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An unusual cause of ischemic claudication: a case report1
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CLINICAL NOTE
An Unusual Cause of Ischemic Claudication: A Case Report Michael Fredericson, MD, Brandee L. Waite, MD ABSTRACT. Fredericson M, Waite BL. An unusual cause of ischemic claudication: a case report. Arch Phys Med Rehabil 2003;84:766-7. A 56-year-old woman with a chief complaint of left lowerextremity numbness was referred by her gynecologist to the physical medicine clinic for workup of presumed lumbosacral radiculopathy. She had no history of low back pain, and her symptoms were elicited only with exercise. Results of her neurologic examination and lumbosacral radiographs were normal. Her medical history was significant for advanced cervical cancer, successfully treated with local surgery followed by high-dose pelvic radiation and chemotherapy 2 years before the current onset of symptoms. Subsequent workup with Doppler and arteriogram studies discovered a 3-cm area of diffuse stenosis of the left external iliac artery for which she was successfully treated with balloon angioplasty. This case presents an unusual cause of left leg claudication secondary to left iliac artery stenosis 2 years after pelvic radiation for cervical cancer and shows the necessity for a detailed evaluation of patients’ medical histories. Key Words: Arterial stenosis; Case report; Cervical cancer; Intermittent claudication; Rehabilitation. © 2003 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation TEDMAN’S MEDICAL DICTIONARY defines intermittent claudication (IC) as “a condition caused by ischemia of the S muscles, characterized by attacks of lameness and pain brought on by walking, chiefly in the calf muscles.”1 The definition does not differentiate between vascular, neurogenic, or other causes of pain. However, 3 criteria are used to precisely characterize IC: cramping pain that occurs in the thigh or calf with walking, pain that is relieved by slowing cadence or standing still for a time, and pain that recurs after walking a similar distance and is again relieved by slowing or standing for the same period of time.1 We present an unusual case of left lower-extremity claudication secondary to left iliac artery stenosis 2 years after pelvic radiation for ovarian cancer. CASE DESCRIPTION A 56-year-old woman was referred to our physical medicine and rehabilitation clinic by her gynecologist for presumed lumbar radiculopathy with numbness in the left lower leg for 1 month. Numbness was diffuse in the leg with occasional radiation into the toes, was provoked by walking more than 1 to 2 blocks, and was associated with a cramping sensation of the
From the Division of Physical Medicine and Rehabilitation, Department of Functional Restoration, Stanford University School of Medicine, Stanford, CA. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the author(s) or upon any organization with which the author(s) is/are associated. Reprint requests to Michael Fredericson, MD, Stanford University Medical Center, Physical Medicine and Rehabilitation, 300 Pasteur Dr, Edwards Bldg, Rm R105B, Stanford, CA 94305-5336, e-mail: [email protected]. 0003-9993/03/8405-7428$30.00/0 doi:10.1016/S0003-9993(03)04857-8
Arch Phys Med Rehabil Vol 84, May 2003
posterior calf and constant swelling in the left leg. She denied any associated back, hip, or knee pain, and there were no bowel or bladder complaints. Symptoms were present only with walking, never while the patient was sitting or standing, and were relieved by walking slowly for several minutes. Her medical history was significant for squamous cell carcinoma of the cervix, stage IIIB, diagnosed 2 years previously, for which she underwent local surgery followed by high-dose radiation therapy and chemotherapy (2 rounds of cisplatin/ 5-fluorouracil). She also had a diverting colostomy performed because of a persistent rectovaginal fistula 1 year later. She had done well since that time until the abrupt onset of leg numbness and discomfort 1 month before presenting to our clinic. The results of recent magnetic resonance imaging (MRI) of the patient’s abdomen were negative for recurrent cancer. The only other abnormality noted was evidence of bilateral hydronephrosis, which later was correlated with an arteriogram finding of renal artery stenosis. Her only medication was ranitidine. She had no known allergies, denied ever using tobacco, and was not know to be hypercholesterolemic. On physical examination, the patient had mild pain and limited range of motion (ROM) with lumbar extension and rotation to the left. Flexion and bilateral side bending were within normal limits. Hip ROM was full and pain free in all planes. There was no tenderness to palpation of the lumbar spine, sacroiliac joint, sciatic notch, or greater trochanters. Left calf circumference was 2cm greater than right, and left thigh circumference was 2.5cm greater than right. Grade 1 to 2⫹ edema was present throughout the left lower extremity. The right femoral pulse was slightly attenuated, but she had intact popliteal and dorsal pedis pulses. There was no gross asymmetry in skin temperature, ischemic changes in the lower extremity, or pallor with elevation of the left foot. The patient was able to perform 10 toe raises on both feet, but this was associated with exertion-related numbness and cramping in the left leg. She was able to walk on her heels and perform 10 repetitive squats without pain or asymmetric weakness. The results of the remainder of the motor examination were normal in all lower-extremity myotomes. Sensation was intact through the L2-S1 dermatomes to light touch and pinprick. Muscle stretch reflexes were normal and symmetric. Babinski reflex was negative, and there was absence of clonus at the ankles. There was a negative Tinel’s sign at the fibular head. Straight-leg raising was limited by hamstring tightness at 70° bilaterally. The patient was sent for anteroposterior and lateral radiographs of the lumbosacral spine, the results of which were unremarkable. Because of the high suspicion for vascular occlusion, she then was scheduled for arterial Doppler studies; they revealed high-grade left iliac artery stenosis. Pre- and postexercise ankle brachial indexes were .94 to .96 on the right and .36 to .48 on the left. The patient was able to exercise for only 37 seconds before the onset of left calf numbness and she requested to stop exercising at 1 minute 26 seconds secondary to the discomfort. She subsequently was referred to the vascular surgery department for an arteriogram that demonstrated a 3-cm area of diffuse narrowing limited to the left external iliac artery, except for 85% stenosis of the right renal artery. A successful balloon angioplasty was performed at the time of the
ISCHEMIC CLAUDICATION, Fredericson
study, with immediate resolution of the patient’s symptoms with exercise. After angioplasty, left ankle brachial index improved to 1.02 and 1.11 pre- and postexercise, respectively (normal range, ⬎1.0); right ankle brachial index improved to 1.1 and 1.2 pre- and postexercise, respectively. DISCUSSION A detailed history will reveal important factors to help the clinician narrow the differential between neurogenic and vascular claudication. Neurogenic claudication causes vague, poorly localized leg pain, dysesthesias, and paresthesias in the anterior and posterior thigh and calf related to walking or posturing that compromises the path of the affected nerve.2 The exact pathoanatomy is unknown; however, vascular and mechanical compressive etiologies have been proposed.3 Vascular claudication results in pain, dysesthesias, and paresthesias associated with ambulation, physical exertion, or elevation. Accordingly, relief of symptoms occurs with a flexed spine posture if the etiology is neurogenic and with slowed cadence, rest, or dependent positioning if the etiology is vascular. In addition, pallor and pulselessness are markers of vascular compromise and are absent in pure neurogenic claudication. Differentiating between these 2 etiologies can be difficult. The differential diagnosis of unilateral leg numbness also includes lumbosacral radiculopathy, spinal stenosis, peripheral entrapment, and, rarely, severe anemia from slow blood loss. In this case, there was also a history of cancer treatment with pelvic radiation, which suggested increased likelihood of vascular compromise. Previous irradiation of the pelvic area has been shown to increase incidence of arterial occlusive disease in the iliac arteries and the distal aorta.4 Radiation causes atherosclerotic changes in experimental animals by selective injury to the internal elastic lamina.5 Additionally, atherosclerotic side effects are more common in patients who also have had radiation reactions in other pelvic organs and in smokers.5 This patient’s history of rectovaginal fistula secondary to radiation qualifies her for increased risk of atherosclerotic complications. The swelling in her left lower extremity can be attributed to pelvic surgery with dissection of the groin nodes as well as to radiation therapy, both of which can lead to scarring of the lymphatics. Other than intrinsic vascular disease secondary to the radiation, the biggest concern in this patient was the possibility of extrinsic compression secondary to metastatic disease. The repeat pelvic MRI showed no evidence of recurrent cancer. Increased signal within the perirectal soft tissues, which had not changed significantly from a prior MRI, was thought to be compatible with radiation changes. We were unable to detect any iliac artery changes on routine pelvic MRI; however, magnetic resonance angiography, with special sequences to view the pelvic vessels, had it been performed, likely would have detected these changes. Most patients with IC can be treated with behavior modification, structured exercise programs, or pharmacologic inter-
767
vention. In the United States, there are currently only 2 drugs approved by the US Food and Drug Administration for use in claudication— oxpentifylline and cilostazol. Naftidrofuryl is approved in Europe, but not yet in the United States. There are several other promising agents currently being investigated for use in treating claudication (eg, prostaglandins, angiogenic growth factors, L-arginine).6,7 Surgical treatment is reserved for those with acute arterial occlusion (manifested by pain, pallor, paresthesia, paralysis, “polar” temperature), rest pain, tissue necrosis, disabling pain refractory to more conservative treatment, or for young patients with aorto-iliac occlusion.1 Options include balloon angioplasty, stenting, and atherectomy. Angioplasty is appropriate for patients with discrete local lesions less than 10cm in length with unaffected adjacent segments. Angioplasty of the iliac vessels has produced better results than angioplasty of femoral-popliteal vessels, infrapopliteal vessels, or bypass graft stenoses. Local hematoma and restenosis are the most common complications. Stents often are used in patients who have had failed angioplasty. The iliac artery is the most common site for stenting. Atherectomy is reserved for lesions unlikely to resolve with angioplasty, for example, calcified lesions, diffusely diseased arterial segments, or eccentric stenoses.8 CONCLUSION IC is a common complaint presented to physical medicine and rehabilitation clinics. A detailed history usually will guide the clinician in narrowing the differential between neurogenic and vascular causes. This case discusses a patient with a history of cervical cancer and rectovaginal fistula who was treated with high-dose radiation therapy and 2 years later presented with abrupt onset of claudication symptoms from stenosis of the external iliac artery. References 1. Stedman TL. Stedman’s medical dictionary. 27th ed. Baltimore: Lippincott, Williams & Wilkins; 2000. 2. Criado E, Ramadan F, Keagy BA, Johnson G Jr. Intermittent claudication. Surg Gynecol Obstet 1991;173:163-70. 3. Connor PM, Goodhart C, Grana WA. Ischemic claudication mimicking lumbar disk herniation in the athlete. Orthopedics 1993;16: 613-5. 4. Fritz JM, Delitto A, Welch WC, Erhard RE. Lumbar spinal stenosis: a review of current concepts in evaluation, management, and outcome measurements. Arch Phys Med Rehabil 1998;79: 700-8. 5. Petersson F, Swedenborg J. Atherosclerotic occlusive disease after radiation for pelvic malignancies. Acta Chir Scand 1990;156:36771. 6. Hiatt WR. New treatment options in intermittent claudication: the US experience. Int J Clin Pract Suppl 2001;119:20-7. 7. Donnelly R. Assessment and management of intermittent claudication: importance of secondary prevention. Int J Clin Pract Suppl 2001;119:2-9. 8. Haji-Aghaii M, Fogarty TJ. Balloon angioplasty, stenting, and role of atherectomy. Surg Clin North Am 1998;78:593-616.
Arch Phys Med Rehabil Vol 84, May 2003
CLINICAL NOTE
An Unusual Cause of Ischemic Claudication: A Case Report Michael Fredericson, MD, Brandee L. Waite, MD ABSTRACT. Fredericson M, Waite BL. An unusual cause of ischemic claudication: a case report. Arch Phys Med Rehabil 2003;84:766-7. A 56-year-old woman with a chief complaint of left lowerextremity numbness was referred by her gynecologist to the physical medicine clinic for workup of presumed lumbosacral radiculopathy. She had no history of low back pain, and her symptoms were elicited only with exercise. Results of her neurologic examination and lumbosacral radiographs were normal. Her medical history was significant for advanced cervical cancer, successfully treated with local surgery followed by high-dose pelvic radiation and chemotherapy 2 years before the current onset of symptoms. Subsequent workup with Doppler and arteriogram studies discovered a 3-cm area of diffuse stenosis of the left external iliac artery for which she was successfully treated with balloon angioplasty. This case presents an unusual cause of left leg claudication secondary to left iliac artery stenosis 2 years after pelvic radiation for cervical cancer and shows the necessity for a detailed evaluation of patients’ medical histories. Key Words: Arterial stenosis; Case report; Cervical cancer; Intermittent claudication; Rehabilitation. © 2003 by the American Congress of Rehabilitation Medicine and the American Academy of Physical Medicine and Rehabilitation TEDMAN’S MEDICAL DICTIONARY defines intermittent claudication (IC) as “a condition caused by ischemia of the S muscles, characterized by attacks of lameness and pain brought on by walking, chiefly in the calf muscles.”1 The definition does not differentiate between vascular, neurogenic, or other causes of pain. However, 3 criteria are used to precisely characterize IC: cramping pain that occurs in the thigh or calf with walking, pain that is relieved by slowing cadence or standing still for a time, and pain that recurs after walking a similar distance and is again relieved by slowing or standing for the same period of time.1 We present an unusual case of left lower-extremity claudication secondary to left iliac artery stenosis 2 years after pelvic radiation for ovarian cancer. CASE DESCRIPTION A 56-year-old woman was referred to our physical medicine and rehabilitation clinic by her gynecologist for presumed lumbar radiculopathy with numbness in the left lower leg for 1 month. Numbness was diffuse in the leg with occasional radiation into the toes, was provoked by walking more than 1 to 2 blocks, and was associated with a cramping sensation of the
From the Division of Physical Medicine and Rehabilitation, Department of Functional Restoration, Stanford University School of Medicine, Stanford, CA. No commercial party having a direct financial interest in the results of the research supporting this article has or will confer a benefit upon the author(s) or upon any organization with which the author(s) is/are associated. Reprint requests to Michael Fredericson, MD, Stanford University Medical Center, Physical Medicine and Rehabilitation, 300 Pasteur Dr, Edwards Bldg, Rm R105B, Stanford, CA 94305-5336, e-mail: [email protected]. 0003-9993/03/8405-7428$30.00/0 doi:10.1016/S0003-9993(03)04857-8
Arch Phys Med Rehabil Vol 84, May 2003
posterior calf and constant swelling in the left leg. She denied any associated back, hip, or knee pain, and there were no bowel or bladder complaints. Symptoms were present only with walking, never while the patient was sitting or standing, and were relieved by walking slowly for several minutes. Her medical history was significant for squamous cell carcinoma of the cervix, stage IIIB, diagnosed 2 years previously, for which she underwent local surgery followed by high-dose radiation therapy and chemotherapy (2 rounds of cisplatin/ 5-fluorouracil). She also had a diverting colostomy performed because of a persistent rectovaginal fistula 1 year later. She had done well since that time until the abrupt onset of leg numbness and discomfort 1 month before presenting to our clinic. The results of recent magnetic resonance imaging (MRI) of the patient’s abdomen were negative for recurrent cancer. The only other abnormality noted was evidence of bilateral hydronephrosis, which later was correlated with an arteriogram finding of renal artery stenosis. Her only medication was ranitidine. She had no known allergies, denied ever using tobacco, and was not know to be hypercholesterolemic. On physical examination, the patient had mild pain and limited range of motion (ROM) with lumbar extension and rotation to the left. Flexion and bilateral side bending were within normal limits. Hip ROM was full and pain free in all planes. There was no tenderness to palpation of the lumbar spine, sacroiliac joint, sciatic notch, or greater trochanters. Left calf circumference was 2cm greater than right, and left thigh circumference was 2.5cm greater than right. Grade 1 to 2⫹ edema was present throughout the left lower extremity. The right femoral pulse was slightly attenuated, but she had intact popliteal and dorsal pedis pulses. There was no gross asymmetry in skin temperature, ischemic changes in the lower extremity, or pallor with elevation of the left foot. The patient was able to perform 10 toe raises on both feet, but this was associated with exertion-related numbness and cramping in the left leg. She was able to walk on her heels and perform 10 repetitive squats without pain or asymmetric weakness. The results of the remainder of the motor examination were normal in all lower-extremity myotomes. Sensation was intact through the L2-S1 dermatomes to light touch and pinprick. Muscle stretch reflexes were normal and symmetric. Babinski reflex was negative, and there was absence of clonus at the ankles. There was a negative Tinel’s sign at the fibular head. Straight-leg raising was limited by hamstring tightness at 70° bilaterally. The patient was sent for anteroposterior and lateral radiographs of the lumbosacral spine, the results of which were unremarkable. Because of the high suspicion for vascular occlusion, she then was scheduled for arterial Doppler studies; they revealed high-grade left iliac artery stenosis. Pre- and postexercise ankle brachial indexes were .94 to .96 on the right and .36 to .48 on the left. The patient was able to exercise for only 37 seconds before the onset of left calf numbness and she requested to stop exercising at 1 minute 26 seconds secondary to the discomfort. She subsequently was referred to the vascular surgery department for an arteriogram that demonstrated a 3-cm area of diffuse narrowing limited to the left external iliac artery, except for 85% stenosis of the right renal artery. A successful balloon angioplasty was performed at the time of the
ISCHEMIC CLAUDICATION, Fredericson
study, with immediate resolution of the patient’s symptoms with exercise. After angioplasty, left ankle brachial index improved to 1.02 and 1.11 pre- and postexercise, respectively (normal range, ⬎1.0); right ankle brachial index improved to 1.1 and 1.2 pre- and postexercise, respectively. DISCUSSION A detailed history will reveal important factors to help the clinician narrow the differential between neurogenic and vascular claudication. Neurogenic claudication causes vague, poorly localized leg pain, dysesthesias, and paresthesias in the anterior and posterior thigh and calf related to walking or posturing that compromises the path of the affected nerve.2 The exact pathoanatomy is unknown; however, vascular and mechanical compressive etiologies have been proposed.3 Vascular claudication results in pain, dysesthesias, and paresthesias associated with ambulation, physical exertion, or elevation. Accordingly, relief of symptoms occurs with a flexed spine posture if the etiology is neurogenic and with slowed cadence, rest, or dependent positioning if the etiology is vascular. In addition, pallor and pulselessness are markers of vascular compromise and are absent in pure neurogenic claudication. Differentiating between these 2 etiologies can be difficult. The differential diagnosis of unilateral leg numbness also includes lumbosacral radiculopathy, spinal stenosis, peripheral entrapment, and, rarely, severe anemia from slow blood loss. In this case, there was also a history of cancer treatment with pelvic radiation, which suggested increased likelihood of vascular compromise. Previous irradiation of the pelvic area has been shown to increase incidence of arterial occlusive disease in the iliac arteries and the distal aorta.4 Radiation causes atherosclerotic changes in experimental animals by selective injury to the internal elastic lamina.5 Additionally, atherosclerotic side effects are more common in patients who also have had radiation reactions in other pelvic organs and in smokers.5 This patient’s history of rectovaginal fistula secondary to radiation qualifies her for increased risk of atherosclerotic complications. The swelling in her left lower extremity can be attributed to pelvic surgery with dissection of the groin nodes as well as to radiation therapy, both of which can lead to scarring of the lymphatics. Other than intrinsic vascular disease secondary to the radiation, the biggest concern in this patient was the possibility of extrinsic compression secondary to metastatic disease. The repeat pelvic MRI showed no evidence of recurrent cancer. Increased signal within the perirectal soft tissues, which had not changed significantly from a prior MRI, was thought to be compatible with radiation changes. We were unable to detect any iliac artery changes on routine pelvic MRI; however, magnetic resonance angiography, with special sequences to view the pelvic vessels, had it been performed, likely would have detected these changes. Most patients with IC can be treated with behavior modification, structured exercise programs, or pharmacologic inter-
767
vention. In the United States, there are currently only 2 drugs approved by the US Food and Drug Administration for use in claudication— oxpentifylline and cilostazol. Naftidrofuryl is approved in Europe, but not yet in the United States. There are several other promising agents currently being investigated for use in treating claudication (eg, prostaglandins, angiogenic growth factors, L-arginine).6,7 Surgical treatment is reserved for those with acute arterial occlusion (manifested by pain, pallor, paresthesia, paralysis, “polar” temperature), rest pain, tissue necrosis, disabling pain refractory to more conservative treatment, or for young patients with aorto-iliac occlusion.1 Options include balloon angioplasty, stenting, and atherectomy. Angioplasty is appropriate for patients with discrete local lesions less than 10cm in length with unaffected adjacent segments. Angioplasty of the iliac vessels has produced better results than angioplasty of femoral-popliteal vessels, infrapopliteal vessels, or bypass graft stenoses. Local hematoma and restenosis are the most common complications. Stents often are used in patients who have had failed angioplasty. The iliac artery is the most common site for stenting. Atherectomy is reserved for lesions unlikely to resolve with angioplasty, for example, calcified lesions, diffusely diseased arterial segments, or eccentric stenoses.8 CONCLUSION IC is a common complaint presented to physical medicine and rehabilitation clinics. A detailed history usually will guide the clinician in narrowing the differential between neurogenic and vascular causes. This case discusses a patient with a history of cervical cancer and rectovaginal fistula who was treated with high-dose radiation therapy and 2 years later presented with abrupt onset of claudication symptoms from stenosis of the external iliac artery. References 1. Stedman TL. Stedman’s medical dictionary. 27th ed. Baltimore: Lippincott, Williams & Wilkins; 2000. 2. Criado E, Ramadan F, Keagy BA, Johnson G Jr. Intermittent claudication. Surg Gynecol Obstet 1991;173:163-70. 3. Connor PM, Goodhart C, Grana WA. Ischemic claudication mimicking lumbar disk herniation in the athlete. Orthopedics 1993;16: 613-5. 4. Fritz JM, Delitto A, Welch WC, Erhard RE. Lumbar spinal stenosis: a review of current concepts in evaluation, management, and outcome measurements. Arch Phys Med Rehabil 1998;79: 700-8. 5. Petersson F, Swedenborg J. Atherosclerotic occlusive disease after radiation for pelvic malignancies. Acta Chir Scand 1990;156:36771. 6. Hiatt WR. New treatment options in intermittent claudication: the US experience. Int J Clin Pract Suppl 2001;119:20-7. 7. Donnelly R. Assessment and management of intermittent claudication: importance of secondary prevention. Int J Clin Pract Suppl 2001;119:2-9. 8. Haji-Aghaii M, Fogarty TJ. Balloon angioplasty, stenting, and role of atherectomy. Surg Clin North Am 1998;78:593-616.
Arch Phys Med Rehabil Vol 84, May 2003