Mycotic Arteriovenous Fistula in a Two-Year-Old Child

By DOUGLAS COHEN AND L. P. RoY

F ISTULAS between the aorta or its major branches and the inferior vena eava are rare. Polak and Skarbek 1 (1963) could not find any examples

recorded in Great Britain in the previous 40 years, while there were only 13 reported cases in other countries. Gresham and Johnson TM (1959) were able to trace 27 previous cases since 1831. All these cases were either atheromatous in origin or were traumatic. Beatl et aF reported 4 cases of fistula between the aorta and the inferior vena cava which were successfully repaired, and listed 7 other cases of survival following operation for spontaneous aorto-inferior vena caval fistulas, all of them secondary to rupture of arteriosclerotic aneu- rysms of the abdominal aorta into the inferior vena eava.

Blood stream infections can result in mycotic aneurysms and such aneurysms may rupture into an adjacent vein resulting in an arteriovenous fistula. Cham- berlain s reviewed 19 eases of mycotic aneurysm and repor ted 3 of his own, all in adults, of which 13 were associated with bacterial endoearditis and the remainder with pneumonia, septicemia, septic arthritis, and infective trauma. These aneurysms were small; none ruptured into a vein producing an arterio- venous fistula. The commonest organisms were streptococci, gonococci and typhoid. Eight of the aneurysms reported were in the aorta.

No case similar to the present one appears to have been previously recorded.

CASE REPOI~T

K. H., a 21~ year old female child, was admit ted to the Royal Alexandra Hospital for Children on January 3, 1966, with a 3 day history of fever, associated with vomiting and diarrhea. Twenty-four hours prior to admission she had developed an erythematous rash and it was noted that the middle 2 fingers of the left hand were swollen.

She looked moderately i l l and her temperature was 39.4~ There were no cardiac murmurs and her blood pressure was 100/50. Hemoglobin was 14.5 Gm. and a stained smear was reported as normal.

A presumptive diagnosis of septicemic osteomyelitis was made, a blood culture was taken and a lumbar puncture performed. Staphylococcus aureus was grown from the blood culture; the cerebrospinal fluid was normal to examination and culture was sterile. X-ray of the left hand showed no bony abnormality, bu t 48 hours after admission there was pus in the pulp space of the middle finger, which was incised and drained. Staphylococcus aureus was cultured. After 3 days the rash present on admission had faded, but the child remained febrile. Blood taken on January 14 for determination of antibacterial activity again grew staphylococcus aureus in spite of being bactericidal at a dilution of 1 in 16 to the organism originally cultured.

Hemoglobin level on January 15 had fallen to 7.3 Gm. and a blood transfusion was given, She became afebrile on January 22 and remained so thereafter.

On admission antibiotic therapy was commenced wi th intravenous sulphadimidine,

DOUGLAS COHEN, M.S., F.R.A.C.S.: Surgeon, Adolph Basser Institute of Cardiology, Royal Alexandra Hospital for Children, Camperdown, Sydney, Australia. L. P. Roy, M. B. : Medical Registrar, Royal Alexandra Hospital [or Children, Camperdown, Sydney, Aus- tralia.

From the Alexandra Hospital for Children, Camperdown, Sydney, Australia.

343

JOURNAL OF PEDIATRIC SURGERY~ VOL. 2, No. 4 (AUGUST), 1967

344 C O H E N A N D ROY

Fig. 1

ehloramphenicol and penicillin. Following isolation of staphyloccus aureus, sulphadimidine was suspended and 24 hours later penicillin was suspended because the organism proved insensitive to this drug, and methicillin was added. On January 14 probenecid was added. On January 19, because of a further pyrexial episode, fusidic acid was commenced and from January 22 she remained afebrile.

On the afternoon of January 20 the child suddenly became tachypneic and was found to have developed congestive cardiac failure. For the first t ime a continuous murmur was heard over the abdomen. She was given digitalis. The next day the murmur was louder, was best heard over the lower abdomen, and radiated to both femoral areas, being louder on the r ight side. The blood pressure was 100/0 in bo th arms and in the right leg, and 80 /0 in the left leg.

It was thought tha t an arteriovenous fistula had developed between two large intra- abdominal vessels. Her congestive cardiac failure slowly responded to t reatment with digitalis and injections of mercurial diuretic.

We thought it might be risky to cannulate the aorta by either femoral vessel because of the danger of perforation and therefore used the brachial route. In order to demonstrate the AV fistula a Seldinger catheter was introduced through the left brachial artery and passed into the upper abdominal aorta. An aortogram showed an arteriovenous fistula between the right common lilac artery and the inferior vena cava (Fig. 1 ).

The child had once again become anemic and she was again transfused and thereafter maintained a normal hemoglobin. Since her cardiac failure was slowly but progressively coming under control, and the abdominal lesion appeared to be stable, it was decided to defer surgery for two to three months, in order to allow the lesion an opportunity to become more fibrotic.

MYCOTIC AllTERIOVENOUS FISTULA. 345

An x-ray of the left hand on February 10 revealed osteomyelitis of the middle phalanx of the left middle finger. The finger had continued to drain after a second incision and remained swollen and indurated, but slowly resolved. Methieillin was replaeed by cloxaeillin on February 14 and she was continued on eloxaeillin, probeneeid and fnsidic acid until March 9, when antibiotic therapy was suspended.

On April 5, 1966, the area of the fistula was approached through a right transverse extraperitoneal abdominal ineision. There was a dense fibrotic reaction around the area of the aortic and eaval bifurcations. Slings were passed around the aorta and inferior vena cava above and around both iliac arteries and veins below the site of the arteriovenous communication. The anterior aspects of the aortic and eaval bifurcations, together with the intervening arteriovenous fistula, were then disseeted as clear as possible, following which the aorta, inferior cava and both common iliac vessels were clamped and the anterior aspect of the inferior vena eava was opened widely. The fistula from the posterior wall of the cava into the r ight eommon iliac artery could be clearly seen and it was possible to divide the fistula leaving a hole in the posterior wall of the eava and in the anterolateral aspect of the right common iliac artery. These holes, together with the opening into the anterior wall of the eava were then repaired with 5-0 silk.

The postoperative course was uneventful and the color and temperature of the right leg remained normal at all times. However, the right femoral pulse was decreased in ampli tude and has remained so. Digoxin was suspended and the child was discharged well on April 20. One month later, she was asymptomatic and the only abnormal finding was the poor volume right femoral pulse together with impalpable distal pulses in the r ight leg.

DISCUSSION

The etiology of the arteriovenous fistula in this case was bacterial. It seems probable that the septicemia originated in an osteomyelitic focus in the middle phalanx of the left middle finger.

It is highly probable that the vessel wall was attacked outside by infection of the vasa vasorum, or possibly by being involved in an inflammatory process originating between the aorta and the inferior vena cava. An abscess in this situation could have resulted in inflammatory softening and perforation of the adjacent walls of the two vessels with the formation of an arterio-venous fistula.

Goodhart 4 (1877) was the first to postulate that a bacterial process could be the cause of an aneurysm, while Eppinger ~ (1887) showed that an aneurysm could form as a result of an abscess of the vessel wall by an infected embolus. He pointed out that this was likely to occur either in bacterial endocarditis or in other septic conditions in which the embolus lodged at a bifurcation or sudden narrowing of the vessel.

The first case of mycotic arteriovenous fistula was reported by Simmonds 6 in 1904. This patient had rheumatic heart disease and had developed strep- tococcal puerperal septicemia. The fistula was between the aorta and the right common iliac vein. In 1921, Floyd 7 reported a case where the communication was between the femoral artery and vein, and was able to find only two others in the literature, that of Simmonds 6 and one described by Libman 8 also be- tween the femoral artery and vein arising at the origin of the profunda femoris. Staphylococcus aureus was cultured from the lesion. All three patients died. A review of mycotic aneurysms by Barker 9 makes no mention of this complication, although rupture of mycotic aneurysms is reported and their surgical management is discussed.

346 COHEN AND BOY

SUMMARY

An arteriovenous fistula in a 2 year old girl which developed following staphylococcal septicemia was treated successfully surgically. This appears to be the first reported survival from this condition and the first reported case in childhood.

S U M M A R I O I N I N T E R L I N G U A

Es reportate un caso in que un fistula se disveloppava inter le dextere extero-iliac e le vena cave inferior. Le patiente esseva un infante de duo annos de etate. Le fistula sequeva osteomyelitis septicemic. Post dominio del congestive insufl~cientia cardiac que esseva etiam presente, le fistula esseva dividite, e le vasos esseva reparate separatemente. In iste caso le fistula habeva sin dubita resultate ab infection. Le presente caso pare esser le prime de su genere unquam reportate in un patiente pediatric.

A C K N O W L E D G M E N T S

We are grateful to Professor T. Stapleton for permission to publish this case, to Dr. Douglas Stuckey and Dr. Denby Bowdler for their help, criticism and assistance in man- agement, and to Dr. John Celermajer, who carried out the angiographic investigation.

R E F E R E N C E S

1. Polak, A., and Skarbek, A.: Spontane- ous rupture of aortic aneurysm into inferior veua cava. Brit. Med. J. i : 1717, 1963.

la. Gresham, G.A., and Johnson, H . R . M . : Rupture of aortic aneurysms into the gut. Brit. Heart J. 21:518 (1959).

2. Beall, A. C., Cooley, D. A., Morris, G. C., and DeBakey, M. E.: Perfora- lion of arteriosclerotic aneurysms into inferior vena cava. Archives of Sur- gery 86:809-818, 1963.

3. Chamberlain, E. N.: Bacterial aneurysm. Brit. Heart J. 5:121, 1943.

4. Goodhart, J. F. : Cases of aneorysm from embolism. Trans. Path. Soe. Lond. 28: 106, 1877.

5. Eppinger, H.: Pathogenesis (Histogen- esis und Aetiologie) der Aneurysmen einschliesslich des Aneurysma equi verminosum. Arch. Klin. Chir. 35, 1887.

6. Simmonds: Mykotisches Aneurysma der Aorta. Muench Med. Wschr. 51:627, 1904.

7. Floyd, R.: A large mycotic embolic arteriovenous aneurysm of the fe- moral vessels, Surg. Gynec. Obstet. 33:560, 1921.

8. Libman, E.: Embolic Aneurysms. Mt. Sinai Hosp. Rep. 5:481, 1905.

9. Barker, W. F.: Mycotic aneurysms. Ann. Surg. 139:84, 1954.