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CASE REPORT
Year : 2019  |  Volume : 8  |  Issue : 4  |  Page : 276-280

An unusual presentation of infrarenal aortoiliac occlusion with metastatic gas gangrene


Department of Radiodiagnosis and Imageology, Kurnool Medical College and Government General Hospital, Andhra Pradesh, India

Date of Submission15-Jan-2016
Date of Acceptance13-Aug-2019
Date of Web Publication16-Dec-2019

Correspondence Address:
Dr. J Abdul Gafoor
Department of Radiodiagnosis, Kurnool Medical College, Kurnool - 518 002, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JDRNTRUHS.JDRNTRUHS_8_19

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  Abstract 


Gas gangrene is a term reserved for fulminant soft tissue infections caused by Clostridium species. The posttraumatic form of gas gangrene is caused by Clostridium perfringens. Nontraumatic, or spontaneous, gas gangrene is even more rare and is usually caused by Clostridium septicum in two-thirds of cases and by C. perfringens in one-third of cases; the underlying malignancy is often present. It also usually affects patients with some degree of underlying immunocompromised or vascular insufficiency. We report a case of a 30-year-old man with no history of prior trauma presented with pain, paresis of both lower limbs, and developed discolouration of both feet clinically diagnosed to be Leriche's syndrome with dry gangrene. Computed tomography angiography showed infrarenal aortoiliac occlusion and rapidly progressive gas gangrene involving even bladder wall and bone marrow, probably caused by Clostridium septicum. Although several case reports discuss the setting of bowel malignancy, our case illustrates acute onset of aortic iliac occlusion resulting in mesenteric infarction, which is the probable source for the infection.

Keywords: Aortoiliac occlusion, gas gangrene, mesenteric infarction


How to cite this article:
Gafoor J A, Reddy O J, Harinath D, Bhavana N. An unusual presentation of infrarenal aortoiliac occlusion with metastatic gas gangrene. J NTR Univ Health Sci 2019;8:276-80

How to cite this URL:
Gafoor J A, Reddy O J, Harinath D, Bhavana N. An unusual presentation of infrarenal aortoiliac occlusion with metastatic gas gangrene. J NTR Univ Health Sci [serial online] 2019 [cited 2020 Feb 27];8:276-80. Available from: http://www.jdrntruhs.org/text.asp?2019/8/4/276/273134




  Introduction Top


Clostridial myonecrosis, or gas gangrene, is a rare but often rapidly fatal infection.

It often occurs as a complication of recent trauma or surgery. Even rarer is nontraumatic, or spontaneous, gas gangrene. Acute aortoiliac occlusion is an unusual but catastrophic condition causing acute limb ischemia and is associated with early and high rates of morbidity and mortality. It is often caused by either embolism or thrombosis of the abdominal aorta and its large terminal branches. The case reported here illustrates spontaneous myonecrosis with infrarenal aortoiliac occlusion with lack of obvious predisposing event.


  Case History Top


A 30-year-old male patient presented to the emergency department with acute onset of pain and weakness of both lower limbs and loss of bladder control. He is an occasional smoker and an alcoholic. He is not a known diabetic and hypertensive. He met with an accident 2 years back with closed fracture of the left tibia and fibula and was treated conservatively. There was no history of recent trauma, head injury, or seizures. On general physical examination, the patient was moderately built and nourished, afebrile with blood pressure 140/90mmHg, respiratory rate 16/min, radial pulse rate 78/min, regular, normal volume, while absent pulses were felt in the both lower limbs along with dry shrunken and discolored feet. Cardiovascular and respiratory system examination was noncontributory. There was no organomegaly or palpable masses felt in the abdomen. Examination of the skeletal system including the neck and spine was normal. Neurological examination revealed a conscious, alert patient responding to verbal commands. Ocular movements were normal. On examination of the motor system, muscle power was decreased in both lower limbs (1–2/5) along with sensory loss. Superficial and deep tendon reflexes were elicitable normally, while the plantar response was mute bilaterally. There were no meningeal signs or pyramidal signs. Routine complete blood picture values were normal. It was clinically diagnosed to be Leriche's syndrome with dry gangrene. The patient was referred for Doppler study showing undetectable flow in the abdominal aorta including the aortic bifurcation, common iliac arteries, bilateral external iliac arteries, femoral, and popliteal arteries and was advised computed tomography angiography (CTA). Immediate CTA revealed occlusion with thrombus extending from infrarenal abdominal aorta, 6.6 cm proximal to bifurcation of aorta with absent flow distal to it [Figure 1]. Absent flow in inferior mesenteric artery with delayed enhancement of bowel wall was noted in distal colon and nonenhancing rectal loop [Figure 2]. Air loculi were noted in the lumen of inferior venacava, bilateral external iliac veins, femoral veins, popliteal veins, and deep superficial veins of the right leg [Figure 3]. Hypodense thrombus was noted in the left renal vein. Extensive intermuscular, intramuscular and fascial air loculi were noted in the bilateral gluteal region, perineal region, both hip joint cavities, the entire right lower limb up to midtarsal region, and proximal left lower limb [Figure 4]. Air loculi were noted inside the pelvic bones including sacrum and in the right femoral neck [Figure 5]. Air inside the bladder wall (gangrenous cystitis), perirectal, and pelvis soft tissues was also noted [Figure 6]. Before further investigations could be carried out, the patient succumbed to cardiorespiratory arrest.
Figure 1: (a) Sagittal (precontrast), (b) (postcontrast), reformatted images showing hypodense thrombus (arrow) just below the origin of renal arteries and (c) axial CT angiogram image showing the hypodense thrombus (arrow). (d) Volume rendering image showing the aortoiliac occlusion in infrarenal aorta

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Figure 2: (a) Axial and (b) coronal images of CT angiogram showing delayed enhancement of distal colon (arrow) with air loculi in mesenteric vessels and in pelvic muscle tissues

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Figure 3: (a), (b) Coronal images of CT angiogram study showing air loculi in inferior vena cava (IVC) (arrow) and in popliteal vessels of right lower limb (arrow).(c) Axial image showing hypodense thrombus in left renal vein

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Figure 4: (a) Coronal and (b) axial images showing extensive intermuscular, intramuscular, and fascial air loculi in perineal, pelvic, and lower limbs (predominantly in right more than left)

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Figure 5: (a), (b) Coronal reformatted images in bone window showing air loculi in pelvic bones (arrow) and in left hip joint space (bone marrow necrosis)

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Figure 6: (a) Axial, (b) coronal, and (c) sagittal images showing air loculi (arrow) in wall of bladder (gangrenous cystitis)

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  Discussion Top


Acute aortic occlusion occurs as a sudden event without previous history of claudication. Symptoms are variable depending on completeness of obstruction, but the most common symptoms are pain, pallor, pulselessness, paresthesia, and paralysis.[1] In preexisting atherosclerotic disease, the degree and severity of symptoms may be lessened due to development of collateral circulation. In the absence of collateral circulation, the patient experiences sudden onset of severe low back, buttocks, and lower extremity pain. This is accompanied by a flaccid paralysis of the lower extremities if spinal cord infarction occurs, or by the gradual onset of lower extremity weakness if ischemia of peripheral nerves or the cauda equina occurs. Most of the ventral two-thirds of the spinal cord includes the pyramidal tracts supplied by lumbar artery from aorta; thus, with occlusion of this system, a flaccid paralysis occurs with initially absent deep tendon reflexes that become hyperactive over time.

Bowel and bladder incontinence are also characteristic. The skin is cool and pale distal to the occlusion. Skin may withstand severe ischemia for as long as 24 h; however, muscle sustains permanent damage after 8–12 h.

Etiologies of acute aortic occlusion

There are several causes of acute occlusion, including saddle embolism, acute thrombosis with or without preexisting aortoiliac atherosclerotic disease, thrombosis of an abdominal aortic aneurysm, aortic dissection, and posttraumatic abdominal aortic occlusion. Acute thrombosis resulting from a hypercoagulable state or vasculitis has also been implicated. Acute thrombosis should not be confused with Leriche's syndrome, a slowly progressing disease due to atherosclerotic lesions of the aortic wall that reduces the diameter of the lumen until it reaches a critical stenosis of 80% of the cross-sectional area. Isolated aortoiliac occlusive disease primarily occurs in younger patients, more commonly in females, and has a higher incidence of smoking and hypercholesterolemia as associated risk factors.[2] In the younger patients, the possibility of a hypercoagulable state should be considered. Hypercoagulable states play an increasing role in aortic occlusive disease, especially when they are superimposed on preexisting arteritis or atherosclerotic disease. The primary causes of hypercoagulability include antithrombin III deficiency or a lower functional level of the inhibitor, protein C and S deficiency, prostacyclin deficiency, disorders of the fibrinolytic system including low or abnormal plasminogen states and deficiency of plasminogen activator, dysfibrinogenemia, and the presence of lupus anticoagulant factor. The secondary causes include malignancy, pregnancy, heparin-induced thrombocytopenia with arterial thrombosis, homocystinuria, nephrotic syndrome, diabetes, hyperlipidemia, use of oral contraceptives, and vasculitis.[2] Rarely, patients who present with complete acute occlusion of aorta can have symptoms related to intestinal and renal ischemia. Acute mesenteric ischemia sets an emergency and requires early diagnosis and intervention. With the progression of bowel ischemia to gangrene, a series of events will culminate to death.[3] In the setting of bowel mucosal injury or compromise by tumor, hematologic seeding with the organism can occur, leading to distant sites of myonecrosis. The condition is most probably due to infection from Clostridium species.

As in our case, in the absence of localizing physical examination findings of gas gangrene, acute occlusion of abdominal aorta resulting in bowel ischemia resulted in rapidly progressive metastatic gas gangrene along with development of gangrenous cystitis and bone marrow necrosis, and the exact sequence of events remains unclear.

Gas gangrene is a rapidly spreading infection. It can advance as fast as 2 cm/h. The incubation period varies from 6 h to 2 days. Spontaneous clostridial mynecrosis occurs without trauma, is most often caused by Clostridium septicum, and has been described in specific clinical settings such as neutropenia, colonic malignancy, inflammatory bowel disease, bowel ischemia, and haemolytic uremic syndrome.

Metastatic gangrene is extremely devastating, with overall mortality rates ranging from 67% to 100%, with death occurring in the first 24–48 h.

Ultrasound remains the diagnostic method of choice, with accuracy approaching 100% and bedside availability for the unstable patient. Multidetector computed tomography (MDCT) with shorter acquisition time, noninvasive, and three-dimensional processing capabilities of CTA has become the most frequently used imaging moadality. Magnetic resonance angiography (MRA) is used for the evaluation of patients with aortoiliac occlusive disease because it can visualize the entire arterial tree without the use of arterial puncture or standard ionic contrast agents. Digital subtraction angiography, which is the gold standard for diagnosis of allarterial occlusive diseases, especially if anatomic questions remain, has largely been replaced by CTA and MRA and is increasingly been selectively performed for endovascular intervention.

Management of aortoiliac occlusive disease presents a unique challenge due to the complex interplay of factors that must be considered, including the underlying pathology, anatomic defect, presence of distal disease, degree of ischemia, comorbid conditions, functional status, ambulation potential, and suitability of anatomy for successful revascularization. Management of acute aortic occlusion in the emergency department consists of prompt recognition, provision of supportive care, immediate surgical consultation, and initiation of a limited diagnostic workup. Since outcome is time-dependent, early recognition and referral are essential elements of management.

The most difficult aspect of management relates to the diagnostic evaluation. A bedside ultrasound is the best first step; its availability, ease of performance, and sensitivity make it ideally suited for this situation. If ultrasound is not available or is technically inadequate, a CT scan should be considered. Nevertheless, urgently needed surgery should not be deferred until angiography is performed. Ultimately, the treatment of acute aortic occlusion is surgery. If acute occlusion from embolism or thrombosis is suspected, high-dose heparin should be initiated in the emergency department and the patient should be taken for emergent embolectomy. Treatment for gas gangrene requires prompt antibiotic therapy and aggressive surgical debridement. In all cases, wide debridement of necrotic tissue should be performed immediately. Myonecrosis in a limb often requires amputation. In some institutions, hyperbaric oxygen therapy has been successfully used to treat clostridial myonecrosis (CM).[4] However, the rarity of immediate availability of hyperbaric chamber facilities limits use of this modality.


  Conclusion Top


The combination of absent femoral pulses and decreased motor or sensory function in the lower extremities demands emergency treatment. In the absence of a previous history of peripheral vascular disease, the patient can be taken directly to the operating room. Angiography can be done in those patients with a history of peripheral vascular disease, but obviously only if the clinical state seems acceptable to warrant a delay in surgery. Life-long anticoagulation is mandatory for all patients.[5] Because of the rarity of this disorder, confusion may exist concerning diagnosis and management. Even with proper management, mortality remains high. A decrease in mortality and morbidity has been seen with the use of anticoagulation and embolectomy.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to b'e reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Sen I, Stephen E, Agarwal S. Clinical profile of aortoiliac occlusive disease and outcomes of aortobifemoral bypass in India. J Vasc Surg 2013;7 (2 Suppl):20S-5S.  Back to cited text no. 1
    
2.
Frost S, Jorden RC. FACEP acute abdominal aortic occlusion. J Emerg Med 1992;10:139-45.  Back to cited text no. 2
    
3.
Dossa CD, Shepard AD, Reddy DJ, Jones CM, Elliott JP, Smith RF, et al. Acute aortic occlusion a 40-year experience. Arch Surg 1994;129:603-8.  Back to cited text no. 3
    
4.
Abella BS, Kuchinic P, Hiraoka T, Howes DS. Atraumatic clostridial myonecrosis: Case report and literature review. J Emerg Med 2003;24:401-5.  Back to cited text no. 4
    
5.
Surowiec SM, Isiklar H, Sreeram S, Weiss VJ, Lumsden AB. Acute occlusion of the abdominal aorta. Am J Surg 1998;176:193-7.  Back to cited text no. 5
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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