http://informahealthcare.com/mor ISSN 1439-7595 (print), 1439-7609 (online) Mod Rheumatol, 2015; 25(2): 298–302 © 2014 Japan College of Rheumatology DOI: 10.3109/14397595.2013.874753
CASE REPORT
Bilateral carpal tunnel syndrome due to gouty tophi: conservative and surgical treatment in different hands of the same patient Kenji Onuma1, Hisako Fujimaki1, Tomonori Kenmoku1, Koji Sukegawa1, Shotaro Takano1, Kentaro Uchida1, Naonobu Takahira2, and Masashi Takaso1 1Department of Orthopaedic Surgery, Kitasato University School of Medicine, Sagamihara, Kanagawa, Japan 2Department of Rehabilitation, Kitasato University School of Allied Health Sciences, Sagamihara, Kanagawa, Japan
Abstract Gouty tophi are an uncommon cause of carpal tunnel syndrome. We describe a case of bilateral carpal tunnel syndrome due to gouty tophi. Gouty tophi in the right wrist developed slowly, but developed acutely in flexor tendons in the left wrist. Symptoms were numbness and finger movement dysfunction in both hands. The right hand was treated surgically, while the left hand was treated by medication. Both hands improved under a well-controlled serum uremic acid level.
Introduction The most common causes of secondary carpal tunnel syndromes are tenosynovitis in rheumatoid arthritis, malunion of distal radius fracture, and amyloidosis in patients under dialysis. In contrast, carpal tunnel syndrome due to gouty tophi of the flexor tendon is far less common. Phalen [1] reported that only 2 cases in 439 patients with carpal tunnel syndrome (0.4 %) were secondary to gout, while Rich et al. [2] reported cases in 15 hands (0.6 %) which were secondary to gout in a large series of 2,649 cases undergoing carpal tunnel release, of which only 2 cases occurred in bilateral hands (0.08 %) [2]. In this report, we describe a case of bilateral carpal tunnel syndrome due to gouty tophi.
Case report A 39-year-old male, a right-handed truck driver, experienced hyperuricemia of approximately 5 years’ duration, during which time he reported several episodes of acute gout in both ankles. However, treatment for hyperuricemia was intermittent. He had a history of atopic dermatitis which had been treated by dermatologists. He had no family history of hyperuricemia. He presented with several nodes in his extremities and ear auricles, several of which were biopsied and diagnosed as gouty tophi. He had a 1-year history of a slowly enlarging mass on the volar aspect of his right wrist with numbness of the hand (Figure 1). In September 2011, he developed acute-onset swelling and inflammatory reaction in his left forearm and hand resembling acute aseptic cellulitis with the contracture of fingers and numbness over the area supplied by the median nerve. Dermatologists diagnosed this condition as a gout attack or aseptic cellulitis and began treatment with antibiotics. Serum uremic acid level at this time was 10.3 mg/dl (normal Correspondence to: Kenji Onuma, Department of Orthopaedic Surgery, Kitasato University School of Medicine, 1-15-1 Kitasato Minami-ku, Sagamihara, Kanagawa 252-0374, Japan. Tel: +81-42-7789343. Fax:+8142-7785850. E-mail: [email protected]
Keywords Carpal tunnel syndrome, Conservative treatment, Flexor tendon, Gouty tophus, Hyperuricemia History Received 17 December 2012 Accepted 13 March 2013 Published online 29 March 2013
range 3.2–7.2 mg/dl), while serum creatinine and blood urea nitrogen levels were both within normal range. Uremic acid clearance was 6.5 ml/min (normal range 7.3–14.7 ml/min), and urinary the uric acid level was 0.24 mg/kg/h (normal range 0.48–0.51 mg/ kg/h). This hyperuricemia was therefore classified as the urate under excretion type. Treatment for hyperuricemia by allopurinol and benzbromarone was begun from September 2011. Although the acute reaction in his left forearm resolved by 2 weeks from onset, the contracture and numbness of the fingers remained. On biopsy, the lump on the right wrist was diagnosed as a gouty tophus. His dermatologist consulted our orthopedic hand clinic for these hand problems. Bilateral contracture of the fingers and a lump on the wrist were observed (Figure 1). Numbness of the right hand in the area supplied by the median nerve was severe, whereas that of the left hand was improving. Although Tinel’s sign was observed at both wrists, Phalen’s test was not observed because of the limited volar flexion in both wrists. Grip strength of the right and left hands was 9 and 4 kg, respectively. Motor nerve conduction velocity tests revealed delayed distal latency of the right and left median nerves of 6.0 and 7.2 ms, respectively, with low amplitude of the left hand, at 8.11 and 2.81 mV, respectively, and an absent median sensory response in both. Electrodiagnosis was bilateral carpal tunnel syndrome. Ultrasonography of the volar aspect of the wrists showed movement dysfunction of the right flexor tendons compared to the left. Magnetic resonance imaging (MRI) of the right and left wrists showed a diffuse swollen lesion around the flexor digitorum profundus and superficialis tenosynovia bilaterally, and displacement of both median nerves (Figures 2, 3). Computed tomography (CT) imaging showed calcifications in both volar regions. Carpal tunnel release and excision of gouty tophus of the right wrist was done in January 2012, at which time the serum uric acid level was within normal range (6.5 mg/dl). All flexor tendons, synovium, and excursion floor of the flexor tendons were heavily infiltrated with a white chalky substance (Figures 4, 5). Tendons and floor were adherent with each other. Excision of tophi and
Case report 299
DOI 10.3109/14397595.2013.874753
Figure 1. The patient’s hands on initial visit to our orthopedic hand clinic. Both volar wrists were swollen (white triangles) (a, b full active extension of fingers; c, d full active flexion of fingers. White arrow indicates nodule in the proximal interphalangeal joint of the index finger)
tendolysis was done for contracture of fingers. The transverse carpal ligament was divided to decompress the median nerve at the carpal tunnel. Conservative treatment by medication and rehabilitation was continued for both hands. At postoperative month 6, the patient did not complain of numbness in either hand. The lumps on both volar wrists had resolved. Although contracture of the fingers of both hands improved, extension remained disturbed in the right hand (Figure 6). Grip strength of the right and left hands improved to 15.7 and 21.8 kg, respectively, while motor nerve conduction velocity tests showed improved distal latency in both right and left median nerves to 4.9 and 4.3 ms, and improved amplitude to 7.51 and 11.21 mV, respectively. Following treatment with allopurinol (200 mg/day) and benzbromarone (50 mg/day) for 1 year (postoperative month 9; September 2012), serum uremic acid level
was 3.9 mg. Medication is ongoing as of writing. No gout attack occurred between the first and last observations.
Discussion Gouty tophi can occur in the ear auricle, Achilles tendon, olecranon bursa, eye, nose, bronchi, heart muscle, small intestine, penis, buttocks, fingers and pharynx. Although the pathogenesis of gouty tophi is still unknown, their conspicuous absence from skeletal muscle, liver, spleen, lungs, and nerve tissue suggests that they are more likely to be derived from avascular than vascular tissues, such as cartilage, epiphyseal bone, synovial membrane, bursa, ligaments, and tendons [3]. In their characterization of carpal tunnel syndrome caused by tophaceous gout on MRI and CT imaging, Chen et al. [4] reported that tophi showed from
300
K. Onuma et al.
Mod Rheumatol, 2015; 25(2): 298–302
Figure 2. Sagittal T1-weighted fat saturated post-gadolinium (TR = 250 ms, TE = 5 ms) image at the wrist (a right wrist, b left wrist). Diffuse lesions are shown around the flexor digitorum superficialis and profundus extending into the carpal tunnel in both wrists
low to intermediate signal intensity on T1-weighted images with heterogeneous signal intensity on T2-weighted images; heterogeneous enhancement on gadolinium-enhanced MR imaging; and varying degrees of calcification on CT imaging. In our case, although pathological diagnosis was done for the right wrist region but not the left wrist, the 2 regions showed similar characteristics in MRI and CT imaging, and both were diagnosed as having gouty tophus in the flexor tendons. A consensus on therapeutic strategies for the treatment of carpal tunnel syndrome due to gouty tophi has yet to be obtained. Most case reports of carpal tunnel syndrome due to gouty tophus have described the availability of surgical treatment in the chronic phase [2, 5–8]. Several authors have reported complications of surgical treatment. Ogilvie and Kay [9] reported a case undergoing carpal tunnel release for acute-phase gout under an uncontrolled uric acid level, and noted that the patient had complete relief of pain but continued to discharge gouty material from the wound for 6 weeks; while Pai and Tseng [10] described a case of an attack of gout following carpal tunnel release under uncontrolled hyperuricemia. In our case, these complications were not encountered, possibly because surgical treatment for the right wrist was performed under a well-controlled uric acid level. These complications should be considered in surgical planning for these patients.
Figure 3. Axial T1-weighted fat saturated post-gadolinium (TR = 250 ms, TE = 5 ms) image at the level of the distal radioulnar joint (a right wrist, b left wrist). Diffuse lesions occur widely around the flexor digitorum superficialis and profundus, and compress the median nerve in both wrists (white arrows)
In contrast, some authors have described conservative treatment by medication for mild cases [11–13]. In our case, as the right hand had severe contracture of the fingers and numbness by slowly growing gouty tophus whereas the left had only mild contracture with improving numbness following an acute gout attack, our initial plan was to conduct surgery on the right wrist and consider the left wrist at a later stage. However, he had not complained of numbness in the left hand following medication for hyperuricemia for 4 months and the contracture of the fingers was improving, and although electrodiagnosis showed that carpal tunnel syndrome was more severe on the left than on right, we decided to continue conservative treatment for the left wrist. Subsequently, contracture of the fingers and the disturbance of flexion were significantly improved at last observation compared to that at his first visit to our hand surgery clinic. Moreover, the wrist lump and finger nodule in the proximal interphalangeal joint of the index finger of left hand had both shrunk (Figure 6). From this clinical course, we suggest a wait-and-see approach for patients with mild numbness and contracture and the continuation of medication for hyperuricemia. With regard to the surgery for his right wrist, although the numbness, contracture of the fingers and grip strength were improved compared to the preoperative status, the disturbance of finger extension remained at 6 months after surgery, which we considered due to
DOI 10.3109/14397595.2013.874753
Case report 301
Figure 5. Gouty tophi and synovia removed from the tendons
Figure 4. Operative findings. The flexor digitorum superficialis is surrounded by chalky white tissue which forms adhesions with the tendons
Figure 6. The patient’s right and left hands at final observation 6 months after surgery (a, b full active extension of fingers; c, d full active flexion of fingers). Both hands show improved range of motion. The swollen volar aspect of the wrists has decreased in size. The nodule in the proximal interphalangeal joint of the left index finger is smaller than in Figure 1 (white arrow)
the decreased elasticity of tendons following severe urate crystal infiltration. Moore and Weiland [7] suggested that the approach to restoration of tendon function should be similar to that in reconstruction of rheumatoid tendon rupture, which utilizes a side-to-side tendon suture, tendon transfer, and occasionally a bridge graft. Lin et al. [ 8 ] utilized resection of the flexor digitorum superficialis tendons to facilitate function of the flexor digitorum profundus tendons. In our case, however, both flexor tendons were heavily infiltrated, and this approach
302
K. Onuma et al.
was not feasible. Contracture under these conditions might be permanent despite continued medication and rehabilitation. Although further surgery to restore tendon function is indicated, our patient has declined additional surgery. Several authors reported that extensor or flexor tendon rupture also occurred as a result of tendon degeneration due to extensive tophaceous deposits [7, 14]. Early diagnosis and control of serum uric acid level might have prevented heavy infiltration of urate crystals into tendons and resulted in the preservation of hand function. In their series of 20 patients with carpal tunnel syndrome due to tophi (carpal tunnel releases for 12 patients and medication only for 8 patients), Chan et al. [4] reported that persistent or recurrent carpal tunnel syndrome was present in 3 wrists. We consider that ongoing control of uric acid level will prevent the recurrence of carpal tunnel syndrome. In conclusion, although surgery will generally provide an immediate improvement in excursion of the flexor tendons and severe sensory loss, surgeons should bear in mind that medication for hyperuricemia is one option for the treatment of carpal tunnel syndrome due to gout, and is important in the perioperative period. Nevertheless, early diagnosis and control of the serum uric acid level is more important than surgery in avoiding permanent dysfunction.
Conflict of interest None.
References 1. Phalen GS. The carpal-tunnel syndrome. Seventeen years’ experience in diagnosis and treatment of six hundred fifty-four hands. J Bone Jt Surg. 1966;48-A:211–28.
Mod Rheumatol, 2015; 25(2): 298–302
2. Rich JT, Bush DC, Lincoski CJ, Harrington TM. Carpal tunnel syndrome due to tophaceous gout. Orthopedics. 2004;27: 862–3. 3. Straub LR, Smith JW. The surgery of gout in the upper extremity. J Bone Jt Surg. 1961;43-A:731–74. 4. Chen CK, Chung CB, Yeh L, Pan HB, Yang CF, Lai PH et al. Carpal tunnel syndrome caused by tophaceous gout: CT and MR imaging features in 20 patients. Am J Roentgenol. 2000;175: 655–9. 5. Hernández-Cortés P, Caba M, Gómez-Sánchez R, Gómez-Morales M. Digital flexion contracture and severe carpal tunnel syndrome due to tophaceous infiltration of wrist flexor tendon: first manifestation of gout. Orthopedics. 2011;34:e797–9. 6. Therimadasamy A, Peng YP, Putti TC, Wilder-Smith EP. Carpal tunnel syndrome caused by gouty tophus of the flexor tendons of the fingers: sonographic features. J Clin Ultrasound. 2011;39: 463–5. 7. Moore JR, Weiland AJ. Gouty tenosynovitis in the hand. J Hand Surg. 1985;10-A:291–5. 8. Lin YC, Chen CH, Fu YC, Lin GT, Chang JK, Hu ST. Carpal tunnel syndrome and finger movement dysfunction caused by tophaceous gout: a case report. Kaohsiung J Med Sci. 2009;25: 34–9. 9. Ogilvie C, Kay NRM. Fulminating carpal tunnel syndrome due to gout. J Hand Surg. 1987;13-B:42–3. 10. Pai CH, Tseng CH. Acute carpal tunnel syndrome caused by tophaceous gout. J Hand Surg. 1993;18-A: 667–9. 11. O’Hara LJ, Levin M. Carpal tunnel syndrome and gout. Arch Intern Med. 1967;120:180–4. 12. Champion D. Gouty tenosynovitis and the carpal tunnel syndrome. Med J Aust. 1969;17:1030–2. 13. Fujimoto WY, Seegmiller JE. Medical treatment of compression neuropathy of median and ulnar nerves in gout. Arth Rheum. 1968;11:99–100. 14. Iwamoto T, Toki H, Iraki K, Yamanaka H, Momohara S. Multiple extensor tendon rupture caused by tophaceous gout. Mod Rheumatol. 2010;20:210–2.
Copyright of Modern Rheumatology (Informa Healthcare) is the property of Informa Healthcare and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
CASE REPORT
Bilateral carpal tunnel syndrome due to gouty tophi: conservative and surgical treatment in different hands of the same patient Kenji Onuma1, Hisako Fujimaki1, Tomonori Kenmoku1, Koji Sukegawa1, Shotaro Takano1, Kentaro Uchida1, Naonobu Takahira2, and Masashi Takaso1 1Department of Orthopaedic Surgery, Kitasato University School of Medicine, Sagamihara, Kanagawa, Japan 2Department of Rehabilitation, Kitasato University School of Allied Health Sciences, Sagamihara, Kanagawa, Japan
Abstract Gouty tophi are an uncommon cause of carpal tunnel syndrome. We describe a case of bilateral carpal tunnel syndrome due to gouty tophi. Gouty tophi in the right wrist developed slowly, but developed acutely in flexor tendons in the left wrist. Symptoms were numbness and finger movement dysfunction in both hands. The right hand was treated surgically, while the left hand was treated by medication. Both hands improved under a well-controlled serum uremic acid level.
Introduction The most common causes of secondary carpal tunnel syndromes are tenosynovitis in rheumatoid arthritis, malunion of distal radius fracture, and amyloidosis in patients under dialysis. In contrast, carpal tunnel syndrome due to gouty tophi of the flexor tendon is far less common. Phalen [1] reported that only 2 cases in 439 patients with carpal tunnel syndrome (0.4 %) were secondary to gout, while Rich et al. [2] reported cases in 15 hands (0.6 %) which were secondary to gout in a large series of 2,649 cases undergoing carpal tunnel release, of which only 2 cases occurred in bilateral hands (0.08 %) [2]. In this report, we describe a case of bilateral carpal tunnel syndrome due to gouty tophi.
Case report A 39-year-old male, a right-handed truck driver, experienced hyperuricemia of approximately 5 years’ duration, during which time he reported several episodes of acute gout in both ankles. However, treatment for hyperuricemia was intermittent. He had a history of atopic dermatitis which had been treated by dermatologists. He had no family history of hyperuricemia. He presented with several nodes in his extremities and ear auricles, several of which were biopsied and diagnosed as gouty tophi. He had a 1-year history of a slowly enlarging mass on the volar aspect of his right wrist with numbness of the hand (Figure 1). In September 2011, he developed acute-onset swelling and inflammatory reaction in his left forearm and hand resembling acute aseptic cellulitis with the contracture of fingers and numbness over the area supplied by the median nerve. Dermatologists diagnosed this condition as a gout attack or aseptic cellulitis and began treatment with antibiotics. Serum uremic acid level at this time was 10.3 mg/dl (normal Correspondence to: Kenji Onuma, Department of Orthopaedic Surgery, Kitasato University School of Medicine, 1-15-1 Kitasato Minami-ku, Sagamihara, Kanagawa 252-0374, Japan. Tel: +81-42-7789343. Fax:+8142-7785850. E-mail: [email protected]
Keywords Carpal tunnel syndrome, Conservative treatment, Flexor tendon, Gouty tophus, Hyperuricemia History Received 17 December 2012 Accepted 13 March 2013 Published online 29 March 2013
range 3.2–7.2 mg/dl), while serum creatinine and blood urea nitrogen levels were both within normal range. Uremic acid clearance was 6.5 ml/min (normal range 7.3–14.7 ml/min), and urinary the uric acid level was 0.24 mg/kg/h (normal range 0.48–0.51 mg/ kg/h). This hyperuricemia was therefore classified as the urate under excretion type. Treatment for hyperuricemia by allopurinol and benzbromarone was begun from September 2011. Although the acute reaction in his left forearm resolved by 2 weeks from onset, the contracture and numbness of the fingers remained. On biopsy, the lump on the right wrist was diagnosed as a gouty tophus. His dermatologist consulted our orthopedic hand clinic for these hand problems. Bilateral contracture of the fingers and a lump on the wrist were observed (Figure 1). Numbness of the right hand in the area supplied by the median nerve was severe, whereas that of the left hand was improving. Although Tinel’s sign was observed at both wrists, Phalen’s test was not observed because of the limited volar flexion in both wrists. Grip strength of the right and left hands was 9 and 4 kg, respectively. Motor nerve conduction velocity tests revealed delayed distal latency of the right and left median nerves of 6.0 and 7.2 ms, respectively, with low amplitude of the left hand, at 8.11 and 2.81 mV, respectively, and an absent median sensory response in both. Electrodiagnosis was bilateral carpal tunnel syndrome. Ultrasonography of the volar aspect of the wrists showed movement dysfunction of the right flexor tendons compared to the left. Magnetic resonance imaging (MRI) of the right and left wrists showed a diffuse swollen lesion around the flexor digitorum profundus and superficialis tenosynovia bilaterally, and displacement of both median nerves (Figures 2, 3). Computed tomography (CT) imaging showed calcifications in both volar regions. Carpal tunnel release and excision of gouty tophus of the right wrist was done in January 2012, at which time the serum uric acid level was within normal range (6.5 mg/dl). All flexor tendons, synovium, and excursion floor of the flexor tendons were heavily infiltrated with a white chalky substance (Figures 4, 5). Tendons and floor were adherent with each other. Excision of tophi and
Case report 299
DOI 10.3109/14397595.2013.874753
Figure 1. The patient’s hands on initial visit to our orthopedic hand clinic. Both volar wrists were swollen (white triangles) (a, b full active extension of fingers; c, d full active flexion of fingers. White arrow indicates nodule in the proximal interphalangeal joint of the index finger)
tendolysis was done for contracture of fingers. The transverse carpal ligament was divided to decompress the median nerve at the carpal tunnel. Conservative treatment by medication and rehabilitation was continued for both hands. At postoperative month 6, the patient did not complain of numbness in either hand. The lumps on both volar wrists had resolved. Although contracture of the fingers of both hands improved, extension remained disturbed in the right hand (Figure 6). Grip strength of the right and left hands improved to 15.7 and 21.8 kg, respectively, while motor nerve conduction velocity tests showed improved distal latency in both right and left median nerves to 4.9 and 4.3 ms, and improved amplitude to 7.51 and 11.21 mV, respectively. Following treatment with allopurinol (200 mg/day) and benzbromarone (50 mg/day) for 1 year (postoperative month 9; September 2012), serum uremic acid level
was 3.9 mg. Medication is ongoing as of writing. No gout attack occurred between the first and last observations.
Discussion Gouty tophi can occur in the ear auricle, Achilles tendon, olecranon bursa, eye, nose, bronchi, heart muscle, small intestine, penis, buttocks, fingers and pharynx. Although the pathogenesis of gouty tophi is still unknown, their conspicuous absence from skeletal muscle, liver, spleen, lungs, and nerve tissue suggests that they are more likely to be derived from avascular than vascular tissues, such as cartilage, epiphyseal bone, synovial membrane, bursa, ligaments, and tendons [3]. In their characterization of carpal tunnel syndrome caused by tophaceous gout on MRI and CT imaging, Chen et al. [4] reported that tophi showed from
300
K. Onuma et al.
Mod Rheumatol, 2015; 25(2): 298–302
Figure 2. Sagittal T1-weighted fat saturated post-gadolinium (TR = 250 ms, TE = 5 ms) image at the wrist (a right wrist, b left wrist). Diffuse lesions are shown around the flexor digitorum superficialis and profundus extending into the carpal tunnel in both wrists
low to intermediate signal intensity on T1-weighted images with heterogeneous signal intensity on T2-weighted images; heterogeneous enhancement on gadolinium-enhanced MR imaging; and varying degrees of calcification on CT imaging. In our case, although pathological diagnosis was done for the right wrist region but not the left wrist, the 2 regions showed similar characteristics in MRI and CT imaging, and both were diagnosed as having gouty tophus in the flexor tendons. A consensus on therapeutic strategies for the treatment of carpal tunnel syndrome due to gouty tophi has yet to be obtained. Most case reports of carpal tunnel syndrome due to gouty tophus have described the availability of surgical treatment in the chronic phase [2, 5–8]. Several authors have reported complications of surgical treatment. Ogilvie and Kay [9] reported a case undergoing carpal tunnel release for acute-phase gout under an uncontrolled uric acid level, and noted that the patient had complete relief of pain but continued to discharge gouty material from the wound for 6 weeks; while Pai and Tseng [10] described a case of an attack of gout following carpal tunnel release under uncontrolled hyperuricemia. In our case, these complications were not encountered, possibly because surgical treatment for the right wrist was performed under a well-controlled uric acid level. These complications should be considered in surgical planning for these patients.
Figure 3. Axial T1-weighted fat saturated post-gadolinium (TR = 250 ms, TE = 5 ms) image at the level of the distal radioulnar joint (a right wrist, b left wrist). Diffuse lesions occur widely around the flexor digitorum superficialis and profundus, and compress the median nerve in both wrists (white arrows)
In contrast, some authors have described conservative treatment by medication for mild cases [11–13]. In our case, as the right hand had severe contracture of the fingers and numbness by slowly growing gouty tophus whereas the left had only mild contracture with improving numbness following an acute gout attack, our initial plan was to conduct surgery on the right wrist and consider the left wrist at a later stage. However, he had not complained of numbness in the left hand following medication for hyperuricemia for 4 months and the contracture of the fingers was improving, and although electrodiagnosis showed that carpal tunnel syndrome was more severe on the left than on right, we decided to continue conservative treatment for the left wrist. Subsequently, contracture of the fingers and the disturbance of flexion were significantly improved at last observation compared to that at his first visit to our hand surgery clinic. Moreover, the wrist lump and finger nodule in the proximal interphalangeal joint of the index finger of left hand had both shrunk (Figure 6). From this clinical course, we suggest a wait-and-see approach for patients with mild numbness and contracture and the continuation of medication for hyperuricemia. With regard to the surgery for his right wrist, although the numbness, contracture of the fingers and grip strength were improved compared to the preoperative status, the disturbance of finger extension remained at 6 months after surgery, which we considered due to
DOI 10.3109/14397595.2013.874753
Case report 301
Figure 5. Gouty tophi and synovia removed from the tendons
Figure 4. Operative findings. The flexor digitorum superficialis is surrounded by chalky white tissue which forms adhesions with the tendons
Figure 6. The patient’s right and left hands at final observation 6 months after surgery (a, b full active extension of fingers; c, d full active flexion of fingers). Both hands show improved range of motion. The swollen volar aspect of the wrists has decreased in size. The nodule in the proximal interphalangeal joint of the left index finger is smaller than in Figure 1 (white arrow)
the decreased elasticity of tendons following severe urate crystal infiltration. Moore and Weiland [7] suggested that the approach to restoration of tendon function should be similar to that in reconstruction of rheumatoid tendon rupture, which utilizes a side-to-side tendon suture, tendon transfer, and occasionally a bridge graft. Lin et al. [ 8 ] utilized resection of the flexor digitorum superficialis tendons to facilitate function of the flexor digitorum profundus tendons. In our case, however, both flexor tendons were heavily infiltrated, and this approach
302
K. Onuma et al.
was not feasible. Contracture under these conditions might be permanent despite continued medication and rehabilitation. Although further surgery to restore tendon function is indicated, our patient has declined additional surgery. Several authors reported that extensor or flexor tendon rupture also occurred as a result of tendon degeneration due to extensive tophaceous deposits [7, 14]. Early diagnosis and control of serum uric acid level might have prevented heavy infiltration of urate crystals into tendons and resulted in the preservation of hand function. In their series of 20 patients with carpal tunnel syndrome due to tophi (carpal tunnel releases for 12 patients and medication only for 8 patients), Chan et al. [4] reported that persistent or recurrent carpal tunnel syndrome was present in 3 wrists. We consider that ongoing control of uric acid level will prevent the recurrence of carpal tunnel syndrome. In conclusion, although surgery will generally provide an immediate improvement in excursion of the flexor tendons and severe sensory loss, surgeons should bear in mind that medication for hyperuricemia is one option for the treatment of carpal tunnel syndrome due to gout, and is important in the perioperative period. Nevertheless, early diagnosis and control of the serum uric acid level is more important than surgery in avoiding permanent dysfunction.
Conflict of interest None.
References 1. Phalen GS. The carpal-tunnel syndrome. Seventeen years’ experience in diagnosis and treatment of six hundred fifty-four hands. J Bone Jt Surg. 1966;48-A:211–28.
Mod Rheumatol, 2015; 25(2): 298–302
2. Rich JT, Bush DC, Lincoski CJ, Harrington TM. Carpal tunnel syndrome due to tophaceous gout. Orthopedics. 2004;27: 862–3. 3. Straub LR, Smith JW. The surgery of gout in the upper extremity. J Bone Jt Surg. 1961;43-A:731–74. 4. Chen CK, Chung CB, Yeh L, Pan HB, Yang CF, Lai PH et al. Carpal tunnel syndrome caused by tophaceous gout: CT and MR imaging features in 20 patients. Am J Roentgenol. 2000;175: 655–9. 5. Hernández-Cortés P, Caba M, Gómez-Sánchez R, Gómez-Morales M. Digital flexion contracture and severe carpal tunnel syndrome due to tophaceous infiltration of wrist flexor tendon: first manifestation of gout. Orthopedics. 2011;34:e797–9. 6. Therimadasamy A, Peng YP, Putti TC, Wilder-Smith EP. Carpal tunnel syndrome caused by gouty tophus of the flexor tendons of the fingers: sonographic features. J Clin Ultrasound. 2011;39: 463–5. 7. Moore JR, Weiland AJ. Gouty tenosynovitis in the hand. J Hand Surg. 1985;10-A:291–5. 8. Lin YC, Chen CH, Fu YC, Lin GT, Chang JK, Hu ST. Carpal tunnel syndrome and finger movement dysfunction caused by tophaceous gout: a case report. Kaohsiung J Med Sci. 2009;25: 34–9. 9. Ogilvie C, Kay NRM. Fulminating carpal tunnel syndrome due to gout. J Hand Surg. 1987;13-B:42–3. 10. Pai CH, Tseng CH. Acute carpal tunnel syndrome caused by tophaceous gout. J Hand Surg. 1993;18-A: 667–9. 11. O’Hara LJ, Levin M. Carpal tunnel syndrome and gout. Arch Intern Med. 1967;120:180–4. 12. Champion D. Gouty tenosynovitis and the carpal tunnel syndrome. Med J Aust. 1969;17:1030–2. 13. Fujimoto WY, Seegmiller JE. Medical treatment of compression neuropathy of median and ulnar nerves in gout. Arth Rheum. 1968;11:99–100. 14. Iwamoto T, Toki H, Iraki K, Yamanaka H, Momohara S. Multiple extensor tendon rupture caused by tophaceous gout. Mod Rheumatol. 2010;20:210–2.
Copyright of Modern Rheumatology (Informa Healthcare) is the property of Informa Healthcare and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
