in middle-aged women, the cause or pathophysiology is not clear. Reference Wintzer-Wehekind, Horlick and Ibrahim6 Risk factors of reversible cerebral vasoconstriction syndrome are associated with medicine like cannabis, vasoconstrictive drugs, or steroid. Reference Sorensen2

As indicated by its name, reversible cerebral vasoconstriction syndrome improves without treatment, although calcium channel blockers such as nimodipine might help. No association between intracardiac device and reversible cerebral vasoconstriction syndrome has been reported. In this case, the reasons for reversible cerebral vasoconstriction syndrome were not clear but might include the device itself or association with methylprednisolone. As in atrial septal defect device closure, the pathophysiology of headache after device closure can be attributed to a microembolism, nickel allergy, or steroid treatment but might not be related to any. 7 Further study is needed on this event.

Conclusion

We treated reversible cerebral vasoconstriction syndrome after device closure of ventricular septal defect that was resolved without any consequences.

Acknowledgements

None.

Financial support

This research received no specific grant from any funding agency, commercial, or not-for-profit sectors.

Conflicts of interest

None.

References

Rodes-Cabau, J, Horlick, E, Ibrahim, R, et al. Effect of clopidogrel and aspirin vs aspirin alone on migraine headaches after transcatheter atrial septal defect closure: the CANOA randomized clinical trial. JAMA 2015; 314: 2147–2154.CrossRefGoogle ScholarPubMed
Sorensen, DM. Reversible cerebral vasoconstriction syndrome. JAMA Neurol 2016; 73: 232–233.CrossRefGoogle ScholarPubMed
Morray, BH. Ventricular septal defect closure devices, techniques, and outcomes. Interv Cardiol Clin 2019; 8: 1–10.Google ScholarPubMed
Wilmshurst, PT, Nightingale, S, Walsh, KP, Morrison, WL. Clopidogrel reduces migraine with aura after transcatheter closure of persistent foramen ovale and atrial septal defects. Heart 2005; 91: 1173–1175.CrossRefGoogle ScholarPubMed
Spencer, BT, Qureshi, Y, Sommer, RJ. A retrospective review of clopidogrel as primary therapy for migraineurs with right to left shunt lesions. Cephalalgia 2014; 34: 933–937.CrossRefGoogle ScholarPubMed
Wintzer-Wehekind, J, Horlick, E, Ibrahim, R, et al. Effect of clopidogrel and aspirin vs aspirin alone on migraine headaches after transcatheter atrial septal defect closure: one-year results of the CANOA randomized clinical trial. JAMA Cardiol 2021; 6: 209–213.CrossRefGoogle ScholarPubMed
Headache Classification Subcommittee of the International Headache Society. The international classification of headache disorders: 2nd edition. Cephalalgia 2004; 24 Suppl 1: 9–160.Google Scholar
Figure 0

Figure 1. Transcatheter VSD closure using a cocoon VSD aneurysmal-type occluder 12–8 mm (white arrow).

Figure 1

Figure 2. (a) CT angiography showed multifocal stenosis in multiple intracranial arteries (red arrows) and (b) follow-up CT angiography one month later showed great improvement (white arrows).