Infection DOI 10.1007/s15010-015-0741-6
REVIEW
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons than infectious diseases specialists Emmanuelle Weber · Amandine Gagneux‑Brunon · V. Jacomo · Thibault Rousselon · Frederic Lucht · Elisabeth Botelho‑Nevers
Received: 10 November 2014 / Accepted: 2 February 2015 © Springer-Verlag Berlin Heidelberg 2015
Abstract Purpose Clinical presentation of tuberculosis is pleomorphic. Some forms are rare and better known by surgeons than infectious disease specialists. Methods We describe a rare case of isolated chronic tenosynovitis of the wrist due to Mycobacterium tuberculosis in a 66-year-old man and review similar cases in the literature. Results On literature search, only 23 other cases of tuberculous tenosynovitis were retrieved. Our case is similar, with an insidious classical presentation. The diagnosis was suggested at the surgical presentation by the presence of rice body masses. Conclusion The diagnosis of tuberculous tenosynovitis should be considered in chronic tenosynovitis. Functional prognosis may be committed without adequate treatment. Keywords Tenosynovitis · Wrist · Mycobacterium tuberculosis · Tuberculosis
in high income countries but prevalence remains high due to HIV infection, migrants, immunosuppression and aging [1]. Tuberculosis is the most common cause of death from infectious diseases world-wide [1]. Extra-pulmonary tuberculosis accounts for 15–30 % of cases [2] with musculoskeletal involvement in only 10–15 % of the cases mainly represented by bone and joint localizations [3]. Tuberculous tenosynovitis is therefore a rare form of musculoskeletal tuberculosis [4]. Indeed tenosynovitis is more frequently due to non tuberculous mycobacteria [5]. Tuberculous tenosynovitis may result in bone and tendon destructions, in particular when the treatment is delayed [4]. Consequently, it is crucial to determine the causative agent to treat the patients, as antibiotic regimens differ between tuberculous and non tuberculous mycobacteria [5, 6]. We describe here a rare case of isolated chronic tenosynovitis due to Mycobacterium tuberculosis in a 66-year-old man and review the literature.
Introduction Case report Tuberculosis remains challenging in diagnosis and treatment. The incidence of tuberculosis is decreasing especially E. Weber · A. Gagneux‑Brunon · F. Lucht · E. Botelho‑Nevers (*) Infectious Diseases Department, University Hospital, 42055 Saint‑Etienne Cedex 2, France e-mail: [email protected] V. Jacomo Biomnis Laboratory, 19 Avenue Tony Garnier, 69007 Lyon, France T. Rousselon Department of Orthopaedic Surgery, Private Hospital of Loire, 42030 Saint‑Etienne Cedex 2, France
A 66-year-old Algerian man was admitted in orthopaedic surgery department in March 2013 for a painful swelling of the right wrist gradually evolving for 6 months. He arrived in France 2 years ago. He had a past history of carpal tunnel surgery of the right hand few years ago in Algeria. He also had an idiopathic adrenal insufficiency diagnosed in 2004, and was treated with hydrocortisone 30 mg daily. No other complaints were reported. Physical examination revealed a cystic mass over the flexor tendons of the right wrist. His thenar eminence was swelling and inflammatory. He did not report paresthesia or sensory deficit. Laboratory values showed C reactive protein 16 mg/L and leucocytes count 9.3 g/L (56 % neutrophils, 32 % lymphocytes). X-ray of
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stopped. Subsequently, isoniazid, rifampicin and pyrazinamide were continued for 1 month then only rifampicin and isoniazid were continued for 7 months and stopped with a good evolution.
Discussion
Fig. 1 Macroscopic findings at surgical tenosynovectomy
hand and wrist showed arthrosis of the first proximal interphalangeal and metacarpophalangeal joint and thickening of the soft parts of thenar eminence. Ultrasound confirmed a large synovitis of long flexor of the thumb. Magnetic resonance imaging of the right wrist and hand revealed synovial thickening of flexor thumb sheath showing high signal intensity on T2-weighted image. It was associated with a 12 cm heterogen fluid collection in the thumb sheath and a second cystic area over the carpal tunnel and the flexor of fourth and fifth fingers. In May 2013 a surgical tenosynovectomy and extensive debridement were performed. Macroscopic examination revealed multiple rice body masses (Fig. 1). The Ziehl-Neelsen staining of this material did not show any acid resistant bacteria. On histopathologic examination, epidermic cyst and granulomatous inflammation with giant cells were observed. A mycobacterial strain grew on Bactect MGIT (Becton–Dickinson®) liquid agar, identified as Mycobacterium tuberculosis by polymerase chain reaction. Identification was performed by PCR: after DNA extraction by heat shock and sonication, amplification was made on thermocycler Gene Amp 2740 (Applied Biosystems®) with primers from Genotype MTBC kit (Hain®), followed by hybrizidation DNA–DNA on Genotype MTBc strips (Hain®). The strain of M. tuberculosis was susceptible to all-first line anti-TB agents. He was therefore addressed to our department for treatment. Three samples of sputum and urine were negative on direct examination and in culture for Mycobacterium tuberculosis. Tuberculin skin test was not performed. Thoraco abdomino pelvic CT scan showed bilateral adrenal hypertrophy with bilateral linear calcifications. No signs of pulmonary tuberculosis were found. In July 2013 an antituberculous therapy consisting of four drugs was started with isoniazid (300 mg daily), rifampicin (720 mg daily), dexambutol (1,500 mg daily) and pyrazinamide (1,800 mg daily) associated with hydrocortisone and fludrocortisone for his adrenal deficiency. At day 10 the patient developed a skin rash due to ethambutol that was
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We reported here a rare case of M. tuberculosis tenosynovitis. Indeed, among osteo-articular localizations of tuberculosis, tenosynovitis is rarely reported. In a series of osteoarticular TB, tenosynovitis represented 1 % of these forms, pott’s disease (50 %), peripheral arthritis (30 %) and osteomyelitis (19 %) being the most common osteoarticular TB presentation [7]. Non tuberculous Mycobacteria (NTM) are, by contrast, more frequently responsible for tenosynovitis and their incidence is increasing [5]. If the clinical presentations of NTM synovitis are similar to those of tuberculous ones, the treatment and the management are different showing the importance of a correct identification of the specie. Some difference could help to distinguish both infections [5, 6]. In NTM tenosynovitis, infection is usually acquired by direct inoculation (skin injury or trauma) of the pathogen from an environmental source or non-apparent inoculation (water contamination). The main causative agents are M. marinum, usually secondary to fishing injuries and M. kansasii [5, 6]. By contrast tuberculous tenosynovitis occurs most often secondary to hematogenous spread to the affected joint [5]. Our patient underwent a surgery for carpal tunnel few years ago. It should be account as source of the tuberculosis although, to the best of our knowledge, tuberculosis has been never reported due to a direct inoculation in a nosocomial context. In our case, we may consider that the patient had a second localization of tuberculosis disease but not yet active. Indeed he had chronic adrenal insufficiency with adrenal calcifications on CT-scan. This aspect although not specific of tuberculosis infection [8] was in this context a posteriori considered due to tuberculosis. His tenosynovitis was probably secondary to hematogenous spread. In NTM tenosynovitis the delay in diagnosis seems to be longer: about 1 month to 3 years. In M. tuberculosis tenosynovitis the duration varied from 10 days to 1 year [5]. The clinical course of NTM tenosynovitis is typically protracted but not destructive whereas M. tuberculosis tenosynovitis can lead to bone destructions if untreated [6]. To better characterize tuberculous tenosynovitis we performed a literature search in the medline database and using “tenosynovitis tuberculosis”, “Mycobacterium tuberculosis tenosynovitis” and “tuberculous tenosynovitis” as search terms. Tenosynovitis due to non tuberculous mycobacteria was excluded. By this literature search only 23 cases of tuberculous tenosynovitis were identified. Most cases are reported by surgeons. The
Sex/age (year)
F/53
F/74
F/58
F/34
M/21
M/60
M/21
M/44
M/55
M/45
First author [Ref]
Wanebo [9]
Asaka [10]
Walker [11]
Aboudola [12]
Huang [13]
Le Meur [14]
Komurcu [15]
Amine [16]
Varshney [17]
Jira [18]
Biological findings
Wrist
Wrist
Wrist
Wrist
Hand, forearm
Elbow
Painless swelling/ 1 year
Pain and swelling, fusiform mass/6 months
foot
Achilles tendon
ND
ND
Nl
ND
ND
MRI: tenosynovitis
ND
MRI: soft tissue with ND intensity lesions with liquefaction and necrosis with tenosynovial proliferation; CXR: nl
Ultrasonography : heterogeneous images
Synovial punction
MTB isolated from joint specimen type Excision
Surgical treatment
Synovial punction
Epithelioid and giant Surgical cell granuloma biopsy with caseation
Caseated inflammation
TST+, AFB+, Non specific chronic Synovial culture+ synovitis, rice fluid bodies
AFB−, PCR+, Granuloma with Punch culture giant cells and biopsy central caseation
Culture−
AFB−, culture+
AFB+, PCR+, Granulomas of Synovial culture+ epithelioide cells biopsy with multiple giant cells with central caseaous necrosis
Granulomatous with Surgical giant cell and case- biopsy ous necrosis, rice bodies
9 months − I + R + E
2 months − I + R + E + P 7 months − I + R
ND
ND − R + STR ND − R + E + STR
6 months − STR ND − I + PA
Duration -antibiotics
Not realized
Not realized
Not realized
2 months − I + R + P 4 months − I + R
3 months − I + R + E + P 9 months − I + R
2 months − I + R + P 7 months − I + R
Debridement, 2 months − I + R + E + P synovec10 months − I + R tomy, carpectomy
Synovectomy 1 month − I + R + E + P 6 months − R + E + P
Excision
Not realized
Surgical TST+, AFB+, Non caseating biopsy culture+ granulomas with epithelioid histiocytes Culture+
Not realized
Necrotising Synovial granulomas with fluid langerhan’s cells
TST+, AFB−, Non specific inflam- Aspiration biopsy Not realized PCR+, matory granuspecimen culture− lomas
Pulmonary culture+ tuberculous at 16 years
Scintiscan: Heart hyperfixation at transearly vascular times plant
MRI : nodes; CXR: nl
CXR: nl
ND
Bacteriological Histological findings findings
Pott TST+, AFB−, Granulomatous disease culture+ inflammation
Context
ND MRI, Ctscan: absecess; thoracic scan: infiltration of the right upper lobe; CXR: nl
ND
Radiological findings
Increased MRI : extensive ND CRP 25 mg/l inflamatory activity
ND
ND
ND
ND
Elevated ERS
Wrist, hand, Nl back
Involved joints
Inflammatory pain and Wrist, ankle, Nl swelling/15 months elbow
Pain, swelling, mass at the extensor area/6 months
Swelling, pain/ 6 months
Mass /2 years
Recurrent swelling/2 months
Painful swelling/6 months
Mass, swelling and tenderness/ND
Painful swelling and masses, back pain/3 years
Symptoms/evolution
Table 1 Description of tenosynovitis tuberculosis published cases
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons
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M/57
Chavan [28]
Wrist
Three swellings mobile/3 years
Wrist
Painful swelling, Hand diminished range of motion/6 months
Painless swelling/5 months
Wrist
M/32
Forearm, elbow
Probst [27]
Pain and swelling/3 months
M/46
M/19
Singh [23]
Mass lesion with swell- Wrist ing/4 months
Mrabet [26]
M/24
Higuchi [22]
ND
Nl
ND
Biological findings
ND
Elevated ERS
Nl
Nl
Nl
Elevated ERS
Nl
Swelling, pain/5 years Thigh, wrist Elevated ERS
Soft tissue nods, pain/5 years
F/68
Batra [21]
Pain, swelling, nights Ankle sweats, weight loss, erythema/3 months
Achilles tendon
F/56
F/53
Ogut [20]
Diminished range of motion, swelling, pain/5 months
Hand, wrist
Hung [25]
F/19
Ogut [20]
Pain and swelling/1 month
Involved joints
Node and carpal tunnel Hand, wrist syndrome/6 years
F/58
Munoz [19]
Symptoms/evolution
Marques [24] F/60
Sex/age (year)
First author [Ref]
Table 1 continued
Kidney transplant
Context
DM; RA
ND
ND
MRI: extraskeletal synovial Koch’s or giant cell tumor of tendon sheath
ND
MRI: multiple lobu- ND lated cystic lesion expansion with inflammation of tissue and sheats
MRI: expansive lesion ND surrounding extensors; CXR: nl
CXR: nl
Ultrasound scan syno- ND vial thickening; MRI: chronic tenosynovitis; CXR: nl
MRI: focal area of ND signal alteration encasing the biceps tendon; CXR: nl
MRI : tenosynovitis; CXR : axillar adenopathy and upper lobe node
CT: cortical irregularityND near greater trochanter; MRI: cystic mass heterogenous; CXR: nl
CXR: nl
MRI: cystic mass with ND lobulated contours; CXR: nl
Tenosynovitis; CXR: nl
Radiological findings
Surgical biopsy
Granulomatous infl- Surgical biopsy lammation
Necrotizing granulomatous inflamamtion
Synovial biopsy
MTB isolated from joint specimen type
ND
AFB−,PCR+, culture−
ND
AFB+, culture+
AFB+, culture+
PCR+
Surgical biopsy
Synovial fluid
Caseous necrosis with granuloma formation, rice bodies
Surgical biopsy
Caseating granulo- Surgical biopsy maswith epithelioid histiocyte and multi-nucleated giant cells
Granulomatous Surgical biopsy inflammation with caseous necrosis, rice bodies
Granulomas with focal central necrosis, rice bodies
Granulomas with Surgical biopsy multinucleate giant cells
Granulation synovitis
TST+, AFB−, Epithelial granuloma Surgical biopsy culture−, with giant cells PCR+ and caseous necrosis
AFB−, PCR+, Chronic inflamma- Synovial culture+ tion with multiple fluid caseating and non caseating epithelioid granulomas with giant cellls
TST−
AFB-
AFB−, PCR−, ND culture
Bacteriological Histological findings findings
Duration -antibiotics
2 months − I + R + E + P + pyroxidine 10 months − I + R
3 months − ND
ND
ND
Tenosynovec- 4 months−I + R + tomy + E + STR debridement 3 months − I + R
Synovectomy 2 months − I + R + E + P 10 months − I + R
Excision
Surgical 6 months − I + R + E + P drainage and cleaning
Excision
Synovectomy ND
Debridement, 6 months − I + R + E + P excision 9 months − I + R
Debridement 2 months − I + R + E + P and surgical 4 months − I + R drainage
Debridement, 2 months − I + R + E + P excision 4 months−I + R
Debridement ND − I + E + Levofloxacin
Surgical treatment
E. Weber et al.
Nl
M/55
M/66
Seung [30]
Present case
M male, F female, ERS erythrocyte rate sedimentation, ND not determinated, CAI chronic adrenal insufficiency, DM diabete mellitus, RA rheumatoid arthritis, AFS acid fast stain, PCR polymerase chain reaction, US ultra sond, CXR chest X-ray, MRI magnetic resonance image, TST tuberculin skin test, Nl normal, I isoniazid, R rifampicin, E ethambutol, P pyrazinamide, STR streptomycin, PA acide paraaminosalicylic
Synovectomy ND – I + R + E + P US: synovitis; X-ray: CAI arthrosis; MRI: tenosynovitis; CXR: nl Hand, wrist
M/65 Lee 3 [29]
Mass, pain
Joint pain, tenderness and swelling/2 months
Surgical biopsy AFB-, PCR +, Non specific culture + granulomatous inflammation, rice bodies
Synovial biopsy Not realized AFB−, culture+ Caseating granulomatous inflammation Doppler ultrasound: DM severe hyperemia, normal joint space; MRI: tenosynovitis with mild subcutaneous soft tissue swelling Hand, wrist, Elevated ERS shoulder
2 months − I + R + E + P 8 months−I + R
ND I + R + E + P Not realized Nl Wrist Flexor weakness/1 week
ND
Synovial fluid PCR+ CXR : calcified DM nodules; MRI : flexor tenosynovitis, ulnar bursitis
Biological findings Sex/age (year) First author [Ref]
Table 1 continued
Symptoms/evolution
Involved joints
Radiological findings
Context
Bacteriological Histological findings findings
MTB isolated from joint specimen type
Surgical treatment
Duration -antibiotics
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons
characteristics of these 23 cases and those of our case were summarized in Table 1 [9–30]. Our case is similar to those of the literature with an usual presentation. Tenosynovitis tuberculosis affected middle-aged men and women (mean age 47 years; range 19–74). The presentation is insidious; more than 50 % had tendon swelling and painful lasting for months. Wrist is mainly affected (n = 15/23) as in our case followed by the hand (n = 6/23). Our patient physical examination revealed aspecific cystic mass as most cases of the literature and usually general symptoms were absent. Concomitant other musculoskeletal localizations are rare (1 case of pott’s disease [9]). No case of concomitant pulmonary tuberculosis was reported as described in Table 1. Among the 14 cases with chest radiography abnormalities, only two had nodules of the lung and tuberculin skin test was positive in 5/6 cases. Moreover, evidence of active chest disease is present in less than half of the cases of extrapulmonary tuberculosis reported in the literature [31]. Negative chest radiography or negative skin tuberculin test did not exclude the diagnosis of tenosynovitis tuberculosis (Table 1). Tuberculous tenosynovitis occurred usually in immunocompetent patients (18/23) as in our case. Immunosuppressive conditions are rarely associated with extrapulmonary localization unlike pulmonary tuberculosis [32]. As in our case definite diagnosis requires culture from surgical debridement (n = 12/17) or PCR allowing rapid instauration of the specific treatment. Macroscopic finding as rice bodies (n = 6/15) may help in the diagnosis of tuberculosis despite being not specific [33]. In front of these finding, surgeons should suspected tuberculosis and performed mycobacterial cultures. Direct examination is rarely positive (n = 5/15). Antituberculous therapy consisting of isoniazid, pyrazinamide, dexambutol and rifampicin for 2 months followed by a bitherapy for 3–10 months was used in our case as in literature as recommended [34]. Shortest regimens were not associated with a poor outcome (see Table 1). Surgery was usually performed for diagnostic or therapeutic issue [35].
Conclusion Isolated tenosynovitis should be known as a rare but possible extrapulmonary tuberculosis presentation, notably by infectious disease specialists and hand surgeons to perform adequate sampling and diagnosis. The diagnosis remains difficult because of insidious and aspecific presentation. Diagnostic should be considered in chronic tenosynovitis. Indeed functional prognosis may be compromised without adequate treatment. Conflict of interest The authors state that there is no conflict of interest.
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References 1. Floyd K, Baddeley A, Dean A, Dias HM, Falzon D, Garcia I, Glaziou P, Hiatt T, Law I, Lienhardt C, Nguyen L, Sismanidis C, Timimi H, Van Gemert W, Zignol M. Global tuberculosis report 2013. Geneva: World Health Organization; 2013. 2. Sandgren A, Hollo V, van der Werf MJ. Extrapulmonary tuberculosis in the European Union and European Economic Area, 2002 to 2011. Euro Surveill. 2013;21:18 pii: 20431. 3. Sharma SK, Mohan A. Extrapulmonary tuberculosis. Indian J Med Res. 2004;120:316–53. 4. Pattamapaspong N, Muttarak M, Sivasomboon C. Tuberculosis arthritis and tenosynovitis. Semin Musculoskelet Radiol. 2011;15:459–69. 5. Hsiao C-H, Cheng A, Huang Y-T, Liao C-H, Hsueh P-R. Clinical and pathological characteristics of mycobacterial tenosynovitis and arthritis. Infection. 2013;41:457–64. 6. Piersimoni C, Scarparo C. Extrapulmonary infections associated with nontuberculous mycobacteria in immunocompetent persons. Emerg Infect Dis. 2009;15:1351–8. 7. Boussel L, Marchand B, Blineau N, Pariset C, Hermier M, Picaud G, et al. Imaging of osteoarticular tuberculosis. J Radiol. 2002;83:1025–34. 8. Hindman N, Israel GM. Adrenal gland and adrenal mass calcification. Eur Radiol. 2005;15:1163–7. 9. Wanebo H. Tuberculous tenosynovitis in a patient with hyperuricemia. Calif Med. 1966;104:484–7. 10. Asaka T, Takizawa Y, Kariya T, Nitta E, Yasuda T, Fujita M, et al. Tuberculous tenosynovitis in the elbow joint. Intern Med. 1996;35:162–5. 11. Walker UA, Gutfleisch J, Peter HH. Case Number 23: tuberculous tenosynovitis. Ann Rheum Dis. 2002;61:384. 12. Aboudola S, Sienko A, Carey RB, Johnson S. Tuberculous tenosynovitis. Hum Pathol. 2004;35:1044–6. 13. Huang G-S, Lee C-H, Chen C-Y. Clinical images: tuberculous rice bodies of the wrist. Arthritis Rheum. 2005;52:1950. 14. Le Meur A, Arvieux C, Guggenbuhl P, Cormier M, Jolivet Gougeon A. Tenosynovitis of the wrist due to resistant Mycobacterium tuberculosis in a heart transplant patient. J Clin Microbiol. 2005;43:988–90. 15. Komurcu M, Botanlioglu H, Erdem H, Albay A. Delayed and misdiagnosis of wrist tuberculosis. Int J Infect Dis. 2006;10:337. 16. Amine B, Benbouazza K, Bahiri R, Hajjaj-Hassouni N. Multifocal tuberculous tenosynovitis. Joint Bone Spine. 2006;73:474–5. 17. Varshney MK, Trikha V, Gupta V. Isolated tuberculosis of achilles tendon. Joint Bone Spine. 2007;74:103–6. 18. Jira M, Qacif H, Sekkach Y, El Qatni M, Elouennass M, Ghafir D. Tuberculous tenosynovitis: an uncommon manifestation. Rev Med Interne. 2007;28:56–8.
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19. Muñoz P, Rodríguez M, Giannella M, Vega A, Anaya F, Serrano MJR, et al. A painful hand in a kidney transplant recipient. Nephrol Dial Transpl. 2007;22:971–2. 20. Oğüt T, Gökçe A, Kesmezacar H, Durak H, Botanlioğlu H, Erginer R. Isolated tuberculous tenosynovitis of the achilles tendon: a report of two cases. Acta Orthop Traumatol Turc. 2007;41:314–20. 21. Batra S, Ab Naell M, Barwick C, Kanvinde R. Tuberculous pyomyositis of the thigh masquerading as malignancy with concomitant tuberculous flexor tenosynovitis and dactylitis of the hand. Singap Med J. 2007;48:1042–6. 22. Higuchi S, Ishihara S, Kobayashi H, Arai T. A mass lesion of the wrist: a rare manifestation of tuberculosis. Intern Med. 2008;47:313–6. 23. Singh AP, Chadha M, Singh AP, Mahajan S. Isolated tuberculous biceps tenosynovitis bicipitoradial bursitis: a case report. J Shoulder Elbow Surg. 2009;18:e30–3. 24. Marques VB, Vieira HP, Alcantara ACC, Braga FN, Rocha FA, Medeiros MC. Tenosynovitis and carpal tunnel syndrome from mycobacterium tuberculosis—a rare manifestation of extrapulmonary tuberculosis. Acta Reumatol Port. 2010;35:82–4. 25. Hung M-H, Ho K-C, Huang K-F. Tuberculous tenosynovitis with rice body formation. ANZ J Surg. 2011;81:572. 26. Mrabet D, Ouenniche K, Mizouni H, Ounaies M, Khémiri C, Sahli H, et al. Tuberculosis tenosynovitis of the extensor tendons of the wrist. BMJ Case Rep. 2011;2011. doi:10.1136/bcr.06.2011.4347 27. Probst FA, Koch M, Lohmeyer J, Machens H-G, Schantz J-T. Tuberculous extensor tenosynovitis of the hand. Arch Orthop Trauma Surg. 2012;132:1141–5. 28. Chavan S, Sable SS, Tekade S, Punia P. Tuberculous tenosynovitis presenting as ganglion of wrist. Case Rep Surg. 2012;2012: 143921. 29. Seung OP, Sulaiman W. Osteoarticular tuberculosis mimicking rheumatoid arthritis. Mod Rheumatol. 2012;22:931–3. 30. Lee S-M, Lee W-J, Song A-R. Tuberculous tenosynovitis and ulnar bursitis of the wrist. Ann Rehabil Med. 2013;37:572–6. 31. Davidson PT, Horowitz I. Skeletal tuberculosis. A review with patient presentations and discussion. Am J Med. 1970;48:77–84. 32. Talbot JC, Bismil Q, Saralaya D, Newton D aG, Frizzel RM, Shaw DL. Musculoskeletal tuberculosis in Bradford—a 6-year review. Ann R Coll Surg Engl. 2007;89:405–9. 33. Iyengar K, Manickavasagar T, Nadkarni J, Mansour P, Loh W. Bilateral recurrent wrist flexor tenosynovitis and rice body formation in a patient with sero-negative rheumatoid arthritis: a case report and review of literature. Int J Surg Case Rep. 2011;2:208–11. 34. Treatment of Tuberculosis. Guidelines. 4th ed. Geneva: World Health Organization; 2010. 35. Dlimi F, Bellarbi S, Mahfoud M, Berrada MS, El Bardouni A, El Yaacoubi M. Tuberculosis of the hand and wrist: different aspects of 30 cases. Chir Main. 2011;30:198–204.
REVIEW
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons than infectious diseases specialists Emmanuelle Weber · Amandine Gagneux‑Brunon · V. Jacomo · Thibault Rousselon · Frederic Lucht · Elisabeth Botelho‑Nevers
Received: 10 November 2014 / Accepted: 2 February 2015 © Springer-Verlag Berlin Heidelberg 2015
Abstract Purpose Clinical presentation of tuberculosis is pleomorphic. Some forms are rare and better known by surgeons than infectious disease specialists. Methods We describe a rare case of isolated chronic tenosynovitis of the wrist due to Mycobacterium tuberculosis in a 66-year-old man and review similar cases in the literature. Results On literature search, only 23 other cases of tuberculous tenosynovitis were retrieved. Our case is similar, with an insidious classical presentation. The diagnosis was suggested at the surgical presentation by the presence of rice body masses. Conclusion The diagnosis of tuberculous tenosynovitis should be considered in chronic tenosynovitis. Functional prognosis may be committed without adequate treatment. Keywords Tenosynovitis · Wrist · Mycobacterium tuberculosis · Tuberculosis
in high income countries but prevalence remains high due to HIV infection, migrants, immunosuppression and aging [1]. Tuberculosis is the most common cause of death from infectious diseases world-wide [1]. Extra-pulmonary tuberculosis accounts for 15–30 % of cases [2] with musculoskeletal involvement in only 10–15 % of the cases mainly represented by bone and joint localizations [3]. Tuberculous tenosynovitis is therefore a rare form of musculoskeletal tuberculosis [4]. Indeed tenosynovitis is more frequently due to non tuberculous mycobacteria [5]. Tuberculous tenosynovitis may result in bone and tendon destructions, in particular when the treatment is delayed [4]. Consequently, it is crucial to determine the causative agent to treat the patients, as antibiotic regimens differ between tuberculous and non tuberculous mycobacteria [5, 6]. We describe here a rare case of isolated chronic tenosynovitis due to Mycobacterium tuberculosis in a 66-year-old man and review the literature.
Introduction Case report Tuberculosis remains challenging in diagnosis and treatment. The incidence of tuberculosis is decreasing especially E. Weber · A. Gagneux‑Brunon · F. Lucht · E. Botelho‑Nevers (*) Infectious Diseases Department, University Hospital, 42055 Saint‑Etienne Cedex 2, France e-mail: [email protected] V. Jacomo Biomnis Laboratory, 19 Avenue Tony Garnier, 69007 Lyon, France T. Rousselon Department of Orthopaedic Surgery, Private Hospital of Loire, 42030 Saint‑Etienne Cedex 2, France
A 66-year-old Algerian man was admitted in orthopaedic surgery department in March 2013 for a painful swelling of the right wrist gradually evolving for 6 months. He arrived in France 2 years ago. He had a past history of carpal tunnel surgery of the right hand few years ago in Algeria. He also had an idiopathic adrenal insufficiency diagnosed in 2004, and was treated with hydrocortisone 30 mg daily. No other complaints were reported. Physical examination revealed a cystic mass over the flexor tendons of the right wrist. His thenar eminence was swelling and inflammatory. He did not report paresthesia or sensory deficit. Laboratory values showed C reactive protein 16 mg/L and leucocytes count 9.3 g/L (56 % neutrophils, 32 % lymphocytes). X-ray of
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stopped. Subsequently, isoniazid, rifampicin and pyrazinamide were continued for 1 month then only rifampicin and isoniazid were continued for 7 months and stopped with a good evolution.
Discussion
Fig. 1 Macroscopic findings at surgical tenosynovectomy
hand and wrist showed arthrosis of the first proximal interphalangeal and metacarpophalangeal joint and thickening of the soft parts of thenar eminence. Ultrasound confirmed a large synovitis of long flexor of the thumb. Magnetic resonance imaging of the right wrist and hand revealed synovial thickening of flexor thumb sheath showing high signal intensity on T2-weighted image. It was associated with a 12 cm heterogen fluid collection in the thumb sheath and a second cystic area over the carpal tunnel and the flexor of fourth and fifth fingers. In May 2013 a surgical tenosynovectomy and extensive debridement were performed. Macroscopic examination revealed multiple rice body masses (Fig. 1). The Ziehl-Neelsen staining of this material did not show any acid resistant bacteria. On histopathologic examination, epidermic cyst and granulomatous inflammation with giant cells were observed. A mycobacterial strain grew on Bactect MGIT (Becton–Dickinson®) liquid agar, identified as Mycobacterium tuberculosis by polymerase chain reaction. Identification was performed by PCR: after DNA extraction by heat shock and sonication, amplification was made on thermocycler Gene Amp 2740 (Applied Biosystems®) with primers from Genotype MTBC kit (Hain®), followed by hybrizidation DNA–DNA on Genotype MTBc strips (Hain®). The strain of M. tuberculosis was susceptible to all-first line anti-TB agents. He was therefore addressed to our department for treatment. Three samples of sputum and urine were negative on direct examination and in culture for Mycobacterium tuberculosis. Tuberculin skin test was not performed. Thoraco abdomino pelvic CT scan showed bilateral adrenal hypertrophy with bilateral linear calcifications. No signs of pulmonary tuberculosis were found. In July 2013 an antituberculous therapy consisting of four drugs was started with isoniazid (300 mg daily), rifampicin (720 mg daily), dexambutol (1,500 mg daily) and pyrazinamide (1,800 mg daily) associated with hydrocortisone and fludrocortisone for his adrenal deficiency. At day 10 the patient developed a skin rash due to ethambutol that was
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We reported here a rare case of M. tuberculosis tenosynovitis. Indeed, among osteo-articular localizations of tuberculosis, tenosynovitis is rarely reported. In a series of osteoarticular TB, tenosynovitis represented 1 % of these forms, pott’s disease (50 %), peripheral arthritis (30 %) and osteomyelitis (19 %) being the most common osteoarticular TB presentation [7]. Non tuberculous Mycobacteria (NTM) are, by contrast, more frequently responsible for tenosynovitis and their incidence is increasing [5]. If the clinical presentations of NTM synovitis are similar to those of tuberculous ones, the treatment and the management are different showing the importance of a correct identification of the specie. Some difference could help to distinguish both infections [5, 6]. In NTM tenosynovitis, infection is usually acquired by direct inoculation (skin injury or trauma) of the pathogen from an environmental source or non-apparent inoculation (water contamination). The main causative agents are M. marinum, usually secondary to fishing injuries and M. kansasii [5, 6]. By contrast tuberculous tenosynovitis occurs most often secondary to hematogenous spread to the affected joint [5]. Our patient underwent a surgery for carpal tunnel few years ago. It should be account as source of the tuberculosis although, to the best of our knowledge, tuberculosis has been never reported due to a direct inoculation in a nosocomial context. In our case, we may consider that the patient had a second localization of tuberculosis disease but not yet active. Indeed he had chronic adrenal insufficiency with adrenal calcifications on CT-scan. This aspect although not specific of tuberculosis infection [8] was in this context a posteriori considered due to tuberculosis. His tenosynovitis was probably secondary to hematogenous spread. In NTM tenosynovitis the delay in diagnosis seems to be longer: about 1 month to 3 years. In M. tuberculosis tenosynovitis the duration varied from 10 days to 1 year [5]. The clinical course of NTM tenosynovitis is typically protracted but not destructive whereas M. tuberculosis tenosynovitis can lead to bone destructions if untreated [6]. To better characterize tuberculous tenosynovitis we performed a literature search in the medline database and using “tenosynovitis tuberculosis”, “Mycobacterium tuberculosis tenosynovitis” and “tuberculous tenosynovitis” as search terms. Tenosynovitis due to non tuberculous mycobacteria was excluded. By this literature search only 23 cases of tuberculous tenosynovitis were identified. Most cases are reported by surgeons. The
Sex/age (year)
F/53
F/74
F/58
F/34
M/21
M/60
M/21
M/44
M/55
M/45
First author [Ref]
Wanebo [9]
Asaka [10]
Walker [11]
Aboudola [12]
Huang [13]
Le Meur [14]
Komurcu [15]
Amine [16]
Varshney [17]
Jira [18]
Biological findings
Wrist
Wrist
Wrist
Wrist
Hand, forearm
Elbow
Painless swelling/ 1 year
Pain and swelling, fusiform mass/6 months
foot
Achilles tendon
ND
ND
Nl
ND
ND
MRI: tenosynovitis
ND
MRI: soft tissue with ND intensity lesions with liquefaction and necrosis with tenosynovial proliferation; CXR: nl
Ultrasonography : heterogeneous images
Synovial punction
MTB isolated from joint specimen type Excision
Surgical treatment
Synovial punction
Epithelioid and giant Surgical cell granuloma biopsy with caseation
Caseated inflammation
TST+, AFB+, Non specific chronic Synovial culture+ synovitis, rice fluid bodies
AFB−, PCR+, Granuloma with Punch culture giant cells and biopsy central caseation
Culture−
AFB−, culture+
AFB+, PCR+, Granulomas of Synovial culture+ epithelioide cells biopsy with multiple giant cells with central caseaous necrosis
Granulomatous with Surgical giant cell and case- biopsy ous necrosis, rice bodies
9 months − I + R + E
2 months − I + R + E + P 7 months − I + R
ND
ND − R + STR ND − R + E + STR
6 months − STR ND − I + PA
Duration -antibiotics
Not realized
Not realized
Not realized
2 months − I + R + P 4 months − I + R
3 months − I + R + E + P 9 months − I + R
2 months − I + R + P 7 months − I + R
Debridement, 2 months − I + R + E + P synovec10 months − I + R tomy, carpectomy
Synovectomy 1 month − I + R + E + P 6 months − R + E + P
Excision
Not realized
Surgical TST+, AFB+, Non caseating biopsy culture+ granulomas with epithelioid histiocytes Culture+
Not realized
Necrotising Synovial granulomas with fluid langerhan’s cells
TST+, AFB−, Non specific inflam- Aspiration biopsy Not realized PCR+, matory granuspecimen culture− lomas
Pulmonary culture+ tuberculous at 16 years
Scintiscan: Heart hyperfixation at transearly vascular times plant
MRI : nodes; CXR: nl
CXR: nl
ND
Bacteriological Histological findings findings
Pott TST+, AFB−, Granulomatous disease culture+ inflammation
Context
ND MRI, Ctscan: absecess; thoracic scan: infiltration of the right upper lobe; CXR: nl
ND
Radiological findings
Increased MRI : extensive ND CRP 25 mg/l inflamatory activity
ND
ND
ND
ND
Elevated ERS
Wrist, hand, Nl back
Involved joints
Inflammatory pain and Wrist, ankle, Nl swelling/15 months elbow
Pain, swelling, mass at the extensor area/6 months
Swelling, pain/ 6 months
Mass /2 years
Recurrent swelling/2 months
Painful swelling/6 months
Mass, swelling and tenderness/ND
Painful swelling and masses, back pain/3 years
Symptoms/evolution
Table 1 Description of tenosynovitis tuberculosis published cases
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons
13
13
M/57
Chavan [28]
Wrist
Three swellings mobile/3 years
Wrist
Painful swelling, Hand diminished range of motion/6 months
Painless swelling/5 months
Wrist
M/32
Forearm, elbow
Probst [27]
Pain and swelling/3 months
M/46
M/19
Singh [23]
Mass lesion with swell- Wrist ing/4 months
Mrabet [26]
M/24
Higuchi [22]
ND
Nl
ND
Biological findings
ND
Elevated ERS
Nl
Nl
Nl
Elevated ERS
Nl
Swelling, pain/5 years Thigh, wrist Elevated ERS
Soft tissue nods, pain/5 years
F/68
Batra [21]
Pain, swelling, nights Ankle sweats, weight loss, erythema/3 months
Achilles tendon
F/56
F/53
Ogut [20]
Diminished range of motion, swelling, pain/5 months
Hand, wrist
Hung [25]
F/19
Ogut [20]
Pain and swelling/1 month
Involved joints
Node and carpal tunnel Hand, wrist syndrome/6 years
F/58
Munoz [19]
Symptoms/evolution
Marques [24] F/60
Sex/age (year)
First author [Ref]
Table 1 continued
Kidney transplant
Context
DM; RA
ND
ND
MRI: extraskeletal synovial Koch’s or giant cell tumor of tendon sheath
ND
MRI: multiple lobu- ND lated cystic lesion expansion with inflammation of tissue and sheats
MRI: expansive lesion ND surrounding extensors; CXR: nl
CXR: nl
Ultrasound scan syno- ND vial thickening; MRI: chronic tenosynovitis; CXR: nl
MRI: focal area of ND signal alteration encasing the biceps tendon; CXR: nl
MRI : tenosynovitis; CXR : axillar adenopathy and upper lobe node
CT: cortical irregularityND near greater trochanter; MRI: cystic mass heterogenous; CXR: nl
CXR: nl
MRI: cystic mass with ND lobulated contours; CXR: nl
Tenosynovitis; CXR: nl
Radiological findings
Surgical biopsy
Granulomatous infl- Surgical biopsy lammation
Necrotizing granulomatous inflamamtion
Synovial biopsy
MTB isolated from joint specimen type
ND
AFB−,PCR+, culture−
ND
AFB+, culture+
AFB+, culture+
PCR+
Surgical biopsy
Synovial fluid
Caseous necrosis with granuloma formation, rice bodies
Surgical biopsy
Caseating granulo- Surgical biopsy maswith epithelioid histiocyte and multi-nucleated giant cells
Granulomatous Surgical biopsy inflammation with caseous necrosis, rice bodies
Granulomas with focal central necrosis, rice bodies
Granulomas with Surgical biopsy multinucleate giant cells
Granulation synovitis
TST+, AFB−, Epithelial granuloma Surgical biopsy culture−, with giant cells PCR+ and caseous necrosis
AFB−, PCR+, Chronic inflamma- Synovial culture+ tion with multiple fluid caseating and non caseating epithelioid granulomas with giant cellls
TST−
AFB-
AFB−, PCR−, ND culture
Bacteriological Histological findings findings
Duration -antibiotics
2 months − I + R + E + P + pyroxidine 10 months − I + R
3 months − ND
ND
ND
Tenosynovec- 4 months−I + R + tomy + E + STR debridement 3 months − I + R
Synovectomy 2 months − I + R + E + P 10 months − I + R
Excision
Surgical 6 months − I + R + E + P drainage and cleaning
Excision
Synovectomy ND
Debridement, 6 months − I + R + E + P excision 9 months − I + R
Debridement 2 months − I + R + E + P and surgical 4 months − I + R drainage
Debridement, 2 months − I + R + E + P excision 4 months−I + R
Debridement ND − I + E + Levofloxacin
Surgical treatment
E. Weber et al.
Nl
M/55
M/66
Seung [30]
Present case
M male, F female, ERS erythrocyte rate sedimentation, ND not determinated, CAI chronic adrenal insufficiency, DM diabete mellitus, RA rheumatoid arthritis, AFS acid fast stain, PCR polymerase chain reaction, US ultra sond, CXR chest X-ray, MRI magnetic resonance image, TST tuberculin skin test, Nl normal, I isoniazid, R rifampicin, E ethambutol, P pyrazinamide, STR streptomycin, PA acide paraaminosalicylic
Synovectomy ND – I + R + E + P US: synovitis; X-ray: CAI arthrosis; MRI: tenosynovitis; CXR: nl Hand, wrist
M/65 Lee 3 [29]
Mass, pain
Joint pain, tenderness and swelling/2 months
Surgical biopsy AFB-, PCR +, Non specific culture + granulomatous inflammation, rice bodies
Synovial biopsy Not realized AFB−, culture+ Caseating granulomatous inflammation Doppler ultrasound: DM severe hyperemia, normal joint space; MRI: tenosynovitis with mild subcutaneous soft tissue swelling Hand, wrist, Elevated ERS shoulder
2 months − I + R + E + P 8 months−I + R
ND I + R + E + P Not realized Nl Wrist Flexor weakness/1 week
ND
Synovial fluid PCR+ CXR : calcified DM nodules; MRI : flexor tenosynovitis, ulnar bursitis
Biological findings Sex/age (year) First author [Ref]
Table 1 continued
Symptoms/evolution
Involved joints
Radiological findings
Context
Bacteriological Histological findings findings
MTB isolated from joint specimen type
Surgical treatment
Duration -antibiotics
Tenosynovitis: a rare presentation of tuberculosis better known by hand surgeons
characteristics of these 23 cases and those of our case were summarized in Table 1 [9–30]. Our case is similar to those of the literature with an usual presentation. Tenosynovitis tuberculosis affected middle-aged men and women (mean age 47 years; range 19–74). The presentation is insidious; more than 50 % had tendon swelling and painful lasting for months. Wrist is mainly affected (n = 15/23) as in our case followed by the hand (n = 6/23). Our patient physical examination revealed aspecific cystic mass as most cases of the literature and usually general symptoms were absent. Concomitant other musculoskeletal localizations are rare (1 case of pott’s disease [9]). No case of concomitant pulmonary tuberculosis was reported as described in Table 1. Among the 14 cases with chest radiography abnormalities, only two had nodules of the lung and tuberculin skin test was positive in 5/6 cases. Moreover, evidence of active chest disease is present in less than half of the cases of extrapulmonary tuberculosis reported in the literature [31]. Negative chest radiography or negative skin tuberculin test did not exclude the diagnosis of tenosynovitis tuberculosis (Table 1). Tuberculous tenosynovitis occurred usually in immunocompetent patients (18/23) as in our case. Immunosuppressive conditions are rarely associated with extrapulmonary localization unlike pulmonary tuberculosis [32]. As in our case definite diagnosis requires culture from surgical debridement (n = 12/17) or PCR allowing rapid instauration of the specific treatment. Macroscopic finding as rice bodies (n = 6/15) may help in the diagnosis of tuberculosis despite being not specific [33]. In front of these finding, surgeons should suspected tuberculosis and performed mycobacterial cultures. Direct examination is rarely positive (n = 5/15). Antituberculous therapy consisting of isoniazid, pyrazinamide, dexambutol and rifampicin for 2 months followed by a bitherapy for 3–10 months was used in our case as in literature as recommended [34]. Shortest regimens were not associated with a poor outcome (see Table 1). Surgery was usually performed for diagnostic or therapeutic issue [35].
Conclusion Isolated tenosynovitis should be known as a rare but possible extrapulmonary tuberculosis presentation, notably by infectious disease specialists and hand surgeons to perform adequate sampling and diagnosis. The diagnosis remains difficult because of insidious and aspecific presentation. Diagnostic should be considered in chronic tenosynovitis. Indeed functional prognosis may be compromised without adequate treatment. Conflict of interest The authors state that there is no conflict of interest.
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E. Weber et al.
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