Acute enlargement, morphological changes, and rupture of intracranial infectious aneurysm in infective endocarditis. Serial imaging Monique Boukobza, Olivier Naggara, Xavier Duval, Jean-Pierre Laissy To cite this version: Monique Boukobza, Olivier Naggara, Xavier Duval, Jean-Pierre Laissy. Acute enlargement, morphological changes, and rupture of intracranial infectious aneurysm in infective endocarditis. Serial imaging. Journal of Clinical Neuroscience, 2020, 82, pp.237 - 240. �10.1016/j.jocn.2020.11.001�. �hal03492751� HAL Id: hal-03492751 https://hal.science/hal-03492751 Submitted on 15 Dec 2022 HAL is a multi-disciplinary open access archive for the deposit and dissemination of scientific research documents, whether they are published or not. The documents may come from teaching and research institutions in France or abroad, or from public or private research centers. L’archive ouverte pluridisciplinaire HAL, est destinée au dépôt et à la diffusion de documents scientifiques de niveau recherche, publiés ou non, émanant des établissements d’enseignement et de recherche français ou étrangers, des laboratoires publics ou privés. Distributed under a Creative Commons Attribution - NonCommercial 4.0 International License Version of Record: https://www.sciencedirect.com/science/article/pii/S0967586820316179 Manuscript_fd3021783e1c657c2c1827fcd79a87fb Title: Acute enlargement, morphological changes, and rupture of intracranial infectious aneurysm in infective endocarditis. Serial Imaging Key words: Intracranial infectious aneurysm; Infective endocarditis; aneurysm rupture; aneurysm enlargement; aneurysm morphological change; contrast enhanced MRA. Abstract A 72-year-old man received a transcatheter aortic valve implantation (TAVI) 2 years ago for leakage of the degenerative bioprosthesis with Corevalve n°31 implantation, presented infective endocarditis (IE) (streptococcus sanguinis) of the bioprosthetic aortic valve. One month after antibiotic treatment was initiated, he presented a left-sided hemiplegia, a right frontal hematoma. MRI/contrast-enhanced magnetic resonance angiography (CE MRA) revealed 2 infectious intracranial aneurysms (IIAs) of the right (10mm) and left middle cerebral artery (MCA) (M2 segment, 5mm). The right MCA IIA was treated within 1 day by glue-embolization. Seven days later, the patient acutely developed motor aphasia. CE MRA showed significant enlargement (15mm) and morphologic change of the ruptured left MCA IIA. This IIA was treated with Onyx-embolization. This case adds additional evidence that IIAs, during IE, can show rapid growth and morphological change over a 7day course and emphasizes the imperative need of close imaging follow-up when IIAs are managed by antibiotic therapy. Introduction The natural history, including growth and rupture, of unruptured intracranial infectious aneurysms (IIAs) remains unknown, even if it is considered that they have a high risk of rupture. They may develop rapidly compared to noninfectious aneurysm. Scarce radiological studies have reported their evolution over time. We report a 72-year-old man, with infective endocarditis (IE) who presented with a ruptured IIA of the left middle cerebral artery (MCA) that increased in size from 5 mm to 15 mm in 7 days before rupture with resultant hematoma Case 1 © 2020 published by Elsevier. This manuscript is made available under the CC BY NC user license https://creativecommons.org/licenses/by-nc/4.0/ A 72-year-old man was admitted 1 month ago for fevers, anemia. Two hemocultures were positive for Streptococcus sanguinis, originating from torpid dental infection. An antibiotic treatment was initiated with [gentamycin and amoxicillin]. Transthoracic echocardiography (TTE) and transesophageal echocardiography (TEE) did not show any valvular dysfunction His history consisted in an aortic valve replacement by stentless bioprosthesis O’Brien n°29 associated with single internal mammary- left anterior descending (LAD) artery coronary artery bypass graft (CABP) at 60 years of age for tight aortic stenosis; he then underwent 7 years later a right coronary revascularization with a bare and a coated stent angioplasty on the right coronary trifurcation 1year later. He further received a transcatheter aortic valve implantation (TAVI) for leakage of the degenerative bioprosthesis with Corevalve n°31 implantation. He presented abruptly a left-sided hemiplegia and dysarthria. Brain-CT and MRI in emergency showed a right frontal intraparenchymal hemorrhage (25x15 cm) and a subarachnoid hemorrhage (SAH) at the left fronto-temporal convexity. MRI showed 3 microinfarcts, a territorial ischemia and 3 cerebellar microbleeds. Contrast-enhanced magnetic resonance angiography (CE MRA) revealed 2 IIAs of the right (10mm) and left MCA (Fig 1 – a) also identified on cone-beam CT and catheter angiography (M2 segment, 5mm) during the endovascular procedure (Fig 1-b -c-d). The right MCA IIA was treated within 1 day by glue-embolization. Seven days later, under antibiotic therapy (gentamycin and amoxicillin) he abruptly presented a motor aphasia. Brain-CT showed left temporal intraparenchymal hemorrhage (47x22cm) with mass effect. Fluid-attenuated inversion recovery (FLAIR) sequence showed a hyperintense arterial rosary-like appearance assumed to slow flow in the left sylvian cistern (Fig 1 -e). CE MRA demonstrated the previously documented left MCA IIA to have clearly enlarged and ruptured (Fig 1 – f-g-h-i). On digital substraction angiography (DSA), the ruptured aneurysm measured 15mm in diameter and was treated with Onyx embolization (Fig 1-j-k-l). Follow-up MRI/CE MRA (10 days and 3 months respectively) (Fig- 1 –m) and DSA (1month) showed disappearance of both IIAs, without development of new aneurysms Discussion The incidence of IIAs is likely underestimated as IIAs can go undiagnosed given their resolution after antibiotic therapy. Given the rarity of IIA, the paucity of population-based studies, considering that DSA is not routinely performed during IEs, and the risks inherent in DSA - especially in fragile IE patients who are sometimes on anticoagulants- no prospective 2 natural history study has examined the rupture rates of IIA. Only one recent study has tried to determine the clinical course of IIAs. The main factor involving favorable clinical outcome was linked to antibiotic therapy duration [1]. Indeed, only few cases of rapid onset and rupture and of rapid growth of IIA during IE have been reported.Formed by inflammation and destruction of arterial walls - the muscular layer being usually involved- IIAs are pseudoaneurysms and carry a high risk of rupture. Seven cases of rapid appearance and rupture [2-7] have been reported since the seminal review of 5 cases by Saito et al [8], including his own one. These 10 cases of IIAs appeared in a time-interval of 1 to 5 days, except a delay of 20 days for 1 case and 30 days for 2 other cases. In 5 of these 12 cases, a new IIA appeared at the site of the previous focal occlusion site due to septic emboli [3]. Very rapid enlargement and subsequent rupture of an IIA on a previously demonstrated IIA have been documented, to the best of our knowledge, in only 3 cases. Tashima [9] reported 1 case of enlarged IIA of an MCA branch, observed on a second angiography 2 weeks after the first DSA examination. In Koffie et al’ s case [10] an IIA of an MCA branch, revealed by SAH, enlarged from 1.5 to 5mm in a time interval of 21h with catastrophic intracranial hemorrhage secondary to rupture. In John et al’ s case 2 [3], at 2-week repeat angiography the IIA previously documented was stable, and follow-up angiography 4 weeks later showed an enlargement of the IIA (Table 1). In the absence of knowledge of unfavorable morphological change, the initial morphological pattern, i.e. the saccular IIAs, should be predictive of worse outcome - rupture and/or growth - as reported by Rice et al [1] who consider that these IIAs should be treated interventionnally in addition to antibiotic therapy. Our case adds evidence that IIAs can show rapid increase in size and morphological change over a course of 7 days. Such a rapid transformation over time, with the risk of rupture, is unpredictable and specific to IIAs related to their pathology, unlike non-infectious cerebral aneurysms. One can ask whether at this time the left MCA aneurysm was not treated initially like the right one. Given the experience of this case, such a case would be now treated as soon as possible after the diagnosis. The usefulness of repeated CE- MRA or CTA within 7 days in all IE patients is questionable. Moreover, a consensus exists about the fact that noninvasive imaging cannot often detect IIAs of 3 mm or less. The use of plain MRA does not allow detection of these MA; on the contrary, CE-MRA has shown better sensitivity than noncontrast MRA in non-infectious IA [11]. DSA should be indicated in cases of non-conclusive MRA. 3 The growth of these aneurysms, evaluated by serial MRA/CE MRA in large series [12] led to similar results (10% over 47 months and 3.9% per year, respectively), those of >8mm in diameter being the most concerned. In addition, aneurysm growth was associated with a high risk of rupture. One exceptional case has been reported of early and gradual enlargement, at 20, 30 and 60 days after rupture and beyond the shrinkage of a distal MCA aneurysm, proved of congenital origin on pathologic study [13]. The current case, as those previously reported, highlights 2 important clinical issues: (i) IIAs showing rapid growth, which have a high risk of rupture within a short period, need close brain imaging and often emergent intervention. Imaging monitoring should be even more stringent in IE patients under antibiotic therapy, without forgetting the risks of repeat DSA and the utility of noninvasive imaging, i.e. ACT and CE MRA; (ii) if IIA shows rapid growth with serial imaging, it should be treated without further delay. Conclusion Progression and subsequent rupture of an IIA previously demonstrated by neuroimaging has rarely been documented. Our case adds additional evidence that IIAs are dynamic lesions that can show a rapid enlargement and transformation over a short time period. The risk of rupture clearly reinforces first the necessity of repeated imaging follow-up in IE patients under antibiotic therapy. DSA should be reserved to patients in whom non-invasive vessel imaging – CE MRA/CTA- is inconclusive, and those showing IIA enlargement where active management should not be delayed. Figures Legends Figure 1 a – Contrast-enhanced magnetic resonance angiography (CE MRA): Maximum intensity projection (MIP) shows 2 intracranial infectious aneurysms (IIAs) in a branch of the right (10mm, short arrow) and left middle cerebral artery (MCA) (M2 segment, 5mm, long arrow). b- Digital subtraction angiography (DSA), lateral view, obtained the same day, shows the saccular IIA located on a M4 branch of the right MCA (arrow). c- Intraoperative DSA image shows the catheter advancing to the orifice of the right MCA IIA before the injection of glue. d- Cone-beam angio-CT (CB-ACT) obtained during the endovascular procedure, shows an IIA at the M2 segment of the left MCA (5mm, arrow); 4 e- Fluid-attenuated inversion recovery (FLAIR) MRI sequence, obtained after motor aphasia onset occurred at day 7 of embolization of the right MCA IIA, shows a hyperintense arterial rosary-like appearance assumed to slow flow in the left sylvian cistern, as shown in the serial images of DSA (see below, j-k.) surrounded by an area of parenchymal hyperintensity. Contrast-enhanced magnetic resonance angiography (CE MRA): maximum intensity projection (MIP) (f, g) and axial (h) and sagittal (i) multiplanar reformat (MPR) shows enlargement of the left MCA IIA from 5mm to 15 mm in diameter (arrow) along with morphological changes. The ruptured IIA appears multilobulated with irregular walls. Note the presence of a localized subarachnoid hemorrhage at the left temporal convexity j-k -l- DSA shows a slow fill-in within the IIA and delayed clearance (j-k) and the disappearance of the IIA after glue embolization (l) m: Ten-day follow-up CE MRA show the disappearance of the IIA. References 1 -Rice CJ, Cho SM, Marquardt RJ, Zhang LQ, Khoury J, Hardman J, Wisco D, Hussain MS, Uchino K. Clinical course of infectious intracranial aneurysm undergoing antibiotic treatment. J Neurol Sci. 2019 Aug 15; 403:50-55. 2-Sorkin GC, Jaleel N, Mokin M, Dumont TM, Eller JL, Siddiqui AH. Ruptured mycotic cerebral aneurysm development from pseudoocclusion due to septic embolism. Surg Neurol Int.2013; 4:144. 3-John S, Walsh KM, Hui FK, Sundararajan S, Silverman S, Bain M. Dynamic angiographic nature of cerebral mycotic aneurysms in patients with infective endocarditis. Stroke 2016;47: e8–e10 4-Samaniego EA, Ortega S, Stander L, Leira EC. Development of a cerebral mycotic aneurysm within 5 days. Neurol Clin Pract. 2017; 7: 449-50. 5-Wang JL, Hinduja AP, Powers CJ. Successful coil embolization of a ruptured mycotic aneurysm that developed three days after septic embolic infarction: Case report and review of the literature. J Clin Neurosci. 2017; 39:95–8. 5 6-Brimley C, Griessenauer CJ, Dalal S, Schirmer C, Goren O. Acute Development and Rupture of a Basilar Tip Mycotic Aneurysm. World Neurosurg. 2019; S1878-8750(19)300907. 7- Goldschmidt E, Faraji AH, Salvetti D, Zussman BM, Jadhav A. Intracranial vessel occlusion preceding the development of mycotic aneurysms in patients with endocarditis. BMJ Case Rep. 2019 ;12(12): e015480 8 -Saito A, Kawaguchi T, Hori E, Kanamori M, Nishimura S, Sannohe S, Kaimori M, Sasaki T, Nishijima M. Subarachnoid hemorrhage after an ischemic attack due to a bacterial middle cerebral artery dissecting aneurysm: case report and literature review. Neurol Med Chir (Tokyo). 2014;54 :196-200 9 -Tashima T, Takaki T, Hikita T, Kuroiwa S, Hamanaka N, Takahashi M. Bacterial Intracranial Aneurysm Associated with infective endocarditis: A Case Showing Enlargement of Aneurysm Size No Shinkei Geka. 1995;23: 985–9 10 -Koffie RM, Stapleton CJ, Torok CM, Yoo AJ, Leslie-Mazwi TM, Codd PJ. Rapid growth of an infectious intracranial aneurysm with catastrophic intracranial hemorrhage. J Clin Neurosci. 2015; 22: 603–5. 11 -Cirillo M, Scomazzoni F, Cirillo L, Cadioli M, Simionato F, Iadanza A, Kirchin M, Righi C, Anzalone N. Comparison of 3D TOF-MRA and 3D CE-MRA at 3T for imaging of intracranial aneurysms. Eur J Radiol. 2013; 82: e853-9. 12 -Serrone JC, Tackla RD, Gozal YM, Hanseman DJ, Gogela SL, Vuong SM, Kosty JA, Steiner CA, Krueger BM, Grossman AW, Ringer AJ. Aneurysm growth and de novo aneurysms during aneurysm surveillance. J Neurosurg. 2016; 125:1374-82. 13 -Inoue A, Watanabe H, Kumon Y, Ohtsuka Y, Tagawa M, Seyama T Takechi A, Mizuno Y, Kitazawa R, Ohnishi T. A case of peripheral middle cerebral artery aneurysm showing repeated morphological changes. No Shinkei Geka.2015;43:713–9. 6 Blood culture IIA (DSA) Valve lesion Species Size location (mm) M/48 Native* Enterococcus fecalis Koffie, 20159 M/49 Aortic, mechanical Cocci + John, 201610, Case 2 F/31 Aortic, prosthesis Streptococcus viridans Tashima, 19958 Age (ys) Sex NA R MCA (M2) 1.5 R MCA, distal 2 IIA 2 L MCA (M2) 2 M/72 Treatment 1 Follow-up 1 NA Antibiotics* scattered SAH Follow-up 2 Treatment 2 Outcome D15:DSA: IIA enlarge clip No residual IIA, asymptomatic H21: coma. Hematoma. IIA 5mm evacuation /clip MCA(M2) Post-op D1: died Coil + Onyx E° 1month: No residual IIA Glue E° No residual IIA Onyx E° No residual IIA Antibiotics* IIA 1 Present case, 2020 Associated findings microhemorrhages, old SAH 3 microinfarcts, ischemic stroke, microbleeds Aortic, Streptococcus Bioprosthesis sanguinis IIA 1 R MCA, distal IIA 2 L MCA (M2) D14: stable 4w: resolve D14: stable 4w: enlarge gentamycine, amoxicillin 10 5 D7: hematoma. IIA: 15mm Table 1: Summary of literature cases of rapid enlargement and subsequent rupture of an intracranial infectious aneurysm IIA: intracranial infectious aneurysm; DSA: digital substraction angiography; *: unspecified; NA : not available; D: day; R: Right; MCA: middle cerebral artery; H: hour; L: left; SAH: subarachnoid hemorrhage; w: week; E°: embolization.