- Email: [email protected]
Evidence for nonoperative management of acute limb ischemia in infants
From the Society for Vascular Surgery
Evidence for nonoperative management of acute limb ischemia in infants Jesus M. Matos, MD,a Andres Fajardo, MD,a Michael C. Dalsing, MD,a Raghu Motaganahalli, MD,a George A. Akingba, MD, PhD,a and Michael P. Murphy, MD,a,b Indianapolis, Ind Acute limb ischemia (ALI) in infants is a catastrophic event. We performed a query of our database to determine those with ALI. Twelve patients were identified. The most frequent presentation was cyanotic limbs. Eleven patients were treated nonoperatively with anticoagulation. One patient was treated surgically with Fogarty balloon thrombectomy. There were three deaths all due to associated comorbidities. All had viable limbs on follow-up examination. There were three complications in the patients managed conservatively. Our recommendation for infants presenting with ALI is conservative observation with anticoagulation and intervention only for cases with tissue loss. ( J Vasc Surg 2012;55: 1156-9.)
Acute limb ischemia (ALI) is a catastrophic event that portends a significant probability of limb loss. ALI in the infant population (ⱕ1 year old) is an exceedingly rare clinical event, and consequently, consensus management guidelines do not exist.1-2 Over the past decade, there have been several successful reports of limb salvage in children over a wide range of ages with anticoagulation and/or thrombolytic therapy but no series focusing specifically on the infant population with ALI.3-6 In this report, we provide a review of our experience in the management of ALI in the infant population, demonstrating efficacy in limb salvage with conservative management consisting of anticoagulation and close monitoring of limb viability. METHODS The Indiana University School of Medicine Vascular Surgery Section maintains a database of all vascular interventions and consultations performed by our staff at the Clarian Hospital System (Methodist, University, and Riley). The protocol of this study was approved by institutional review board review. Query of this database for all patients with acute limb ischemia in infants (defined as less than 1 year of age) from 2004 to 2010 was conducted. Data collected and analyzed included age, gender, medical comorbidities, affected extremity, mechanism of occlusion, presenting symptoms, diagnostic studies, treatment, and follow-up data. From the Department of Surgery, Indiana University School of Medicine,a and the Vascular and Cardiac Center for Adult Stem Cell Therapy.b Competition of interest: none. Presented at the 2011 Vascular Annual Meeting of the Society for Vascular Surgery, Chicago, Ill, June 16-18, 2011. Reprint requests: Michael Murphy, MD, Department of Surgery, Indiana University School of Medicine, 1801 N. Senate Blvd., MPC2, #3500, Indianapolis, IN 46202-1228 (e-mail: [email protected]). The editors and reviewers of this article have no relevant financial relationships to disclose per the JVS policy that requires reviewers to decline review of any manuscript for which they may have a competition of interest. 0741-5214/$36.00 Copyright © 2012 by the Society for Vascular Surgery. doi:10.1016/j.jvs.2011.09.092
1156
RESULTS Between November 21, 2004 and April 12, 2010 there were 12 patients identified with ALI7 (Fig 1). Injuries were more common in male patients (n ⫽ 9; 75%) with an average age of 3 months (range, 10 days-8 months). At presentation, seven (59%) patients had absent pulses by examination, and nine (75%) had limb cyanosis (Fig 2). A color-flow duplex ultrasound (DUS) was initially obtained on all patients that provided sufficient information to confirm the diagnosis, mitigating the need for additional imaging studies. Comorbidities included congenital cardiac disease (n ⫽ 5; 42%), pulmonary failure requiring mechanical ventilation (n ⫽ 4; 33%), prematurity (n ⫽ 1; 8%), sepsis (n ⫽ 1; 8%), and intermittent methemoglobinemia (n ⫽ 1; 8%). Intra-arterial catheterizations for diagnostic or monitoring purposes were the cause of ALI in 11 (92%) patients. Vessels of the lower extremity were more commonly injured (n ⫽ 9; 75%), and arteries that were injured were the common femoral artery (n ⫽ 7; 58%), superficial femoral artery (n ⫽ 2; 17%), radial artery (n ⫽ 2; 17%), and brachial artery (n ⫽ 1; 8%). Six of the patients had dopamine running during evaluation of the ischemic event. Twelve patients were anticoagulated with unfractionated heparin (n ⫽ 10) or subcutaneous (SQ) enoxaparin.2 Unfractionated heparin was administered as an initial bolus of 100 units/kg followed by continuous infusion of 20 units/ kg/hr to achieve an activated partial thromboplastin time (APTT) of 2 to 2.5 times normal. DUS was performed serially during the critical period of initiating anticoagulation. All patients were transitioned from heparin to SQ enoxaparin. Patients continued for 3 to 4 weeks with follow-up DUS. There was one failure of anticoagulation with heparin manifesting as ulceration and gangrene 48 hours after initiation of therapy. The patient underwent common femoral artery thrombectomy with a #3 Fogarty balloon catheter and four compartment fasciotomies with restoration of distal pulses. There were two deaths (18%) in the anticoagulation group during treatment for ALI. One patient died 2 days
JOURNAL OF VASCULAR SURGERY Volume 55, Number 4
Fig 1. Arterial duplex ultrasound of 6-month-old infant at time of presentation. Duplex demonstrates a relative absence of flow in the common femoral artery with monophasic waveforms and significantly reduced velocities.
Fig 2. Infant with cyanotic limb at time of presentation.
and the second patient died 9 days after vascular surgery evaluation due to heart failure. Out of the remaining nine patients, we had follow-up data on seven. On follow-up visits, every patient had repeat DUS. Three patients experienced complications felt to be related to limb ischemia: One was found to have a size discrepancy of 1 cm of the foot on the fully functional index limb, a second patient required skin debridement and grafting due to skin necrosis, and the third patient lost a fingernail. One patient had a follow-up DUS 24 days after treatment with enoxaparin that documented persistent occlusion of the common femoral artery with notable development of a collateral branch perfusing the superficial femoral artery (Fig 3). The one patient that was managed with surgical thrombectomy died 30 days after surgery from septic shock unrelated to the surgery or complications of limb ischemia. DISCUSSION Infant vascular injuries are rare and account for ⬍1% of patients with acute limb ischemia seen by vascular surgeons.8 Iatrogenic vascular injuries are more common in the infant population, while older children and adolescent
Matos et al 1157
Fig 3. Arterial duplex ultrasound of a month-old infant 24 days after treatment with anticoagulation, showing occlusion with lack of flow. Blue represents collateral flow formation with green arrow representing refeeding distal flow.
will experience penetrating injuries.9-11 Common causes for infant vascular injuries are cardiac catheterizations and arterial blood pressure monitoring devices.11 “Soft signs” of vascular injury are difficult to evaluate on this population age due to lack of verbalization of pain and diminished or absent sensation.8 In the infant population, active bleeding is the absolute indication for operation.12 There is limited literature when it comes to management of infants with ALI. Surgical management of these injuries represents a technical challenge; not only the small vessel size but also the problem of arterial spasms can cause difficulties. Our institution previously published how open surgical repair in older children with blunt vascular injuries allowed limb salvage in the absence of diagnostic delay.12 There are multiple series that report surgical intervention of pediatric patients1,12-15 and infants2,16,17 with ALI, but according to Lin et al15 operative results in children younger than 2 years leads to less satisfactory outcomes when compared with older children.5,9 Lazarides et al conducted a review of the literature describing outcomes of surgically managed patients in children ⱕ2.5 years of age and found only 48% of patients were able to regain palpable pulses, and limb discrepancy was detected in as many as 15% of patients.3 In terms of a more conservative approach, there are several management guidelines that have been proposed.2,3,18,19 One includes the use of thrombolytic agents as the first-choice therapy for 48 hours followed by microsurgery if no improvement was seen.2 Lazarides et al recommended only operating on infants with definite threat of limb loss and doing systemic heparinization or thrombolytic therapy, knowing that limb length discrepancies will be inevitable.3 The recommended conservative method is the administration of heparin as an initial bolus of 75 to 100 units/kilogram with an infusion depending on age: for the ⬍2 month old, 28 units/kilogram/hour, while older infants require only 20 units/kilogram/hour.8,20 Other
1158 Matos et al
groups recommend the use of thrombolytic agents in a selective group of patients who do not respond to systemic heparin anticoagulation.19,20 Criticism of conservative management includes the possibility of developing compartment syndrome and limb length discrepancies.3,9,11 In the infant population, supportive care and the use of systemic anticoagulation with heparin is the best management for most patients. Limb viability was 100% in this series of infants with ALI managed nonoperatively, and we propose that favorable remodeling in parallel collateral vessels occurs due to changes in flow dynamics with acute occlusion. Our recommendation for infants presenting with ALI is physical examination followed by DUS and systemic anticoagulation with heparin and transitioned to SQ lovenox. Patients should undergo serial examinations to assess changes in perfusion of the index limb and a repeat DUS in 3 to 4 weeks. We further recommend that if adequate perfusion of the limb is established by examination and DUS, then anticoagulation should be stopped. During the observational period, it is important for the vascular surgeon to establish expectations with the family. It is important to warn the family that the child could suffer limb discrepancies. If DUS depicts restoration of flow with collateral formation, anticoagulation can be stopped. Surgical intervention is indicated in patients with tissue loss, active bleeding, or lack of response to systemic anticoagulation. REFERENCES 1. Whitehouse WM, Coran AG, Stanley JC, Kuhns LR, Weintraub WH, Fry WJ. Pediatric vascular trauma. Manifestations, management, and sequelae of extremity arterial injury in patients undergoing surgical treatment. Arch Surg 1976;111:1269-75. 2. Gamba P, Tchaprassian Z, Verlato F, Verlato G, Orzali A, Zanon GF. Iatrogenic vascular lesions in extremely low birth weight and low birth weight neonates. J Vasc Surg 1997;26:643-6. 3. Lazarides MK, Georgiadis GS, Papas TT, Gardikis S, Maltezos C. Operative and nonoperative management of children aged 13 years or younger with arterial trauma of the extremities. J Vasc Surg 2006;43: 72-6; Discussion:6. 4. Kothari SS, Varma S, Wasir HS. Thrombolytic therapy in infants and children. Am Heart J 1994;127:651-7.
JOURNAL OF VASCULAR SURGERY April 2012
5. Perry MO. Iatrogenic injuries of arteries in infants. Surg Gynecol Obstet 1983;157:415-8. 6. Dillon PW, Fox PS, Berg CJ, Cardella JF, Krummel TM. Recombinant tissue plasminogen activator for neonatal and pediatric vascular thrombolytic therapy. J Pediatr Surg 1993;28:1264-8; Discussion:8-9. 7. Norgren L, Hiatt WR, Dormandy JA, Nehler MR, Harris KA, Fowkes FG, et al. Inter-Society Consensus for the Management of peripheral Arterial Disease (TASC II). J Vasc Surg 2007;45 Suppl S:S5-67. 8. Dalsing MC, Cikrit DF, Sawchuk AS. Open surgical repair of children less than 13 years old with lower extremity vascular injury. J Vasc Surg 2005.41 983-7. 9. Smith C, Green RM. Pediatric vascular injuries. Surgery 1981;90: 20-31. 10. Navarre JR, Cardillo PJ, Gorman JF, Clark PM, Martinez BD. Vascular trauma in children and adolescents. Am J Surg 1982;143:229-31. 11. Flanigan DP, Keifer TJ, Schuler JJ, Ryan TJ, Castronuovo JJ. Experience with iatrogenic pediatric vascular injuries. Incidence, etiology, management, and results. Ann Surg 1983;198:430-42. 12. Dalsing MC, Cikrit DF, Sawchuk AP. Open surgical repair of children less than 13 years old with lower extremity vascular injury. J Vasc Surg 2005;41:983-7. 13. Lin PH, Dodson TF, Bush RL, Weiss VJ, Conklin BS, Chen C, et al. Surgical intervention for complications caused by femoral artery catheterization in pediatric patients. J Vasc Surg 2001;34:1071-8. 14. Aspalter M, Domenig CM, Haumer M, Kitzmüller E, Kretschmer G, Hölzenbein TJ. Management of iatrogenic common femoral artery injuries in pediatric patients using primary vein patch angioplasty. J Pediatr Surg 2007;42:1898-902. 15. Shah SR, Wearden PD, Gaines BA. Pediatric peripheral vascular injuries: a review of our experience. J Surg Res 2009;153:162-6. 16. Friedman J, Fabre J, Netscher D, Jaksic T. Treatment of acute neonatal vascular injuries–the utility of multiple interventions. J Pediatr Surg 1999;34:940-5. 17. Chaikof EL, Dodson TF, Salam AA, Lumsden AB, Smith RB 3rd. Acute arterial thrombosis in the very young. J Vasc Surg 1992;16:428-35. 18. Saxena A, Gupta R, Kumar RK, Kothari SS, Wasir HS. Predictors of arterial thrombosis after diagnostic cardiac catheterization in infants and children randomized to two heparin dosages. Cathet Cardiovasc Diagn 1997;41:400-3. 19. Weiner GM, Castle VP, DiPietro MA, Faix RG. Successful treatment of neonatal arterial thromboses with recombinant tissue plasminogen activator. J Pediatr 1998;133:133-6. 20. Monagle P, Chan A, Massicotte P, Chalmers E, Michelson AD. Antithrombotic therapy in children: the Seventh ACCP Conference on Antithrombotic and Thrombolytic Therapy. Chest 2004;126(3 Suppl): 645S-87S. Submitted Jun 16, 2011; accepted Sep 26, 2011.
DISCUSSION Dr George Hamilton (London, United Kingdom). Congratulations on your results; we have a similar experience. What you haven’t mentioned is thrombolysis. As a tertiary referral center, a lot of our children get to us late, having been treated at other hospitals. Our protocol is to start with anticoagulation absolutely monitored with APTT for a period of 6 hours or so. If that doesn’t improve perfusion, systemic thrombolysis is then used. There is a risk, particularly in preterm infants, of cerebral hemorrhage, but that risk actually is worth taking because the results of thrombolysis are universally good except in irreversible ischemia. We would agree absolutely that there is really a very little role for surgical intervention because the results are dreadful. Dr Jesus Matos. The one patient that underwent thrombectomy was transferred from an outside institution, and by the time he got to our institution, it was too late to consider thrombolysis. As you mentioned, there are data to support thrombolysis. In kids that would not tolerate general anesthetic, thrombolysis is always
an adequate option. Unfortunately, in our series we don’t have any kids that underwent thrombolysis treatment. Dr Jonathan Beard (Sheffield, United Kingdom): I’d like to concur with not operating on these babies. My experience with two have been disastrous. It’s like operating on a piece of wet tissue paper. I’m a bit worried about your suggestion that you should wait. You seem to suggest that it is best to wait until the limb is going black before you intervene. Have you thought about other methods of monitoring limb perfusion, maybe compartment pressures, for instance, to see if the compartment pressure is going up? That might be an indication for the need for intervention. Dr Matos. We did not measure compartment pressures on any of our patients. We feel that very close monitoring of these kids by the vascular team several times a day is sufficient. Dr Beard. What features on physical examination do you use to determine intervention?
JOURNAL OF VASCULAR SURGERY Volume 55, Number 4
Dr Matos. Rutherford Class 3 with gangrene and extensive tissue loss. I agree, it’s difficult, because you don’t have the ability to check whether the kid has sensation, whether they’re able to move on command, so it is a little more challenging in that sense. Dr William Turnipseed (Madison, Wisc). I have some anecdotal experience that really predates the more current thrombolytic and antiplatelet medications. I have three patients that I’ve actually followed for almost 20 years. One of my questions will be: Do you have any examples of pediatric infant injuries that predate your study to give you longer follow-up? Because the real critical issue is not what happens in the first 3 months, but for the survivors, information relative to their lifestyle and any morbidity down the road. I can tell you that from my experience, I had three cases that extended over about a 6-year period. They were all very similar. They were all cardiac cases. In each case, a cardiac catheter had knotted in the common iliac, and the external iliac was totally evulsed and pulled out through the groin. This really taught me a lesson, and that is, that if the hypogastric is open and the profunda is open, that anticoagulation actually stands a very good chance of working on these patients. The long-term follow-up has been very interesting. I would have suspected some limb deformity. Your point about generous collateral development in the very young is quite accurate in that all three of these have grown up with normal leg length and normal leg function, after 2 decades of follow-up. Dr Matos. Thank you for your comments. Actually, Dr Dalsing knows, by name, every single patient in this study because he’s been following them very extensively. Unfortunately, it’s difficult for us to follow some of these kids long-term because we are the only tertiary center in the state of Indiana, and a lot of these kids come from out of town. And a lot of time, the follow-up is
Matos et al 1159
based on what the primary care surgeon tells us over the phone. But from what we can tell, Dr Dalsing has been following the longest kid for about 6 years now with no limb discrepancies. Dr Timothy Baxter (Omaha, Neb). My question is about fasciotomies. I’ve had a hard time making that decision in some of these kids. I’ve watched their CPK levels because the physical examination may be hard to interpret. In seeing them back in follow-up, I have seen some gait abnormalities. So the question is, how do you decide about whether to perform a fasciotomy? Dr Matos. The one kid that we performed the open thrombectomy with the fasciotomy had very severe tissue loss with gangrene and had been mismanaged by the outside institution for over 12 hours, so the decision for us to perform the fasciotomy was pretty straightforward. But it is difficult to come to that decision, I agree. Dr Malachi Sheahan (New Orleans, La). Since what you’re describing is the treatment of iatrogenic injuries to the common femoral artery rather than congenital anomalies, I think it would be useful if you expanded the group to include children up to 4 or 5 years of age. Dr Lumsden has one of the largest reported series in which he manages these children fairly aggressively, a lot of them with bypass. Overall, however, the literature suffers from a lack of numbers. So if you have more cases to add to the worldwide experience, I’d be interested to see what happens with these kids up to 2, 3, 4 years old, especially with regard to limb length and functional development. Dr Matos. I agree. Most of the literature out there, including Dr Lumsden’s and Dr Lin’s publication, is from kids ⬎2 years of age that were managed surgically. There is a greater correlation of limb length discrepancy in that age group, but there is not as much in kids younger than 2, which is why we were interested in studying this age group.
Evidence for nonoperative management of acute limb ischemia in infants Jesus M. Matos, MD,a Andres Fajardo, MD,a Michael C. Dalsing, MD,a Raghu Motaganahalli, MD,a George A. Akingba, MD, PhD,a and Michael P. Murphy, MD,a,b Indianapolis, Ind Acute limb ischemia (ALI) in infants is a catastrophic event. We performed a query of our database to determine those with ALI. Twelve patients were identified. The most frequent presentation was cyanotic limbs. Eleven patients were treated nonoperatively with anticoagulation. One patient was treated surgically with Fogarty balloon thrombectomy. There were three deaths all due to associated comorbidities. All had viable limbs on follow-up examination. There were three complications in the patients managed conservatively. Our recommendation for infants presenting with ALI is conservative observation with anticoagulation and intervention only for cases with tissue loss. ( J Vasc Surg 2012;55: 1156-9.)
Acute limb ischemia (ALI) is a catastrophic event that portends a significant probability of limb loss. ALI in the infant population (ⱕ1 year old) is an exceedingly rare clinical event, and consequently, consensus management guidelines do not exist.1-2 Over the past decade, there have been several successful reports of limb salvage in children over a wide range of ages with anticoagulation and/or thrombolytic therapy but no series focusing specifically on the infant population with ALI.3-6 In this report, we provide a review of our experience in the management of ALI in the infant population, demonstrating efficacy in limb salvage with conservative management consisting of anticoagulation and close monitoring of limb viability. METHODS The Indiana University School of Medicine Vascular Surgery Section maintains a database of all vascular interventions and consultations performed by our staff at the Clarian Hospital System (Methodist, University, and Riley). The protocol of this study was approved by institutional review board review. Query of this database for all patients with acute limb ischemia in infants (defined as less than 1 year of age) from 2004 to 2010 was conducted. Data collected and analyzed included age, gender, medical comorbidities, affected extremity, mechanism of occlusion, presenting symptoms, diagnostic studies, treatment, and follow-up data. From the Department of Surgery, Indiana University School of Medicine,a and the Vascular and Cardiac Center for Adult Stem Cell Therapy.b Competition of interest: none. Presented at the 2011 Vascular Annual Meeting of the Society for Vascular Surgery, Chicago, Ill, June 16-18, 2011. Reprint requests: Michael Murphy, MD, Department of Surgery, Indiana University School of Medicine, 1801 N. Senate Blvd., MPC2, #3500, Indianapolis, IN 46202-1228 (e-mail: [email protected]). The editors and reviewers of this article have no relevant financial relationships to disclose per the JVS policy that requires reviewers to decline review of any manuscript for which they may have a competition of interest. 0741-5214/$36.00 Copyright © 2012 by the Society for Vascular Surgery. doi:10.1016/j.jvs.2011.09.092
1156
RESULTS Between November 21, 2004 and April 12, 2010 there were 12 patients identified with ALI7 (Fig 1). Injuries were more common in male patients (n ⫽ 9; 75%) with an average age of 3 months (range, 10 days-8 months). At presentation, seven (59%) patients had absent pulses by examination, and nine (75%) had limb cyanosis (Fig 2). A color-flow duplex ultrasound (DUS) was initially obtained on all patients that provided sufficient information to confirm the diagnosis, mitigating the need for additional imaging studies. Comorbidities included congenital cardiac disease (n ⫽ 5; 42%), pulmonary failure requiring mechanical ventilation (n ⫽ 4; 33%), prematurity (n ⫽ 1; 8%), sepsis (n ⫽ 1; 8%), and intermittent methemoglobinemia (n ⫽ 1; 8%). Intra-arterial catheterizations for diagnostic or monitoring purposes were the cause of ALI in 11 (92%) patients. Vessels of the lower extremity were more commonly injured (n ⫽ 9; 75%), and arteries that were injured were the common femoral artery (n ⫽ 7; 58%), superficial femoral artery (n ⫽ 2; 17%), radial artery (n ⫽ 2; 17%), and brachial artery (n ⫽ 1; 8%). Six of the patients had dopamine running during evaluation of the ischemic event. Twelve patients were anticoagulated with unfractionated heparin (n ⫽ 10) or subcutaneous (SQ) enoxaparin.2 Unfractionated heparin was administered as an initial bolus of 100 units/kg followed by continuous infusion of 20 units/ kg/hr to achieve an activated partial thromboplastin time (APTT) of 2 to 2.5 times normal. DUS was performed serially during the critical period of initiating anticoagulation. All patients were transitioned from heparin to SQ enoxaparin. Patients continued for 3 to 4 weeks with follow-up DUS. There was one failure of anticoagulation with heparin manifesting as ulceration and gangrene 48 hours after initiation of therapy. The patient underwent common femoral artery thrombectomy with a #3 Fogarty balloon catheter and four compartment fasciotomies with restoration of distal pulses. There were two deaths (18%) in the anticoagulation group during treatment for ALI. One patient died 2 days
JOURNAL OF VASCULAR SURGERY Volume 55, Number 4
Fig 1. Arterial duplex ultrasound of 6-month-old infant at time of presentation. Duplex demonstrates a relative absence of flow in the common femoral artery with monophasic waveforms and significantly reduced velocities.
Fig 2. Infant with cyanotic limb at time of presentation.
and the second patient died 9 days after vascular surgery evaluation due to heart failure. Out of the remaining nine patients, we had follow-up data on seven. On follow-up visits, every patient had repeat DUS. Three patients experienced complications felt to be related to limb ischemia: One was found to have a size discrepancy of 1 cm of the foot on the fully functional index limb, a second patient required skin debridement and grafting due to skin necrosis, and the third patient lost a fingernail. One patient had a follow-up DUS 24 days after treatment with enoxaparin that documented persistent occlusion of the common femoral artery with notable development of a collateral branch perfusing the superficial femoral artery (Fig 3). The one patient that was managed with surgical thrombectomy died 30 days after surgery from septic shock unrelated to the surgery or complications of limb ischemia. DISCUSSION Infant vascular injuries are rare and account for ⬍1% of patients with acute limb ischemia seen by vascular surgeons.8 Iatrogenic vascular injuries are more common in the infant population, while older children and adolescent
Matos et al 1157
Fig 3. Arterial duplex ultrasound of a month-old infant 24 days after treatment with anticoagulation, showing occlusion with lack of flow. Blue represents collateral flow formation with green arrow representing refeeding distal flow.
will experience penetrating injuries.9-11 Common causes for infant vascular injuries are cardiac catheterizations and arterial blood pressure monitoring devices.11 “Soft signs” of vascular injury are difficult to evaluate on this population age due to lack of verbalization of pain and diminished or absent sensation.8 In the infant population, active bleeding is the absolute indication for operation.12 There is limited literature when it comes to management of infants with ALI. Surgical management of these injuries represents a technical challenge; not only the small vessel size but also the problem of arterial spasms can cause difficulties. Our institution previously published how open surgical repair in older children with blunt vascular injuries allowed limb salvage in the absence of diagnostic delay.12 There are multiple series that report surgical intervention of pediatric patients1,12-15 and infants2,16,17 with ALI, but according to Lin et al15 operative results in children younger than 2 years leads to less satisfactory outcomes when compared with older children.5,9 Lazarides et al conducted a review of the literature describing outcomes of surgically managed patients in children ⱕ2.5 years of age and found only 48% of patients were able to regain palpable pulses, and limb discrepancy was detected in as many as 15% of patients.3 In terms of a more conservative approach, there are several management guidelines that have been proposed.2,3,18,19 One includes the use of thrombolytic agents as the first-choice therapy for 48 hours followed by microsurgery if no improvement was seen.2 Lazarides et al recommended only operating on infants with definite threat of limb loss and doing systemic heparinization or thrombolytic therapy, knowing that limb length discrepancies will be inevitable.3 The recommended conservative method is the administration of heparin as an initial bolus of 75 to 100 units/kilogram with an infusion depending on age: for the ⬍2 month old, 28 units/kilogram/hour, while older infants require only 20 units/kilogram/hour.8,20 Other
1158 Matos et al
groups recommend the use of thrombolytic agents in a selective group of patients who do not respond to systemic heparin anticoagulation.19,20 Criticism of conservative management includes the possibility of developing compartment syndrome and limb length discrepancies.3,9,11 In the infant population, supportive care and the use of systemic anticoagulation with heparin is the best management for most patients. Limb viability was 100% in this series of infants with ALI managed nonoperatively, and we propose that favorable remodeling in parallel collateral vessels occurs due to changes in flow dynamics with acute occlusion. Our recommendation for infants presenting with ALI is physical examination followed by DUS and systemic anticoagulation with heparin and transitioned to SQ lovenox. Patients should undergo serial examinations to assess changes in perfusion of the index limb and a repeat DUS in 3 to 4 weeks. We further recommend that if adequate perfusion of the limb is established by examination and DUS, then anticoagulation should be stopped. During the observational period, it is important for the vascular surgeon to establish expectations with the family. It is important to warn the family that the child could suffer limb discrepancies. If DUS depicts restoration of flow with collateral formation, anticoagulation can be stopped. Surgical intervention is indicated in patients with tissue loss, active bleeding, or lack of response to systemic anticoagulation. REFERENCES 1. Whitehouse WM, Coran AG, Stanley JC, Kuhns LR, Weintraub WH, Fry WJ. Pediatric vascular trauma. Manifestations, management, and sequelae of extremity arterial injury in patients undergoing surgical treatment. Arch Surg 1976;111:1269-75. 2. Gamba P, Tchaprassian Z, Verlato F, Verlato G, Orzali A, Zanon GF. Iatrogenic vascular lesions in extremely low birth weight and low birth weight neonates. J Vasc Surg 1997;26:643-6. 3. Lazarides MK, Georgiadis GS, Papas TT, Gardikis S, Maltezos C. Operative and nonoperative management of children aged 13 years or younger with arterial trauma of the extremities. J Vasc Surg 2006;43: 72-6; Discussion:6. 4. Kothari SS, Varma S, Wasir HS. Thrombolytic therapy in infants and children. Am Heart J 1994;127:651-7.
JOURNAL OF VASCULAR SURGERY April 2012
5. Perry MO. Iatrogenic injuries of arteries in infants. Surg Gynecol Obstet 1983;157:415-8. 6. Dillon PW, Fox PS, Berg CJ, Cardella JF, Krummel TM. Recombinant tissue plasminogen activator for neonatal and pediatric vascular thrombolytic therapy. J Pediatr Surg 1993;28:1264-8; Discussion:8-9. 7. Norgren L, Hiatt WR, Dormandy JA, Nehler MR, Harris KA, Fowkes FG, et al. Inter-Society Consensus for the Management of peripheral Arterial Disease (TASC II). J Vasc Surg 2007;45 Suppl S:S5-67. 8. Dalsing MC, Cikrit DF, Sawchuk AS. Open surgical repair of children less than 13 years old with lower extremity vascular injury. J Vasc Surg 2005.41 983-7. 9. Smith C, Green RM. Pediatric vascular injuries. Surgery 1981;90: 20-31. 10. Navarre JR, Cardillo PJ, Gorman JF, Clark PM, Martinez BD. Vascular trauma in children and adolescents. Am J Surg 1982;143:229-31. 11. Flanigan DP, Keifer TJ, Schuler JJ, Ryan TJ, Castronuovo JJ. Experience with iatrogenic pediatric vascular injuries. Incidence, etiology, management, and results. Ann Surg 1983;198:430-42. 12. Dalsing MC, Cikrit DF, Sawchuk AP. Open surgical repair of children less than 13 years old with lower extremity vascular injury. J Vasc Surg 2005;41:983-7. 13. Lin PH, Dodson TF, Bush RL, Weiss VJ, Conklin BS, Chen C, et al. Surgical intervention for complications caused by femoral artery catheterization in pediatric patients. J Vasc Surg 2001;34:1071-8. 14. Aspalter M, Domenig CM, Haumer M, Kitzmüller E, Kretschmer G, Hölzenbein TJ. Management of iatrogenic common femoral artery injuries in pediatric patients using primary vein patch angioplasty. J Pediatr Surg 2007;42:1898-902. 15. Shah SR, Wearden PD, Gaines BA. Pediatric peripheral vascular injuries: a review of our experience. J Surg Res 2009;153:162-6. 16. Friedman J, Fabre J, Netscher D, Jaksic T. Treatment of acute neonatal vascular injuries–the utility of multiple interventions. J Pediatr Surg 1999;34:940-5. 17. Chaikof EL, Dodson TF, Salam AA, Lumsden AB, Smith RB 3rd. Acute arterial thrombosis in the very young. J Vasc Surg 1992;16:428-35. 18. Saxena A, Gupta R, Kumar RK, Kothari SS, Wasir HS. Predictors of arterial thrombosis after diagnostic cardiac catheterization in infants and children randomized to two heparin dosages. Cathet Cardiovasc Diagn 1997;41:400-3. 19. Weiner GM, Castle VP, DiPietro MA, Faix RG. Successful treatment of neonatal arterial thromboses with recombinant tissue plasminogen activator. J Pediatr 1998;133:133-6. 20. Monagle P, Chan A, Massicotte P, Chalmers E, Michelson AD. Antithrombotic therapy in children: the Seventh ACCP Conference on Antithrombotic and Thrombolytic Therapy. Chest 2004;126(3 Suppl): 645S-87S. Submitted Jun 16, 2011; accepted Sep 26, 2011.
DISCUSSION Dr George Hamilton (London, United Kingdom). Congratulations on your results; we have a similar experience. What you haven’t mentioned is thrombolysis. As a tertiary referral center, a lot of our children get to us late, having been treated at other hospitals. Our protocol is to start with anticoagulation absolutely monitored with APTT for a period of 6 hours or so. If that doesn’t improve perfusion, systemic thrombolysis is then used. There is a risk, particularly in preterm infants, of cerebral hemorrhage, but that risk actually is worth taking because the results of thrombolysis are universally good except in irreversible ischemia. We would agree absolutely that there is really a very little role for surgical intervention because the results are dreadful. Dr Jesus Matos. The one patient that underwent thrombectomy was transferred from an outside institution, and by the time he got to our institution, it was too late to consider thrombolysis. As you mentioned, there are data to support thrombolysis. In kids that would not tolerate general anesthetic, thrombolysis is always
an adequate option. Unfortunately, in our series we don’t have any kids that underwent thrombolysis treatment. Dr Jonathan Beard (Sheffield, United Kingdom): I’d like to concur with not operating on these babies. My experience with two have been disastrous. It’s like operating on a piece of wet tissue paper. I’m a bit worried about your suggestion that you should wait. You seem to suggest that it is best to wait until the limb is going black before you intervene. Have you thought about other methods of monitoring limb perfusion, maybe compartment pressures, for instance, to see if the compartment pressure is going up? That might be an indication for the need for intervention. Dr Matos. We did not measure compartment pressures on any of our patients. We feel that very close monitoring of these kids by the vascular team several times a day is sufficient. Dr Beard. What features on physical examination do you use to determine intervention?
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Dr Matos. Rutherford Class 3 with gangrene and extensive tissue loss. I agree, it’s difficult, because you don’t have the ability to check whether the kid has sensation, whether they’re able to move on command, so it is a little more challenging in that sense. Dr William Turnipseed (Madison, Wisc). I have some anecdotal experience that really predates the more current thrombolytic and antiplatelet medications. I have three patients that I’ve actually followed for almost 20 years. One of my questions will be: Do you have any examples of pediatric infant injuries that predate your study to give you longer follow-up? Because the real critical issue is not what happens in the first 3 months, but for the survivors, information relative to their lifestyle and any morbidity down the road. I can tell you that from my experience, I had three cases that extended over about a 6-year period. They were all very similar. They were all cardiac cases. In each case, a cardiac catheter had knotted in the common iliac, and the external iliac was totally evulsed and pulled out through the groin. This really taught me a lesson, and that is, that if the hypogastric is open and the profunda is open, that anticoagulation actually stands a very good chance of working on these patients. The long-term follow-up has been very interesting. I would have suspected some limb deformity. Your point about generous collateral development in the very young is quite accurate in that all three of these have grown up with normal leg length and normal leg function, after 2 decades of follow-up. Dr Matos. Thank you for your comments. Actually, Dr Dalsing knows, by name, every single patient in this study because he’s been following them very extensively. Unfortunately, it’s difficult for us to follow some of these kids long-term because we are the only tertiary center in the state of Indiana, and a lot of these kids come from out of town. And a lot of time, the follow-up is
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based on what the primary care surgeon tells us over the phone. But from what we can tell, Dr Dalsing has been following the longest kid for about 6 years now with no limb discrepancies. Dr Timothy Baxter (Omaha, Neb). My question is about fasciotomies. I’ve had a hard time making that decision in some of these kids. I’ve watched their CPK levels because the physical examination may be hard to interpret. In seeing them back in follow-up, I have seen some gait abnormalities. So the question is, how do you decide about whether to perform a fasciotomy? Dr Matos. The one kid that we performed the open thrombectomy with the fasciotomy had very severe tissue loss with gangrene and had been mismanaged by the outside institution for over 12 hours, so the decision for us to perform the fasciotomy was pretty straightforward. But it is difficult to come to that decision, I agree. Dr Malachi Sheahan (New Orleans, La). Since what you’re describing is the treatment of iatrogenic injuries to the common femoral artery rather than congenital anomalies, I think it would be useful if you expanded the group to include children up to 4 or 5 years of age. Dr Lumsden has one of the largest reported series in which he manages these children fairly aggressively, a lot of them with bypass. Overall, however, the literature suffers from a lack of numbers. So if you have more cases to add to the worldwide experience, I’d be interested to see what happens with these kids up to 2, 3, 4 years old, especially with regard to limb length and functional development. Dr Matos. I agree. Most of the literature out there, including Dr Lumsden’s and Dr Lin’s publication, is from kids ⬎2 years of age that were managed surgically. There is a greater correlation of limb length discrepancy in that age group, but there is not as much in kids younger than 2, which is why we were interested in studying this age group.