Multiple Mycotic Aneurysms Infected with Haemophilus influenzae Alexander F. Nath, and Michael J. Clarke, Newcastle upon Tyne, United Kingdom
Mycotic aneurysms are a relatively uncommon phenomenon that can pose a distinct treatment dilemma for the treating surgeon. It is rare to find Haemophilus influenzae as the causative organism, and it is even more rare to find multiple aneurysms in the same patient. We present the first case to our knowledge to be described in the literature of multiple mycotic aneurysms caused by H. influenzae. The treatment presenteddusing high-dose antibiotics and cryopreserved homograftsdhas worked well in this instance. This approach is supported in the literature.
Osler1 first used the term ‘‘mycotic aneurysm’’ in 1885 to describe a nonsyphilitic infection of the arterial wall. Today the term is used to describe any arterial aneurysm that has an infective cause. Mycotic aneurysms specifically infected with Haemophilus influenzae are documented as extremely rare.2 To date there have been 4 reported mycotic aneurysms infected with H. influenzae in the published literature. All were isolated instances and none of the cases noted multiple aneurysms in the same patient. We report a patient with multiple mycotic aneurysms infected with H. influenzae, and we believe that this case is the first of its kind.
CASE REPORT A 57-year-old man presented to his local hospital with a short history of general malaise, back pain, and a tender pulsatile lump in his right groin. Blood results on
Northern Vascular Centre, Freeman Hospital, High Heaton, Newcastle Upon Tyne, United Kingdom. Correspondence to: Alexander F. Nath, MBBS, MRCSEd, Northern Vascular Centre, Freeman Hospital, High Heaton, Newcastle upon Tyne, NE7 7DN, United Kingdom; E-mail: [email protected] Ann Vasc Surg 2014; 28: 741.e11–741.e13 http://dx.doi.org/10.1016/j.avsg.2013.07.016 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: December 11, 2012; manuscript accepted: July 15, 2013; published online: February 2, 2014.
admission revealed a hemoglobin (Hb) level of 12.1 g/ dL, a platelet count of 548 109 L, a white blood cell count (WBC) of 10.4 109 L, a C-reactive protein (CRP) level of 64 mg/L, and an erythrocyte sedimentation rate (ESR) of 38 mm/hr. A computed tomography angiography (CTA) scan of the aorta and lower limbs revealed the presence of a right common femoral artery aneurysm and saccular aneurysmal change of the distal descending thoracic aorta (Fig. 1). A provisional diagnosis of nonspecific inflammatory arteritis of the medium and large vessels was made, and the patient was prescribed high-dose steroids and methotrexate. A repeat CTA scan performed 20 days later at the same hospital revealed progression of the aneurysmal disease. The patient was then transferred to the regional vascular service. Additional review of the CTA scan confirmed a right common femoral artery aneurysm, a multilobulated saccular aneurysm of the distal thoracic aorta, and multiple small saccular aneurysms of the ascending aorta. The morphology of the aneurysms was strongly suspicious of a mycotic etiology, although there was no history of recent infection. An echocardiogram revealed no evidence of endocarditis, and a labeled white cell scan was inconclusive. It was noted that the aneurysmal section in the descending thoracic aorta had increased in size from 35 to 43 mm. A decision was made to undertake repair of the right femoral aneurysm in an attempt to establish a firm diagnosis. This was achieved by excision and in situ replacement with a rifampicin-soaked Dacron graft (DuPont, Kinston, NC)d the intent being to preserve his autologous vein for possible future aortic reconstruction. H. influenzae was grown from the aneurysm wall sent for culture, although the thrombus revealed a negative culture. Initially, the patient was started on intravenous amoxicillin 2 g, 4 times
741.e11
741.e12 Case reports
Annals of Vascular Surgery
Fig. 2. The second of the thoracic aneurysms repaired with a homograft.
Fig. 1. Computed tomography angiography scan revealing the 2 aneurysmal sections of the thoracic aorta. daily, after guidance of sensitivities, for a period of 2 weeks. This was then rationalized to an oral dose for an additional 4 weeks. The ascending aorta and distal thoracic aorta were subsequently repaired in separate procedures 16 and 21 days later, respectively, with excision of the diseased aorta and in situ replacement using cryopreserved homografts (Fig. 2). The culture results were negative from the excised specimens, which was unsurprising given the directed prolonged antibiotic treatment; however, the histologic results were consistent with mycotic aneurysm. The patient made a full recovery and was discharged 13 days later. He has been followed up with annual CTA scans, and at 28 months postsurgery has no signs of recurrent disease and no late aneurysmal dilatation (Fig. 3).
DISCUSSION H. influenzae in particular is an uncommon causative organism for the development of mitotic aneurysms. We have discovered 4 documented reports in the literature.2e5 Mycotic aneurysms themselves are said to be rare; multiple ones are exceptional.6 A recent article by Sedivy et al.6 noted no instances of H. influenzae infection in a case series of 32 patients over 15 years. Our literature search has revealed 2
Fig. 3. Computed tomography scan of reconstruction at 28 months showing no late aneurysmal dilatation or recurrence.
cases of multiple mycotic aneurysms specifically of the aorta,7,8 and neither had H. influenzae isolated. In the first, by Parameswaran,7 Aspergillus niger was found to be the causative organism. The second case, by Szendro et al.,8 was culture-negative at presentation despite heart valve vegetations, leukocytosis, and elevated ESR and CRP levels. In isolation, the medical treatment of mycotic aneurysms carries a very high mortality rate.9e11 Surgical treatment is frequently complicated by the development of recurrent infection. Prosthetic material will become a further focus of infection, leading to the inevitable consequence of rupture and death. In the infrarenal aorta, in situ repair or ligation and extra-anatomic bypass would be the mainstays of treatment. In the thoracic aorta, the options are less straightforward. As a temporizing measure, the aneurysmal section can have an endovascular stent placed before formal repair after antibiotic treatment. Semba et al.12 have described the use of endovascular stent grafts in the treatment of 3 patients with mycotic aneurysms of the thoracic aorta. The first case required a repeat procedure
Vol. 28, No. 3, April 2014
because of migration. The second was stented concurrently with open ruptured abdominal aortic mycotic aneurysm repair. The final case was a secondary procedure after interposition graft rupture. Notably, there were no complications from these procedures or any persistent bacteremia. Cadaveric homografts have been reported to be more resistant to infection.11,13 Knosalla et al.13 described treating 8 patients between 1988e1994 who had mycotic aneurysms of the thoracic aorta. These patients had either previously undergone cardiac transplant or had valvular endocarditis. Of these 8 patients, 6 recovered well and the bacterial infection was fully treated. One of the remaining 2 patients died from salmonella sepsis and the second required additional surgery. Traditionally, there has been some concern in using homografts for aortic replacement because of concerns of late graft deterioration, thrombosis, and calcification.14e20 Kieffer et al.14 have presented their series of using allografts in the replacement of infected infrarenal grafts. They used a mixture of fresh and cryopreserved allografts. They postulated that the initial poor results seen with allografts could be explained by the cryopreservation and thawing techniques at the time, which has improved in recent years. They also suggested that the source of the graft harvest will also play a part. It was noted that the graft segments that were especially prone to late complications had been harvested from the descending aorta. They postulated that this could be because of their lower number of mural elastic fibers. Brown et al.21 have also presented their series of using cryopreserved allografts and have not seen aneurysmal change as a particular problem; however, their follow-up has been midterm only. We have presented a case of multiple mycotic aneurysms with H. influenzae as a causative organism. The patient was previously fit and well with no chest symptoms or previous immunosuppression. We have found no other examples of this in the literature. Our approach to treatment is supported by the literature, and the staged manner of the procedures has, in our opinion, produced the best result for the patient with the least morbidity. We acknowledge the support and contribution to the management of this case from John H. Dark FRCS, Consultant Cardiothoracic Surgeon, Freeman Hospital, Newcastle upon Tyne, United Kingdom.
Case reports 741.e13
REFERENCES 1. Osler W. The Gulstonian Lectures, on Malignant Endocarditis. Br Med J 1885;1:467e70. 2. Takahashi Y, Tsutsumi Y, Monta O, et al. Mycotic aneurysm of the descending thoracic aorta caused by Haemophilus influenzae. J Card Surg 2010;25:218e20. 3. Degener JE, Voesten HG, Nap M, et al. Rupture of an aortic aneurysm caused by Haemophilus influenzae type B infection. Vasa 1993;22:247e50. 4. Adlakha A, Yale SH, Patel R, et al. Haemophilus influenzae serotype f: an unusual cause of a mycotic aneurysm in an adult. Mayo Clin Proc 1994;69:467e8. 5. Whitfield CG, Lonsdale RJ, Rahbour G, et al. Infective abdominal aortic aneurysm due to Haemophilus influenza identified via the polymerase chain reaction. Eur J Vasc Endovasc Surg 2008;36:28e30. 6. Sedivy P, Spacek M, El Samman K, et al. Endovascular treatment of infected aortic aneurysms. Eur J Vasc Endovasc Surg 2012;44:385e94. 7. Parameswaran V. Multiple mycotic aneurysms with a rare fungus, Aspergillus niger: a complex case report. J Vasc Nurs 2008;26:22e6. 8. Szendro G, Golcman L, Barki Y, et al. Evolution of multiple mycotic aneurysms. Eur J Vasc Endovasc Surg 1995;10:250e2. 9. Fisk M, Peck LF, Miyagi K, et al. Mycotic aneurysms: a case report, clinical review and novel imaging strategy. QJM 2012;105:181e8. 10. De Salazar AO, Izaguirre JL, Zubero Z. Surgical treatment of multiple mycotic aneurysms: in the ascending aorta, aortic arch, and descending aorta. Tex Heart Inst J 2003;30:225e8. 11. Hsu RB, Chang CI, Wu IH, et al. Selective medical treatment of infected aneurysms of the aorta in high-risk patients. J Vasc Surg 2009;49:66e70. 12. Semba CP, Sakai T, Slonim SM, et al. Mycotic aneurysms of the thoracic aorta: repair with use of endovascular stentgrafts. J Vasc Interv Radiol 1998;(1 pt 1):33e40. 13. Knosalla C, Weng Y, Yankah AC, et al. Using aortic allograft material to treat mycotic aneurysms of the thoracic aorta. Ann Thorac Surg 1996;61:1146e52. 14. Kieffer E, Gomes D, Chiche L, et al. Allograft replacement for infrarenal aortic graft infection: early and late results in 179 patients. J Vasc Surg 2004;39:1009e17. 15. Halpert B, DeBakey ME, Jordan GL, et al. The fate of homografts and prostheses of the human aorta. Surg Gynecol Obstet 1960;111:659e74. 16. Humphries AW, Hawk WA, De Wolfe VG, et al. Clinicopathologic observations on the fate of arterial freeze-dried homografts. Surgery 1959;45:59e71. 17. Knox WG, Miller RE. Long-term appraisal of aortic and arterial homografts implanted in years 1954-1957. Ann Surg 1970;172:1076e8. 18. Meade JW, Linton RR, Darling RC, et al. Arterial homografts: a long-term clinical follow-up. Arch Surg 1966;93:392e9. 19. Szilagyi DE, McDonald RT, Smith RF, et al. Biologic fate of human arterial homografts. Arch Surg 1957;75:506e29. 20. Szilagyi DE, Rodriguez FJ, Smith RF, et al. Late fate of arterial allografts: observations 6 to 15 years after implantation. Arch Surg 1970;101:721e33. 21. Brown KE, Heyer K, Rodriguez H, et al. Arterial reconstruction with cryopreserved human allografts in the setting of infection: a single centre experience with midterm followup. J Vasc Surg 2009;49:660e6.
Mycotic aneurysms are a relatively uncommon phenomenon that can pose a distinct treatment dilemma for the treating surgeon. It is rare to find Haemophilus influenzae as the causative organism, and it is even more rare to find multiple aneurysms in the same patient. We present the first case to our knowledge to be described in the literature of multiple mycotic aneurysms caused by H. influenzae. The treatment presenteddusing high-dose antibiotics and cryopreserved homograftsdhas worked well in this instance. This approach is supported in the literature.
Osler1 first used the term ‘‘mycotic aneurysm’’ in 1885 to describe a nonsyphilitic infection of the arterial wall. Today the term is used to describe any arterial aneurysm that has an infective cause. Mycotic aneurysms specifically infected with Haemophilus influenzae are documented as extremely rare.2 To date there have been 4 reported mycotic aneurysms infected with H. influenzae in the published literature. All were isolated instances and none of the cases noted multiple aneurysms in the same patient. We report a patient with multiple mycotic aneurysms infected with H. influenzae, and we believe that this case is the first of its kind.
CASE REPORT A 57-year-old man presented to his local hospital with a short history of general malaise, back pain, and a tender pulsatile lump in his right groin. Blood results on
Northern Vascular Centre, Freeman Hospital, High Heaton, Newcastle Upon Tyne, United Kingdom. Correspondence to: Alexander F. Nath, MBBS, MRCSEd, Northern Vascular Centre, Freeman Hospital, High Heaton, Newcastle upon Tyne, NE7 7DN, United Kingdom; E-mail: [email protected] Ann Vasc Surg 2014; 28: 741.e11–741.e13 http://dx.doi.org/10.1016/j.avsg.2013.07.016 Ó 2014 Elsevier Inc. All rights reserved. Manuscript received: December 11, 2012; manuscript accepted: July 15, 2013; published online: February 2, 2014.
admission revealed a hemoglobin (Hb) level of 12.1 g/ dL, a platelet count of 548 109 L, a white blood cell count (WBC) of 10.4 109 L, a C-reactive protein (CRP) level of 64 mg/L, and an erythrocyte sedimentation rate (ESR) of 38 mm/hr. A computed tomography angiography (CTA) scan of the aorta and lower limbs revealed the presence of a right common femoral artery aneurysm and saccular aneurysmal change of the distal descending thoracic aorta (Fig. 1). A provisional diagnosis of nonspecific inflammatory arteritis of the medium and large vessels was made, and the patient was prescribed high-dose steroids and methotrexate. A repeat CTA scan performed 20 days later at the same hospital revealed progression of the aneurysmal disease. The patient was then transferred to the regional vascular service. Additional review of the CTA scan confirmed a right common femoral artery aneurysm, a multilobulated saccular aneurysm of the distal thoracic aorta, and multiple small saccular aneurysms of the ascending aorta. The morphology of the aneurysms was strongly suspicious of a mycotic etiology, although there was no history of recent infection. An echocardiogram revealed no evidence of endocarditis, and a labeled white cell scan was inconclusive. It was noted that the aneurysmal section in the descending thoracic aorta had increased in size from 35 to 43 mm. A decision was made to undertake repair of the right femoral aneurysm in an attempt to establish a firm diagnosis. This was achieved by excision and in situ replacement with a rifampicin-soaked Dacron graft (DuPont, Kinston, NC)d the intent being to preserve his autologous vein for possible future aortic reconstruction. H. influenzae was grown from the aneurysm wall sent for culture, although the thrombus revealed a negative culture. Initially, the patient was started on intravenous amoxicillin 2 g, 4 times
741.e11
741.e12 Case reports
Annals of Vascular Surgery
Fig. 2. The second of the thoracic aneurysms repaired with a homograft.
Fig. 1. Computed tomography angiography scan revealing the 2 aneurysmal sections of the thoracic aorta. daily, after guidance of sensitivities, for a period of 2 weeks. This was then rationalized to an oral dose for an additional 4 weeks. The ascending aorta and distal thoracic aorta were subsequently repaired in separate procedures 16 and 21 days later, respectively, with excision of the diseased aorta and in situ replacement using cryopreserved homografts (Fig. 2). The culture results were negative from the excised specimens, which was unsurprising given the directed prolonged antibiotic treatment; however, the histologic results were consistent with mycotic aneurysm. The patient made a full recovery and was discharged 13 days later. He has been followed up with annual CTA scans, and at 28 months postsurgery has no signs of recurrent disease and no late aneurysmal dilatation (Fig. 3).
DISCUSSION H. influenzae in particular is an uncommon causative organism for the development of mitotic aneurysms. We have discovered 4 documented reports in the literature.2e5 Mycotic aneurysms themselves are said to be rare; multiple ones are exceptional.6 A recent article by Sedivy et al.6 noted no instances of H. influenzae infection in a case series of 32 patients over 15 years. Our literature search has revealed 2
Fig. 3. Computed tomography scan of reconstruction at 28 months showing no late aneurysmal dilatation or recurrence.
cases of multiple mycotic aneurysms specifically of the aorta,7,8 and neither had H. influenzae isolated. In the first, by Parameswaran,7 Aspergillus niger was found to be the causative organism. The second case, by Szendro et al.,8 was culture-negative at presentation despite heart valve vegetations, leukocytosis, and elevated ESR and CRP levels. In isolation, the medical treatment of mycotic aneurysms carries a very high mortality rate.9e11 Surgical treatment is frequently complicated by the development of recurrent infection. Prosthetic material will become a further focus of infection, leading to the inevitable consequence of rupture and death. In the infrarenal aorta, in situ repair or ligation and extra-anatomic bypass would be the mainstays of treatment. In the thoracic aorta, the options are less straightforward. As a temporizing measure, the aneurysmal section can have an endovascular stent placed before formal repair after antibiotic treatment. Semba et al.12 have described the use of endovascular stent grafts in the treatment of 3 patients with mycotic aneurysms of the thoracic aorta. The first case required a repeat procedure
Vol. 28, No. 3, April 2014
because of migration. The second was stented concurrently with open ruptured abdominal aortic mycotic aneurysm repair. The final case was a secondary procedure after interposition graft rupture. Notably, there were no complications from these procedures or any persistent bacteremia. Cadaveric homografts have been reported to be more resistant to infection.11,13 Knosalla et al.13 described treating 8 patients between 1988e1994 who had mycotic aneurysms of the thoracic aorta. These patients had either previously undergone cardiac transplant or had valvular endocarditis. Of these 8 patients, 6 recovered well and the bacterial infection was fully treated. One of the remaining 2 patients died from salmonella sepsis and the second required additional surgery. Traditionally, there has been some concern in using homografts for aortic replacement because of concerns of late graft deterioration, thrombosis, and calcification.14e20 Kieffer et al.14 have presented their series of using allografts in the replacement of infected infrarenal grafts. They used a mixture of fresh and cryopreserved allografts. They postulated that the initial poor results seen with allografts could be explained by the cryopreservation and thawing techniques at the time, which has improved in recent years. They also suggested that the source of the graft harvest will also play a part. It was noted that the graft segments that were especially prone to late complications had been harvested from the descending aorta. They postulated that this could be because of their lower number of mural elastic fibers. Brown et al.21 have also presented their series of using cryopreserved allografts and have not seen aneurysmal change as a particular problem; however, their follow-up has been midterm only. We have presented a case of multiple mycotic aneurysms with H. influenzae as a causative organism. The patient was previously fit and well with no chest symptoms or previous immunosuppression. We have found no other examples of this in the literature. Our approach to treatment is supported by the literature, and the staged manner of the procedures has, in our opinion, produced the best result for the patient with the least morbidity. We acknowledge the support and contribution to the management of this case from John H. Dark FRCS, Consultant Cardiothoracic Surgeon, Freeman Hospital, Newcastle upon Tyne, United Kingdom.
Case reports 741.e13
REFERENCES 1. Osler W. The Gulstonian Lectures, on Malignant Endocarditis. Br Med J 1885;1:467e70. 2. Takahashi Y, Tsutsumi Y, Monta O, et al. Mycotic aneurysm of the descending thoracic aorta caused by Haemophilus influenzae. J Card Surg 2010;25:218e20. 3. Degener JE, Voesten HG, Nap M, et al. Rupture of an aortic aneurysm caused by Haemophilus influenzae type B infection. Vasa 1993;22:247e50. 4. Adlakha A, Yale SH, Patel R, et al. Haemophilus influenzae serotype f: an unusual cause of a mycotic aneurysm in an adult. Mayo Clin Proc 1994;69:467e8. 5. Whitfield CG, Lonsdale RJ, Rahbour G, et al. Infective abdominal aortic aneurysm due to Haemophilus influenza identified via the polymerase chain reaction. Eur J Vasc Endovasc Surg 2008;36:28e30. 6. Sedivy P, Spacek M, El Samman K, et al. Endovascular treatment of infected aortic aneurysms. Eur J Vasc Endovasc Surg 2012;44:385e94. 7. Parameswaran V. Multiple mycotic aneurysms with a rare fungus, Aspergillus niger: a complex case report. J Vasc Nurs 2008;26:22e6. 8. Szendro G, Golcman L, Barki Y, et al. Evolution of multiple mycotic aneurysms. Eur J Vasc Endovasc Surg 1995;10:250e2. 9. Fisk M, Peck LF, Miyagi K, et al. Mycotic aneurysms: a case report, clinical review and novel imaging strategy. QJM 2012;105:181e8. 10. De Salazar AO, Izaguirre JL, Zubero Z. Surgical treatment of multiple mycotic aneurysms: in the ascending aorta, aortic arch, and descending aorta. Tex Heart Inst J 2003;30:225e8. 11. Hsu RB, Chang CI, Wu IH, et al. Selective medical treatment of infected aneurysms of the aorta in high-risk patients. J Vasc Surg 2009;49:66e70. 12. Semba CP, Sakai T, Slonim SM, et al. Mycotic aneurysms of the thoracic aorta: repair with use of endovascular stentgrafts. J Vasc Interv Radiol 1998;(1 pt 1):33e40. 13. Knosalla C, Weng Y, Yankah AC, et al. Using aortic allograft material to treat mycotic aneurysms of the thoracic aorta. Ann Thorac Surg 1996;61:1146e52. 14. Kieffer E, Gomes D, Chiche L, et al. Allograft replacement for infrarenal aortic graft infection: early and late results in 179 patients. J Vasc Surg 2004;39:1009e17. 15. Halpert B, DeBakey ME, Jordan GL, et al. The fate of homografts and prostheses of the human aorta. Surg Gynecol Obstet 1960;111:659e74. 16. Humphries AW, Hawk WA, De Wolfe VG, et al. Clinicopathologic observations on the fate of arterial freeze-dried homografts. Surgery 1959;45:59e71. 17. Knox WG, Miller RE. Long-term appraisal of aortic and arterial homografts implanted in years 1954-1957. Ann Surg 1970;172:1076e8. 18. Meade JW, Linton RR, Darling RC, et al. Arterial homografts: a long-term clinical follow-up. Arch Surg 1966;93:392e9. 19. Szilagyi DE, McDonald RT, Smith RF, et al. Biologic fate of human arterial homografts. Arch Surg 1957;75:506e29. 20. Szilagyi DE, Rodriguez FJ, Smith RF, et al. Late fate of arterial allografts: observations 6 to 15 years after implantation. Arch Surg 1970;101:721e33. 21. Brown KE, Heyer K, Rodriguez H, et al. Arterial reconstruction with cryopreserved human allografts in the setting of infection: a single centre experience with midterm followup. J Vasc Surg 2009;49:660e6.
