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Ulnar tunnel syndrome from giant cell tumor of tendon sheath: A case report
Ulnar tunnel syndrome from giant cell tumor of tendon sheath: A case report Cauley W. Hayes, Jr., M.D., Chattanooga, Tenn.
A
42-year-old Caucasian male warehouse worker had had increasing weakness of the right hand for 6 months. It was accompanied by difficulty with fine movements and a hand thinner in appearance. Two years previously, after several months of mild weakness, an operation on the dorsal ulnar aspect of the wrist had disclosed a solid lesion which was removed piecemeal. The lesion was diagnosed as a giant cell tumor of tendon sheath. Full function returned and persisted until the present episode. Upon examination, there was ulnar intrinsic atrophy in the hand, and the grip was diminished. Tinel's sign could not be elicited. Electromyographic studies showed poly phasic potentials in the first dorsal interosseous and the abductor digiti quinti muscles, with sensory conduction delayed across the wrist. There was mild hypesthesia in the ulnar distribution. Exploration disclosed a slightly compressible mass producing a I cm bulge into the floor of the ulnar tunnel. The ulnar nerve was splayed out over the mass and pushed palmarward and medially just distal to its division into superficial and deep branches (Fig. I). The solid, yellowish-brown tumor extended laterally to the triquetrum and deep to the hypothenar muscles and was approximately 2 by 3 cm in size. Use of part of the previous dorsal wrist incision allowed complete removal. Histologic examination confirmed the diagnosis of giant cell tumor. Healing was uneventful. One week after operation, sensibility had improved, and at 5 weeks after operation there was no difference, as compared with the left hand. At 10 weeks there was good abductor digiti quinti function, though the first dorsal interosseous was weak. The patient returned to his regular work at this time. Full strength and function were noted at 8 months, and there was no evidence of muscular atrophy. He was asymptomatic I year following the operation.
Guyon,1 in his anatomical studies of ulnar nerve relationships at the wrist, predicted neuropathy by compression in 1861. Hunt2 described the first clinical cases of ulnar compression with symptoms of muscle From the Department of Plastic Surgery, University of Tennessee Clinical Education Center-Chattanooga, and the Hand Service, Baroness Erlanger Hospital. Received for publication March 5, 1977. Reprint requests: Cauley W. Hayes, Jr., M.D., Plastic Surgery Associates of Chattanooga, 1010 E. Third St., Chattanooga, TN 37403.
0363-5023/78/0203-0187$00.20/0
atrophy and weakness. Many causes of ulnar nerve compression at the wrist have been reported, and anatomical observations by McFarlane, Mayer, and HugilP indicated that the most likely site of compression of the ulnar nerve was at its exit from the pisohamate tunnel, where the deep or motor branch of the ulnar nerve passes between the pisohamate ligament and the fibrous arch of origin of the hypothenar muscles. Lesions reported to have caused ulnar neuropathy at the wrist include ganglion,4-10 accessory muscles, ulnar artery thrombosis, 9, 10 palmar fasciitis, 10 palmaris brevis hypertrophy, 10 lipoma,l1, 12 anomalies of the hamate bone,13 and traumatic occupational neuritis. 2 This is thought to be the first reported case of compression by a giant cell tumor (fibrous histiocytoma). Kleinert and Hayes 10 attribute to Dupont et a1. 9 the term "ulnar tunnel syndrome" and preferred it to the more common term "Guyon's canal syndrome." Grantham14 stated that a canal "should have a bed or floor and sides and be unroofed." For that reason he prefers the term "loge." Anatomical studies by Lotem, Gloobe, and Nathan 15 and later by McFarlane, Mayer, and HugilP that the roof of the ulnar tunnel is formed by the volar carpal ligament, its medial wall by the pisiform bone, and its floor by the transverse carpalligament. Both reports pointed out the constant presence of a fascial or fibrotic ring at the exit of the tunnel, where the deep (motor) branch of the ulnar nerve passes deeply into the palm. This fascial or fibrous arch is the origin of the abductor and flexor digiti minimi muscles. In the routine ulnar tunnel decompression, this arch may be divided, thereby decompressing the narrow exit. Shea and McClain8 classified the types of ulnar nerve compression syndromes at the wrist on the basis of the site of involvement into three types: type I, a combined sensory and motor deficit; type II, a motor deficit only; and type III, a sensory deficit only. In their 136 collected cases, 30% were type I; 52% were type II; and 18% were type III. They noticed no particular lesion to be consistently associated with any particular type of syndrome, with the possible exception of ulnar artery arteritis, which more frequently produced type
© 1978 American Society for Surgery of the Hand
THE JOURNAL OF HAND SURGERY
187
188 Hayes
The Journal of HAND SURGERY
Fig. 1. Operative photograph showing the ulnar nerve at its bifurcation with the underlying giant cell tumor.
III lesions. As stated by Lotem, Gloobe, and Nathan,15 the anatomic fibrous arch normally is loose and does not compress the motor branch of the ulnar nerve, but such factors as swelling, edema, scarring, inflamma· tion, or mechanical factors obviously can tilt the balance toward compression . In the present case, motor weakness was the initial complaint, but sensory deficit developed, placing the lesion in Shea and McClain's type I category. Any mass in the narrow confines of the ulnar tunnel could produce a similar compression syndrome. No report in the English literature has been found of a giant cell tumor (fibrous histiocytoma) producing the symptom complex observed in this case. REFERENCES I. Guyon F: Note sur une disposition anatomique propre a la face anterieure de la region du poignet et non encore decrite . Bull Soc Anatomique Paris 6:184, 1861 2. Hunt JR: Occupation neuritis of the deep palmar branch of the ulnar nerve. J Nerv Ment Dis 35:673, 1908 3 . McFarlane RM, Mayer JR, Hugill JV: Further observations on the anatomy of the ulnar nerve at the wrist. Hand 8:115,1976 4. Seddon HJ: Carpal ganglion as a cause of paralysis of the deep branch of the ulnar nerve. J Bone Joint Surg 34B:386, 1952
5. Brooks 0: Nerve compression by simple ganglion. J Bone Joint Surg 34-B:391, 1952 6. Vanderpool OW, Chalmers J, Lamb DW, et al: Peripheral compression lesions of the ulnar nerve. J Bone Joint Surg 50-B:792, 1968 7. Richmond DA: Carpal ganglion with ulnar nerve compression J Bone Joint Surg 45-B:513, 1963 8. Shea JD, McClain EJ: Ulnar nerve compression syndromes at and below the wrist. J Bone Joint Surg 51A: 1095 , 1969 9. Dupont C, Clouthier GE , Prevost Y, et al: Ulnar tunnel syndrome at the wrist. J Bone Joint Surg 47-A:757, 1965 10. Kleinert HE, Hayes JE: The ulnar tunnel syndrome. Plast Reconst Surg 47:21,1971 II. McFarland GB, Hoffer MD: Paralysis of the intrinsic muscles of the hand secondary to lipoma in Guyon's tunnel. J Bone Joint Surg 53-A:375, 1971 12. White WC, Hanna DC: Troublesome lipomata of the upper extremity. J Bone Joint Surg 44:1353, 1962 13 . Fenning 18: Deep ulnar nerve paralysis resulting from an anatomic abnormality . J Bone Joint Surg 47-A:1381, 1965 14. Grantham SA: Ulnar compression in the loge de Guyon. JAMA 197:229, 1966 15. Lotem M, Gloobe H, Nathan H: Fibrotic arch around the deep branch of the ulnar nerve in the hand. Plast Reconst Surg. 52:553, 1973
A
42-year-old Caucasian male warehouse worker had had increasing weakness of the right hand for 6 months. It was accompanied by difficulty with fine movements and a hand thinner in appearance. Two years previously, after several months of mild weakness, an operation on the dorsal ulnar aspect of the wrist had disclosed a solid lesion which was removed piecemeal. The lesion was diagnosed as a giant cell tumor of tendon sheath. Full function returned and persisted until the present episode. Upon examination, there was ulnar intrinsic atrophy in the hand, and the grip was diminished. Tinel's sign could not be elicited. Electromyographic studies showed poly phasic potentials in the first dorsal interosseous and the abductor digiti quinti muscles, with sensory conduction delayed across the wrist. There was mild hypesthesia in the ulnar distribution. Exploration disclosed a slightly compressible mass producing a I cm bulge into the floor of the ulnar tunnel. The ulnar nerve was splayed out over the mass and pushed palmarward and medially just distal to its division into superficial and deep branches (Fig. I). The solid, yellowish-brown tumor extended laterally to the triquetrum and deep to the hypothenar muscles and was approximately 2 by 3 cm in size. Use of part of the previous dorsal wrist incision allowed complete removal. Histologic examination confirmed the diagnosis of giant cell tumor. Healing was uneventful. One week after operation, sensibility had improved, and at 5 weeks after operation there was no difference, as compared with the left hand. At 10 weeks there was good abductor digiti quinti function, though the first dorsal interosseous was weak. The patient returned to his regular work at this time. Full strength and function were noted at 8 months, and there was no evidence of muscular atrophy. He was asymptomatic I year following the operation.
Guyon,1 in his anatomical studies of ulnar nerve relationships at the wrist, predicted neuropathy by compression in 1861. Hunt2 described the first clinical cases of ulnar compression with symptoms of muscle From the Department of Plastic Surgery, University of Tennessee Clinical Education Center-Chattanooga, and the Hand Service, Baroness Erlanger Hospital. Received for publication March 5, 1977. Reprint requests: Cauley W. Hayes, Jr., M.D., Plastic Surgery Associates of Chattanooga, 1010 E. Third St., Chattanooga, TN 37403.
0363-5023/78/0203-0187$00.20/0
atrophy and weakness. Many causes of ulnar nerve compression at the wrist have been reported, and anatomical observations by McFarlane, Mayer, and HugilP indicated that the most likely site of compression of the ulnar nerve was at its exit from the pisohamate tunnel, where the deep or motor branch of the ulnar nerve passes between the pisohamate ligament and the fibrous arch of origin of the hypothenar muscles. Lesions reported to have caused ulnar neuropathy at the wrist include ganglion,4-10 accessory muscles, ulnar artery thrombosis, 9, 10 palmar fasciitis, 10 palmaris brevis hypertrophy, 10 lipoma,l1, 12 anomalies of the hamate bone,13 and traumatic occupational neuritis. 2 This is thought to be the first reported case of compression by a giant cell tumor (fibrous histiocytoma). Kleinert and Hayes 10 attribute to Dupont et a1. 9 the term "ulnar tunnel syndrome" and preferred it to the more common term "Guyon's canal syndrome." Grantham14 stated that a canal "should have a bed or floor and sides and be unroofed." For that reason he prefers the term "loge." Anatomical studies by Lotem, Gloobe, and Nathan 15 and later by McFarlane, Mayer, and HugilP that the roof of the ulnar tunnel is formed by the volar carpal ligament, its medial wall by the pisiform bone, and its floor by the transverse carpalligament. Both reports pointed out the constant presence of a fascial or fibrotic ring at the exit of the tunnel, where the deep (motor) branch of the ulnar nerve passes deeply into the palm. This fascial or fibrous arch is the origin of the abductor and flexor digiti minimi muscles. In the routine ulnar tunnel decompression, this arch may be divided, thereby decompressing the narrow exit. Shea and McClain8 classified the types of ulnar nerve compression syndromes at the wrist on the basis of the site of involvement into three types: type I, a combined sensory and motor deficit; type II, a motor deficit only; and type III, a sensory deficit only. In their 136 collected cases, 30% were type I; 52% were type II; and 18% were type III. They noticed no particular lesion to be consistently associated with any particular type of syndrome, with the possible exception of ulnar artery arteritis, which more frequently produced type
© 1978 American Society for Surgery of the Hand
THE JOURNAL OF HAND SURGERY
187
188 Hayes
The Journal of HAND SURGERY
Fig. 1. Operative photograph showing the ulnar nerve at its bifurcation with the underlying giant cell tumor.
III lesions. As stated by Lotem, Gloobe, and Nathan,15 the anatomic fibrous arch normally is loose and does not compress the motor branch of the ulnar nerve, but such factors as swelling, edema, scarring, inflamma· tion, or mechanical factors obviously can tilt the balance toward compression . In the present case, motor weakness was the initial complaint, but sensory deficit developed, placing the lesion in Shea and McClain's type I category. Any mass in the narrow confines of the ulnar tunnel could produce a similar compression syndrome. No report in the English literature has been found of a giant cell tumor (fibrous histiocytoma) producing the symptom complex observed in this case. REFERENCES I. Guyon F: Note sur une disposition anatomique propre a la face anterieure de la region du poignet et non encore decrite . Bull Soc Anatomique Paris 6:184, 1861 2. Hunt JR: Occupation neuritis of the deep palmar branch of the ulnar nerve. J Nerv Ment Dis 35:673, 1908 3 . McFarlane RM, Mayer JR, Hugill JV: Further observations on the anatomy of the ulnar nerve at the wrist. Hand 8:115,1976 4. Seddon HJ: Carpal ganglion as a cause of paralysis of the deep branch of the ulnar nerve. J Bone Joint Surg 34B:386, 1952
5. Brooks 0: Nerve compression by simple ganglion. J Bone Joint Surg 34-B:391, 1952 6. Vanderpool OW, Chalmers J, Lamb DW, et al: Peripheral compression lesions of the ulnar nerve. J Bone Joint Surg 50-B:792, 1968 7. Richmond DA: Carpal ganglion with ulnar nerve compression J Bone Joint Surg 45-B:513, 1963 8. Shea JD, McClain EJ: Ulnar nerve compression syndromes at and below the wrist. J Bone Joint Surg 51A: 1095 , 1969 9. Dupont C, Clouthier GE , Prevost Y, et al: Ulnar tunnel syndrome at the wrist. J Bone Joint Surg 47-A:757, 1965 10. Kleinert HE, Hayes JE: The ulnar tunnel syndrome. Plast Reconst Surg 47:21,1971 II. McFarland GB, Hoffer MD: Paralysis of the intrinsic muscles of the hand secondary to lipoma in Guyon's tunnel. J Bone Joint Surg 53-A:375, 1971 12. White WC, Hanna DC: Troublesome lipomata of the upper extremity. J Bone Joint Surg 44:1353, 1962 13 . Fenning 18: Deep ulnar nerve paralysis resulting from an anatomic abnormality . J Bone Joint Surg 47-A:1381, 1965 14. Grantham SA: Ulnar compression in the loge de Guyon. JAMA 197:229, 1966 15. Lotem M, Gloobe H, Nathan H: Fibrotic arch around the deep branch of the ulnar nerve in the hand. Plast Reconst Surg. 52:553, 1973