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Case Report
Hereditary hemorrhagic telangiectasia with bilateral pulmonary vascular malformations and a cerebral abscess as a complication: A case report
1 MD, PhD student, Clinical Department of Diagnostic and Interventional Radiology, University Hospital Centre Osijek, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
2 Associate Professor, Department of Hematology, Clinic of Internal Medicine, University Hospital Centre Osijek, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
3 Medical student, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
4 Associate Professor, Health Centre Osijek, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
5 MD, PhD, Clinic of Paediatrics, University Hospital Centre Osijek, Croatia; Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
6 MD, PhD, Department of Neurosurgery, University Hospital Centre Osijek, Faculty of Medicine, Josip Juraj Strossmayer University of Osijek, Osijek, Croatia
Address correspondence to:
Zdravka Krivdić Dupan
Clinical Department of Diagnostic and Interventional Radiology, University Hospital Centre Osijek, J. Huttlera 4, Osijek,
Croatia
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Article ID: 100105Z12ZD2022
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How to cite this article
Dupan ZK, Periša V, Guljaš S, Laslo D, Salha T, Tomac V, Koruga N. Hereditary hemorrhagic telangiectasia with bilateral pulmonary vascular malformations and a cerebral abscess as a complication: A case report. J Case Rep Images Surg 2022;8(1):4–8.ABSTRACT
Introduction: Hereditary hemorrhagic telangiectasia (HHT) also known as Osler–Weber–Rendu syndrome is a relatively common, under-recognized autosomal-dominant disorder that results from multisystem vascular dysplasia. It is characterized by telangiectasis and arteriovenous malformations of skin, mucosa, and viscera.
Case Report: A 26-year-old man presented with an 8-day history of headache, fever, cough, right hemiparesis, motor dysphasia, and dysgraphia. The urgent brain computed tomography (CT) scan revealed a bulky rounded left frontal lobe lesion, with peripheral contrast enhancement and marked perilesional edema, indicating a brain abscess or metastasis. A brain magnetic resonance imaging showed an intra-axial hypovascular round enhancing left frontal lobe lesion, with pronounced perilesional edema. Laboratory findings discovered severe polycythemia with normal level of erythropoietin and gas analysis of blood revealed hypoxemia of secondary cause. Computed tomography of the lungs suggested pulmonary arteriovenous malformations (AVMs) in the right and left lung. Contrast-enhanced chest CT scan confirmed AVM of the lungs, while abdominal CT scan ruled out AVM of the liver. The brain abscess was treated neurosurgically. The patient reported positive family history of epistaxis.
Conclusion: Brain abscess may be the first manifestation of HHT and early clinical recognition of HHT in patients with positive family history of epistaxis may help to prevent complications.
Keywords: Arteriovenous malformations, Brain abscess, Hereditary hemorrhagic telangiectasia, Hypoxemia, Polycythemia
SUPPORTING INFORMATION
Author Contributions
Zdravka Krivdić Dupan - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Vlatka Periša - Substantial contributions to conception and design, Revising it critically for important intellectual content, Final approval of the version to be published
Silva Guljaš - Substantial contributions to conception and design, Revising it critically for important intellectual content, Final approval of the version to be published
Dorian Laslo - Substantial contributions to conception and design, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Tamer Salha - Substantial contributions to conception and design, Drafting the article, Final approval of the version to be published
Višnja Tomac - Analysis of data, Revising it critically for important intellectual content, Final approval of the version to be published
Nenad Koruga - Drafting the article, Final approval of the version to be published
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this article.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
Copyright© 2022 Zdravka Krivdić Dupan et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
