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Delayed manifestation of abdominal aortic stenosis in a child presenting 10 years after blunt abdominal trauma José A. Diaz, MD, a Brendan T. Campbell, MD, a Mohammed M. Moursi, MD, b Cristiano Boneti, MD, a Evan R. Kokoska, MD, a Richard J. Jackson, MD, a and Samuel D. Smith, MD, a Little Rock, Ark We report the case of a 13-year-old boy who, at 3 years of age, was a rear seat–restrained passenger in a high-speed motor vehicle crash necessitating segmental small-bowel resection. The patient remained well for 10 years; then he began to have exercise-induced fatigue in his lower extremities. Routine physical examination revealed a bruit and thrill in the mid abdomen and diminished femoral pulses. Aortic stenosis was diagnosed and treated surgically. We discuss the patho- physiology of the lesion and review the literature. This is the first report of abdominal aortic stenosis 10 years after blunt abdominal trauma in a child. ( J Vasc Surg 2006;44:1104-6.) Blunt trauma is common in children and is a major cause of death and disability. Aortic injury after blunt abdominal trauma accounts for less than 0.1% of injuries. 1 Injuries confined to the abdominal aorta are important because of their potential for devastating complications if diagnosis is delayed. Seat belt–associated bunt abdominal trauma may lead to aortic injury. Sudden deceleration and direct force by mechanical compression against the vertebral column have been described as the main factors involved in this type of aortic injury. 2-5 Aortic lesions secondary to blunt abdomi- nal trauma include simple contusion, intimal disruption, intramural hematoma, pseudoaneurysm, and rupture. 5 De- pending on the lesion, presentation may be acute or de- layed (weeks to years after trauma). 2,4,6 Delayed manifes- tation of aortic injuries is unusual in children, and a high suspicion index is essential for their diagnosis. CASE REPORT A 13-year-old boy was involved in a high-speed motor vehicle accident as a seat belt–restrained rear-seat occupant when he was 3 years old. He sustained blunt abdominal trauma that necessi- tated resection of approximately 15 cm of small bowel. The patient recovered uneventfully and remained well for 10 years, until he began complaining of intermittent abdominal discomfort, weight loss, and decreased appetite. The patient also complained of occa- sional cramps in his lower extremities during exercise. Routine physical examination was normal except for a loud bruit and thrill in the mid abdomen and slightly decreased femoral pulses. Signs and symptoms remained stable for 3 months from first consulta- tion to surgery. Abdominal ultrasonography showed infrarenal aortic stenosis and poststenotic dilatation. The ankle-brachial in- dex at rest was as follows: right, 0.98; left, 0.92. The exercise ankle-brachial index was as follows: right, 0.79; left, 0.66. Diag- nosis of infrarenal aortic stenosis was confirmed by a computed tomographic angiogram with three-dimensional reconstruction (Fig 1). At operation, the infrarenal aorta was clamped and opened longitudinally. An area of tight web stenosis was observed at the mid infrarenal aorta (Fig 2). The stenotic web was excised, and patch aortoplasty was performed by using bovine pericardium. Pathologic analysis demonstrated subintimal fibrosis. Immediately after surgery, femoral and pedal pulses were present and bilaterally symmetric. The patient recovered uneventfully from surgery and was discharged home on postoperative day 3 on aspirin. Rest and exercise ankle-brachial indices were normal 30 days after surgery and at the 4-month follow-up. DISCUSSION Abdominal aortic injuries are uncommon after blunt trauma. In a series of 870 patients requiring exploratory laparotomy for blunt abdominal trauma, only 1 had an abdominal aortic injury. 1 Two retrospective studies of 8710 autopsies reported 347 blunt aortic injuries, but only 16 (0.18%) were abdominal aortic injuries. 2 The low incidence of abdominal aortic injury may be due to its retroperitoneal location, 1-6 but this location is also the reason for direct force due to mechanical compres- sion against the vertebra. 2 The proposed mechanism of aortic stenosis is assumed to be the intimal tear and subse- quent disruption of the vasa vasorum leading to subintimal fibrosis, which results in partial occlusion of the abdominal aorta. 3 Manifestations of blunt aortic injury can be broadly di- vided into acute (24 hours) and chronic (24 hours). 2,4 Most cases are acute and present with signs of an acute abdomen and/or neurovascular deficits in the lower ex- tremities. 3 In a review of the literature, we only found seven cases of delayed manifestation of aortic stenosis (Table). Patients’ From the Department of Pediatric Surgery, Arkansas Children’s Hospital, a and the Division of Vascular Surgery, b University of Arkansas for Medical Sciences. Competition of interest: none. Reprint requests: José A. Diaz, MD, Arkansas Children’s Hospital, Depart- ment of Surgery, Slot 837, 800 Marshall Street, Little Rock, AR 72202 (e-mail: [email protected]). 0741-5214/$32.00 Copyright © 2006 by The Society for Vascular Surgery. doi:10.1016/j.jvs.2006.06.040 1104
Transcript
Page 1: Delayed manifestation of abdominal aortic stenosis in a child presenting 10 years after blunt abdominal trauma

Delayed manifestation of abdominal aortic stenosisin a child presenting 10 years after bluntabdominal traumaJosé A. Diaz, MD,a Brendan T. Campbell, MD,a Mohammed M. Moursi, MD,b Cristiano Boneti, MD,a

Evan R. Kokoska, MD,a Richard J. Jackson, MD,a and Samuel D. Smith, MD,a Little Rock, Ark

We report the case of a 13-year-old boy who, at 3 years of age, was a rear seat–restrained passenger in a high-speed motorvehicle crash necessitating segmental small-bowel resection. The patient remained well for 10 years; then he began to haveexercise-induced fatigue in his lower extremities. Routine physical examination revealed a bruit and thrill in the midabdomen and diminished femoral pulses. Aortic stenosis was diagnosed and treated surgically. We discuss the patho-physiology of the lesion and review the literature. This is the first report of abdominal aortic stenosis 10 years after blunt

abdominal trauma in a child. (J Vasc Surg 2006;44:1104-6.)

Blunt trauma is common in children and is a majorcause of death and disability. Aortic injury after bluntabdominal trauma accounts for less than 0.1% of injuries.1

Injuries confined to the abdominal aorta are importantbecause of their potential for devastating complications ifdiagnosis is delayed.

Seat belt–associated bunt abdominal trauma may leadto aortic injury. Sudden deceleration and direct force bymechanical compression against the vertebral column havebeen described as the main factors involved in this type ofaortic injury.2-5 Aortic lesions secondary to blunt abdomi-nal trauma include simple contusion, intimal disruption,intramural hematoma, pseudoaneurysm, and rupture.5 De-pending on the lesion, presentation may be acute or de-layed (weeks to years after trauma).2,4,6 Delayed manifes-tation of aortic injuries is unusual in children, and a highsuspicion index is essential for their diagnosis.

CASE REPORT

A 13-year-old boy was involved in a high-speed motor vehicleaccident as a seat belt–restrained rear-seat occupant when he was3 years old. He sustained blunt abdominal trauma that necessi-tated resection of approximately 15 cm of small bowel. The patientrecovered uneventfully and remained well for 10 years, until hebegan complaining of intermittent abdominal discomfort, weightloss, and decreased appetite. The patient also complained of occa-sional cramps in his lower extremities during exercise. Routinephysical examination was normal except for a loud bruit and thrillin the mid abdomen and slightly decreased femoral pulses. Signsand symptoms remained stable for 3 months from first consulta-tion to surgery. Abdominal ultrasonography showed infrarenal

From the Department of Pediatric Surgery, Arkansas Children’s Hospital,a

and the Division of Vascular Surgery,b University of Arkansas for MedicalSciences.

Competition of interest: none.Reprint requests: José A. Diaz, MD, Arkansas Children’s Hospital, Depart-

ment of Surgery, Slot 837, 800 Marshall Street, Little Rock, AR 72202(e-mail: [email protected]).

0741-5214/$32.00Copyright © 2006 by The Society for Vascular Surgery.

doi:10.1016/j.jvs.2006.06.040

1104

aortic stenosis and poststenotic dilatation. The ankle-brachial in-dex at rest was as follows: right, 0.98; left, 0.92. The exerciseankle-brachial index was as follows: right, 0.79; left, 0.66. Diag-nosis of infrarenal aortic stenosis was confirmed by a computedtomographic angiogram with three-dimensional reconstruction(Fig 1).

At operation, the infrarenal aorta was clamped and openedlongitudinally. An area of tight web stenosis was observed at themid infrarenal aorta (Fig 2). The stenotic web was excised, andpatch aortoplasty was performed by using bovine pericardium.Pathologic analysis demonstrated subintimal fibrosis. Immediatelyafter surgery, femoral and pedal pulses were present and bilaterallysymmetric. The patient recovered uneventfully from surgery andwas discharged home on postoperative day 3 on aspirin. Rest andexercise ankle-brachial indices were normal 30 days after surgeryand at the 4-month follow-up.

DISCUSSION

Abdominal aortic injuries are uncommon after blunttrauma. In a series of 870 patients requiring exploratorylaparotomy for blunt abdominal trauma, only 1 had anabdominal aortic injury.1 Two retrospective studies of8710 autopsies reported 347 blunt aortic injuries, but only16 (0.18%) were abdominal aortic injuries.2

The low incidence of abdominal aortic injury may bedue to its retroperitoneal location,1-6 but this location isalso the reason for direct force due to mechanical compres-sion against the vertebra.2 The proposed mechanism ofaortic stenosis is assumed to be the intimal tear and subse-quent disruption of the vasa vasorum leading to subintimalfibrosis, which results in partial occlusion of the abdominalaorta.3

Manifestations of blunt aortic injury can be broadly di-vided into acute (�24 hours) and chronic (�24 hours).2,4

Most cases are acute and present with signs of an acuteabdomen and/or neurovascular deficits in the lower ex-tremities.3

In a review of the literature, we only found seven cases

of delayed manifestation of aortic stenosis (Table). Patients’
Page 2: Delayed manifestation of abdominal aortic stenosis in a child presenting 10 years after blunt abdominal trauma

JOURNAL OF VASCULAR SURGERYVolume 44, Number 5 Diaz et al 1105

Fig 1. Three-dimensional computed tomographic angiogram showing abdominal aortic stenosis.

Fig 2. A, Tight web stenosis observed at the mid infrarenal aorta. B, Enlarged aortic lumen after stenotic web removal.

1, Aorta.

Table. Delayed manifestation in aortic stenosis

Study Sex Age (yrs) Delay (�24 h) Signs and symptoms Site of stenosis Treatment

Campbell7 F 24 7 mo Bruit—pulsating mass 8 cm proximal to thebifurcation

Dacron* tube graft

Rybak8 F 40 3 d Cold and pulseless limbs 3 cm proximal to thebifurcation

Aortoiliac bypass (bifurcationDacron graft)

Matolo9 F 28 4 mo Abdominal thrill and bruit Infrarenal abdominalaorta (2 rings)

Dacron tube graft

Sloop10 M 46 10 mo Bilateral intermittent claudication Infrarenal abdominalaorta (2 rings)

Tube graft

Bergqvist11 M 42 9 y Bilateral intermittent claudication Infrarenal abdominalaorta

Aortoiliac bypass (bifurcationDacron graft)

Nizzero12 M 44 9 mo Numbness to 30 meters (relievedby rest)

1.5 cm proximal tothe bifurcation

Aortoiliac bypass (bifurcationDacron graft)

Shindo13 M 67 1 y Bilateral intermittent claudication Infrarenal abdominalaorta

Dacron tube graft

*DuPont, Wilmington, Del.

Page 3: Delayed manifestation of abdominal aortic stenosis in a child presenting 10 years after blunt abdominal trauma

JOURNAL OF VASCULAR SURGERYNovember 20061106 Diaz et al

ages ranged from 24 to 64 years. Presentation occurredbetween 3 days and 9 years after the trauma. The mostcommon site of aortic injury was an infrarenal location. Allpatients were treated with an open operation. In our pa-tient, the time between injury and diagnosis was 10 years.This is the first case in which we are aware of delayedpresentation of abdominal aortic stenosis after blunt ab-dominal trauma in a child.

Diagnostic modalities and treatment performed in thiscase did not differ from the standard of care for adultpatients. Percutaneous transluminal angioplasty with orwithout stent placement may be considered an alternativetreatment for this condition. However, concern still re-mains about stent placement in children because of thefixed internal diameter of the device in relationship to theincreasing diameter of the contiguous aorta due to normalgrowth.14

Because the growth rate acceleration starts at the age of12 to 13 years,15 it could be hypothesized that the collat-eral vessels prevented symptoms during childhood butresulted in insufficiency during the growth-accelerationtime. This could explain why our patient became symptom-atic at the age of 13 years.

In conclusion, silent processes such as aortic stenosis inchildren may become clinically evident during pubertybecause of the growth-rate acceleration. Blunt abdominaltrauma should be considered as a cause of aortic stenosis inchildren, even 10 years after the event.

REFERENCES

1. Cox EF. Blunt abdominal trauma. A 5-year analysis of 870 patients

requiring celiotomy [review]. Ann Surg 1984;199:467-74.

2. Muniz AE, Haynes JH. Delayed abdominal aortic rupture in a childwith a seat-belt sign and review of the literature [review]. J Trauma2004;56:194-7.

3. Randhawa MP Jr, Menzoian JO. Seat belt aorta [review]. Ann Vasc Surg1990;4:370-7.

4. Amin A, Alexander JB, O’Malley KF, Doolin E. Blunt abdominal aortictrauma in children: case report [review]. J Trauma 1993;34:293-6.

5. Lassonde J, Laurendeau F. Blunt injury of the abdominal aorta. AnnSurg 1981;194:745-8.

6. Pisters PW, Heslin MJ, Riles TS. Abdominal aortic pseudoaneurysmafter blunt trauma [review]. J Vasc Surg 1993;18:307-9.

7. Campbell DK, Austin RF. Seat-belt injury: injury of the abdominalaorta. Radiology 1969;92:123-4.

8. Rybak JJ, Thomford NR. Acute occlusion of the infrarenal aorta fromblunt trauma. Am Surg 1969;35:444-7.

9. Matolo NM, Danto LA, Wolfman EF Jr. Traumatic aneurysm of theabdominal aorta. Report of two cases and review of the literature. ArchSurg 1974;108:867-9.

10. Sloop RD, Robertson KA. Nonpenetrating trauma of the abdominalaorta with partial vessel occlusion: report of two cases. Am Surg 1975;41:555-9.

11. Bergqvist D, Takolander R. Aortic occlusion following blunt trauma ofthe abdomen. J Trauma 1981;21:319-22.

12. Nizzero A, Miles JT. Blunt trauma to the abdominal aorta. CMAJ1986;135:219-20.

13. Shindo S, Ogata K, Katahira S, Iyori K, Ishimoto T, Kobayashi M, et al.Delayed manifestation of aortic stenosis after blunt abdominal trauma:report of a case. Surg Today 1997;27:76-9.

14. Takach TJ, Anstadt MP, Moore HV. Pediatric aortic disruption. TexHeart Inst J 2005;32:16-20.

15. Vignolo M, Brignone A, Mascagni A, Ravera G, Biasotti B, Aicardi G.Influence of age, sex, and growth variables on phalangeal quantitativeultrasound measures: a study in healthy children and adolescents. CalcifTissue Int 2003;72:681-8.

Submitted May 21, 2006; accepted Jun 22, 2006.


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