Rapid sequential development and rupture of mycotic aneurysms within a period of days in a patient with graft-versus-host disease and angiotropic Scedosporium apiospermum infection

Abstract

Background: Fungal origin mycotic aneurysms are rare and carry a high mortality rate. Scedosporium apiospermum is an ubiquitous fungus which has been described to cause devastating infections in immunocompromised hosts.

Case description: We report a case of a 23-year-old patient with Burkitt's lymphoma and graft-versus-host disease admitted with intracerebral hemorrhage and sequential development of 12 anterior circulation aneurysms from disseminated Scedosporium infection. Despite aggressive surgical and antimicrobial treatment, the patient died 6 months later from multiorgan failure. The notable feature of this case is the rapid angioinvasiveness of the infection with new aneurysm formation within days of clear angiographic imaging despite the apparent lack of skull base osteomyelitis.

Conclusion: We highlight the difficulties in treating fungal mycotic aneurysms and the associated high mortality. While a less aggressive approach might have been appropriate given the known poor outcomes, the age of the patients as well as the family wishes dictated the treatment decisions.

Keywords: Mycotic aneurysm; Scedosporium apiospermum; Surgery.

Figure 1:

Depiction of the most relevant imaging findings from the patient’s treatment. (a) Presentation CT head demonstrating left inferior frontal hemorrhage suspicious of vascular origin, (b-d) initial diagnostic DSA, (b) lateral view of the left ICA injection demonstrating ×1 fusiform aneurysm on the orbitofrontal branch of the left ACA (arrow) and 3 fusiform aneurysms on the frontopolar branch of the left ACA (arrowheads), (c) AP view of the left ICA injection demonstrating fusiform aneurysm at the distal ATA of the left MCA (arrowhead), (d) lateral view of the right ICA injection demonstrating a fusiform aneurysm on the frontopolar branch of the right ACA (arrowhead), (e) CT scan obtained on postbleed day 4 and postoperative day 2 following parent vessel occlusion of the left frontopolar branch of the left ACA demonstrating new IVH and fresh blood in the inferior left frontal lobe suggesting re-bleed, (f-h) DSA obtained following re-bleeding on postbleed day 4, (f) oblique view of the left ICA injection demonstrating interval growth of the untreated left orbitofrontal branch fusiform aneurysm (arrow), (g) lateral view of the right ICA injection showing three new fusiform aneurysms on the A2 segment of the right ACA (arrowhead), (h) AP view of the left ICA injection showing new fusiform aneurysms of the distal AChA and PCOMA (arrowheads), (i and j) postbleed day 8 following further surgery with bilateral A2 occlusion and bifrontal decompression, (i) CT head demonstrating extensive bifrontal decompression and swelling following intraoperative sacrifice of bilateral A2s, (j) intraoperative DSA demonstrating no flow in the left ACA at the A2 segment (arrowhead) with new aneurysm formation at the ACOMA (arrow), (k-l) interval DSA on postbleed day 9, (k) AP injection of the right ICA demonstrating growth of the ACOMA aneurysm (arrow), (l) intraprocedural image on postbleed day 9 during parent vessel occlusion of the right A1 to treat the enlarging ACOMA aneurysm (arrowhead), (m) Interval imaging on postbleed day 10 demonstrating growth of the previously identified AChA and PCOMA aneurysms (arrow), (n) interval imaging on postbleed day 10 demonstrating stable ATA aneurysm, (o) postoperative CT head following pterional craniotomy and parent vessel occlusion of the growing AChA and PCOMA aneurysms and the stable ATA aneurysm, and (p-r) follow-up MRI 1 month following initial presentation demonstrating DWI lesions in both ACA territories, although not extensive and not encompassing the whole territories despite occlusion of both A2s and a DWI lesion in the left thalamus in keeping with hypodensities seen on presentation CT.