Femoral neuropathy as a complication of prolonged bleeding in hemophilia has been well documented since Tallroth reported his first case in 1939 and second case in 1941 . M7 A case of femoral neuropathy secondary to heparin anticoagulation was reported by Calverley and Mulder in 1960 and observed at surgical decompression by Kettlekamp in 1969.4,8 With the widening use of anticoagulation therapy this once rare finding has been noted with greater frequency. 3-6.8 .9, 14.15.17-19 The purpose of this article is to present a case of a heparin induced intrapelvic hematoma causing femoral neuropathy and to correlate the computerized tomographic findings with the previously proposed pathogenesis.
A 73-year-old white male was admitted with exertional burning retrosternal pain. He had a past history of hypertension, and current medications were Dyazide and Persantine. An EKG revealed T-wave inversion on the inferior chest leads. A nitro-drip was begun and titrated to keep the blood pressure to greater than 100 systolic. Persantine 75 mg and aspirin 5 g, orally, three times a day were administered.
Three day post-admission he was taken for cardiac catheterization and found to have 99% occlusion of the superior dominant right coronary artery. A percutaneous trans coronary angioplasty was performed using the right brachial artery for access. Five-thousand units of heparin were administered intravenously prior to the procedure and 10,000 units during the procedure. Post-angioplasty, heparin was continued at 1200 units per hour. Five hours postprocedure, the partial thromboplastin time (PTT) was greater than 150. The following morning, bleeding from the catheterization site was noted and the PTT level was 107. The heparin was discontinued, and the PTT dropped to 54.
The patient went into ventricular fibrillation and was returned to the catheterization suite where the site of the previous angioplasty was found to be thrombosed, therefore, the procedure was repeated. Five-thousand units of heparin preoperatively and 10,000 intraoperatively were administered. Post-procedure heparin at 500 units per hour was continued and the PTT was again noted to be greater than 150, therefore, the heparin was decreased to 300 units per hour. The PTT level six hours following heparin reduction was 50. Two days later, the patient developed spontaneous severe pain in the back with radiation to the left lower extremity. There was noted to be a large thigh ecchymosis with a palpable mass in the left iliac fossa. Pulses were normal and no calf tenderness was present. Sensation was intact except for an area of decrease to pinprick on the medial left thigh. The left patella tendon reflex was absent with the right 2/4. Achilles tendon reflexes were 2/4 bilaterally. The patient experienced considerable pain during the examination rendering muscle grading difficult. Left quadriceps strength was 0/5, adductors 2/5, and hip flexors 1/5 with all distal muscle function normal. All right lower extremity muscle function was normal. Sphincter sensation and tone were normal.
The hemoglobin was decreased at 5.5 with no site of major external blood loss. A chest x-ray was negative and no blood was present in the urine. Because of the falling hemoglobin with severe back and flank pain, a CT scan of the abdomen and pelvis was obtained. The CT scan revealed a large mass on the left side of the abdominal cavity (Fig. 1) extending from L3 to below the inguinal liagment (Fig. 2). It was felt to be consistent with bleeding into the iliacus and psoas muscles.
Fig. 1: Transverse CT image at the level of the sacroiliac joints. The arrows delineate the massive increase in size of the psoas and iliacus muscles on the left as compared to the right.
Fig. 2: Transverse CT image at the level of the roof of the acetabulum. The psoas and iliacus are merging to form a single Compartment. The iliopsoas is slightly larger than on the contralateral side.
After 2 weeks of bed rest, range of motion to the extremity was initiated as well as ambulation with a walker bearing weight as tolerated. At this time left adductor and hip flexor strength had returned to normal, thus, implicating pain as the source of initial decreased grades in these groups. Quadriceps strength remained decreased, now at 1/5. At 3 months follow up, sensation had improved but was still subjectively decreased to pinprick. Quadriceps function improved to grade 4/5 with all other muscle groups grade 5.
The femoral and lateral femoral cutaneous nerves arise from the posterior divisions of the lumbar plexus. The femoral nerve most commonly arises from the second, third, and fourth lumbar nerves and emerges through the lateral border of the psoas to eventually travel in the groove between the iliacus and psoas muscles. As it emerges beneath the inguinal ligament into the femoral triangle it divides into a number of terminal branches.20
The fascia overlying the iliopsoas muscles is attached proximally to the medial accurate ligament of the diaphragm, medially to the vertebral column, while laterally it is continuous with the inner iliac fascia. The iliac fascia is continuous with the transversalis fascia after it firmly inserts on the iliac crest. As the iliopsoas fascia courses medially, the layer becomes confluent with the pectineal fascia to form the iliopectineal fascia which is anchored to the pelvic brim. Deep to the inguinal ligament is a thickened band of fascia, the iliopectineal arch. This arch passes from the junction of the iliac fascia and inguinal ligament laterally while medially it attaches to the pubis between the psoas major and the pectineus just lateral to the femoral artery. This divides the space deep to the inguinal ligament into two compartments, one containing the iliopsoas muscle and femoral nerve while the other contains the femoral sheath and vessels.2'20 The femoral nerve lies beneath the iliac fascia and anterior to the iliacus muscle as it exits the pelvis. As the femoral nerve exits beneath the iliopectineal arch it passes through the iliopsoas fascia to lie anterior to this structure. The fascial spaces of the iliacus and psoas muscles become a singular space distal to the inguinal ligament. The iliopsoas musculotendinous junction is therefore distal to the iliopectineal arch.2,20
Tallroth is generally given credit for describing femoral neuropathy in hemophilia secondary to a report in 1939.17 The condition has been variously reported by Gunther 1923 and 1935, Bulloch and Fildes 1912, Seddon 1930, Birtch 1937, Aggelerand Lucias 1944, Lyons 1953, and Brower 1966.1,2,12,13,21-22 In 1964, Silverstein in a large series found 28 of 206 hemophiliacs, 13.6%, to have peripheral neuropathy secondary to bleeding, 17 of which were femoral neuropathies.22 This figure is higher than subsequent reports for overall incidence of neuropathy, however it is consistent with the ratio of femoral to other neuropathies.5,6,14
Groch and associates in 1959 described a lesion of the lumbosacral plexus secondary to anticoagulation.16 Calverley and Mudler in 1960 claimed to have seen the complication of anticoagulation femoral neuropathy although the case was not described in detail.4 Deboli in 1966 reported two cases of heparin induced anticoagulation femoral neuropathy.19 In 1967, Goodfellow presented 24 patients with femoral neuropathy 23 secondary to hemophilia and one secondary to an anticoagulation complication.5 Kettlekamp, in 1969, reported a case of femoral neuropathy secondary to heparin anticoagulation.8 With the now widespread use of anticoagulation, there has been a marked increase in reports of this findjng 3-22.214.171.124 1. 14-16.19
Different mechanisms for femoral nerve entrapment with bleeding disorders have been proposed.2'5'23 Brower proposed a tear at the musculotendinous junction as the etiology.2 He recreated the hip flexion and femoral trigone swelling in cadavers by injecting saline within the iliopsoas muscle distal to the inguinal ligament. He could not create hip flexion by injection within the iliacus muscle. Kettlekamp's case seemed to support Browers conclusion.8 The femoral nerve in his case was explored, a large tense hematoma was found distal to the inguinal ligament with little blood found through a second incision proximal to the inguinal ligament. Goodfellow in an experiment similar to Brower's had different conclusions.5 Like Brower, he felt that fluid within the psoas muscle would not lead to nerve compression because of the distensible nature of this structure, however, although not recreating hip flexion he felt that fluid injected into the iliacus muscle eventually lead to pressure on the femoral nerve at the inguinal ligament. It was his conclusion that should enough fluid accumulate it could travel down the iliac compartment distal to the inguinal ligament and then traverse proximally into the psoas compartment.
Nobel in 1980 expanded on the previous work of Brower and Goodfellow by dissecting 44 cadavers as well as performing latex injection studies.9 Like Goodfellow, he was able to inject fluid into the iliacus muscle and overcome the block of the inguinal ligament. He also demonstrated retrograde filling of the psoas muscle thus supporting the concept of separate psoas and iliacus compartments bound by epimysium and covered by a common iliopsoas fascia. An interesting finding in Nobel's dissection was additional laminated fascial compartments superficial to the iliacus fascia thus forming further reenforcement to the funnel area of the iliac fossa.
Two basic pathogenic mechanisms have been described. First, iliopsoas musculotendinous junction disruption can lead to bleeding in the femoral triangle with resultant femoral neuropathy and relative confinement of hematoma distal to the inguinal ligament as described by Brower and directly observed by Keetelkamp,2,8 Second, Nobel and Goodfellow have demonstrated by latex and saline injections respectively that fluid within the iliacus muscle proper can traverse the block of the inguinal ligament and enter the femoral triangle thus entrapping the overlying femoral nerve.5,9 It is yet to be determined if one mechanism is more specific for anticoagulation induced neuropathy versus hemophilic neuropathy. One would assume that cases of anticoagulation induced bleeding are associated with abnormal coagulation studies. Although, an elevated PTT was well documented, in this case it is not a constant finding.19
When reviewing the CT scan of this case one can easily see a massive increase in size of the left iliacus and psoas muscles within the pelvis (Fig. I). The mass extends from the distal end of L3 to the level of the hip joint which would correspond approximately to the point at which the iliopsoas traverses beneath the inguinal ligament (Fig. 2 ). These findings, although not ruling out the source of blood as the musculotendinous junction, seem to suggest that bleeding within the muscle proximal to the inguinal ligament is responsible for this case of femoral nerve compression.
1. Aggeler PM. Lucias SP: The neurologic complication of hemophilia. J Nerv Mem Disease 1944; 99:475-500.
2. Brower TD. Wilde AH: Femoral neuropathy in hemophilia. J Bone Joint Surg 1966; 48:487-492.
3. Butterticld WC. Neviaser RJ. Roberts MP: Femoral neuropathy and anticoagulants. Ann Surg 1972; 176:58-61.
4. Calverley JR, Mulder DW: Femoral neuropathy. Neurol 1960: 10:963.
5 . Goodfellow J, D'Afearn CB . Matthews JM: iliacus hematoma: A common complication of haemophilia. J Bone Joint Surg 1967: 49:748-756.
6. Hilgartncr MW: Hemophilia in the Child and Adult. New York, Masson Publishing, 1982, pp 99-120.
7. Jordan HH: Hemophiliac Arthropathies. Springfield, Illinois, Charles C. Thomas. 1958, pp 78-104.
8. Kettlekamp DB, Powers SR: Femoral compression neuropathy in hemorrhagic disorders. Arch Surg 1969; 98:367-368.
9. Nobel W, Marks SC, Kubik SK: The anatomical basis of femoral nerve palsy following iliacus hematoma. J Neurosurg 1980; 52:533-539.
10. Seddons HJ: Haemophilia as a cause of lesions in the nervous system. Brain 1930; 53:306-310.
11. Stern MB, Spiegel P: Femoral neuropathy as a complication of heparin anticoagulation therapy. Clin Orthop 1975; 106:140-142.
12. Tallroth A: Femoral paralysis hemorrhage in iliopsoas muscle causing injury to the femoral nerve - Report of a second case. Acta Chir Scand 1941; 84:124-128.
13. Tallroth A: Hemophilia with spontaneous hemorrhage in the iliopsoas muscle followed by injury to the femoral nerve - Report of a case. Acta Chir Scand 1939; 82:1-9.
14. Brantigan JW, Owens ML, Moudy FG: Femoral neuropathy complicating anticoagulant therapy. Am J Surg 1976; 132:108-109.
15. Galzio R, Lucantoni D, Zenobii M, et al: Femoral neuropathy caused by iliacus hematoma. Sung Neurol 1983; 20:254-257.
16. Groch SN. et al: Problems of anticoagulant therapy in cerebrovascular disease. Neurol 1959; 9:786-793.
17. Mastroianni PO, Roberts MP: Femoral neuropathy and retroperitoneal hemorrhage. Neurosurg 1983; 13:44-47.
18. Simeone JR, Robinson R, Rothman SLG, et al: Computerized tomographic demonstration of a retroperitoneal hematoma causing femoral neuropathy. J Neurosurg 1977; 47:946-948.
19. Wells J, Templeton J : Femoral neuropathy associated with anticoagulation therapy. Clin Orthop 1977; 124:155-160.
20. Hollinshead WH: Anatomy for Surgeons. New York, Harper and Row. 1969, pp 707-708.
21. Lyons JB: Femoral nerve lesion in haemophilia. J Irish Med Assoc 1953; 32:110-111.
22. Silverstein A: Neuropathy in hemophilia. JAMA 1964; 190:554-555.
23. Bigelow NH, Graves RW: Peripheral nerve lesions in hemorrhagic diseases. Arch Neurol Psychiatry 1952; 68:819-830.
24. Duthie RB, Matthews JM, Rizza CR, et al: The Management of Musculoskeletal Problems in the Haemophilias. London, Blackwell Scientific Publications, 1972, pp 63-72.