Received: 20 July 2013 Accepted: 6 January 2014 Disponible en ligne 15 February 2014
Available online at
ScienceDirect www.sciencedirect.com
Review
Tuberculosis of the temporomandibular joint Tuberculose de l’articulation temporomandibulaire C. Assouana, K. Anzouana, N.D. Nguessana, M. Millogoa, K. Horob, E. Konana, N. Zwetyengac,* a
Department of Stomatology and Maxillofacial Surgery, CHU de Treichville Abidjan, Coˆte D’Ivoire, France b Department of Pneumophtisiology, CHU Cocody, Coˆte d’Ivoire, France c Department of Maxillofacial Surgery, Plastique, Aesthetic and Reconstructive Surgery, Hand Surgery, centre hospitalier universitaire, universite´ de Bourgogne, faculte´ de me´decine, boulevard de Lattre-de-Tassigny, 21000 Dijon, France
Summary Introduction. Extrapulmonary and extra-spinal tuberculosis (TB) is rare, even in countries where the disease is endemic. Ten percent of these localizations are cervico-facial. Involvement of the temporomandibular joint (TMJ) is very unusual. We present the features of such a case. Review. We looked for patients managed for TMJ TB in 2 Maxillofacial Surgery departments and in 1 Pneumology & Phthisiology Department since 1992. The second part of the study was a literature review. One case was found in our departments and 15 other cases were found in published data. Most patients were women with mean age of 39.9 years (5 to 68). Pre-auricular swelling was the predominant functional sign, often without fever or change in the health status. The biological and radiological abnormalities were nonspecific (osteolysis, joint pinching, etc.). No lung involvement was observed. The joint recovered its normal function after appropriate management. Discussion. Tuberculosis of the TMJ is difficult to diagnose given its rarity and the non-specific nature of clinical and paraclinical signs. It must be considered in the differential diagnosis for common diseases of the TMJ whether TB is endemic or not. ß 2014 Elsevier Masson SAS. All rights reserved. Keywords: Osteoarticular tuberculosis, Temporomandibular joint, HIV
Re´sume´ Introduction. Les localisations extrapulmonaires et extrarachidiennes de tuberculose sont rares meˆme dans les pays a` forte ende´mie. Dix pour cent de ces localisations sont cervico-faciales. L’atteinte de l’articulation temporomandibulaire (ATM) est exceptionnelle. Au travers d’un cas nous faisons une mise au point de cette localisation particulie`re. Mate´riel et me´thode. La premie`re partie de cette e´tude a consiste´ en une recherche des patients pris en charge dans deux services de Chirurgie maxillofaciale et un service de pneumo-phtisiologie depuis 1992. La deuxie`me partie a consiste´ en une recherche bibliographique. Re´sultats. Un cas a e´te´ trouve´ dans nos services et 15 autres dans la litte´rature. Il y avait une pre´dominance fe´minine avec un aˆge moyen de 39,9 ans (de 5 a` 68 ans). La tume´faction pre´-auriculaire e´tait le signe fonctionnel pre´dominant, souvent sans signes ge´ne´raux. Les anomalies biologiques et radiologiques (oste´olyse, pincement articulaire. . .) n’e´taient pas spe´cifiques. Aucune atteinte pulmonaire n’e´tait note´e. Apre`s une prise en charge ade´quate, l’articulation avait repris un fonctionnement normal. Discussion. L’atteinte tuberculeuse de l’ATM est difficile a` diagnostiquer du fait de sa rarete´ et de ses caracte´ristiques cliniques et paracliniques non spe´cifique. Elle doit faire partie des diagnostics diffe´rentiels des pathologies courantes de l’articulation temporomandibulaire, en milieu ende´mique ou non. ß 2014 Elsevier Masson SAS. Tous droits re´serve´s. Mots cle´s : Tuberculose oste´oarticulaire, Articulation temporomandibulaire, VIH
* Corresponding author. e-mail: [email protected] (N. Zwetyenga). 2213-6533/$ - see front matter ß 2014 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2014.01.008 Rev Stomatol Chir Maxillofac Chir Orale 2014;115:88-93
88
Tuberculosis of the temporomandibular joint
Introduction Tuberculosis remains a problem of public health, in developed countries. According to the World Health Organization (WHO), there are 8.8 million new cases of tuberculosis and 1.45 million deaths related to the disease every year, worldwide [1] HIV infection [2] and the emergence of multiresistant strains of mycobacteria have aggravated this problem. The usual sites of tuberculosis are the lungs and the spine; infections in other sites are rare, even in countries where the disease is endemic [1]. Cervico-facial sites account for about 10% of extrapulmonary cases [3], with a clear predominance of cervical lymph node involvement (more than 90% of cases). Involvement of the temporomandibular joint (TMJ) is very unusual [4]. We present a case of TMJ TB and review the literature for this disease.
Figure 2. 3-D CT-Scan showing joint pinching and condyle lacunae on the right side.
Presentation of a clinical case One case of TMJ tuberculosis was found in the archives of 1 of the 2 maxillofacial surgery units in the Ivory Coast, since 1992. A 37-year-old healthy female patient consulted for pain in the right TMJ that had been increasing for the previous 3 months. The patient, who had no particular medical history, reported that the pain was associated with a progressive limitation of her mouth opening. Her vaccination schedule was up to date, and she had not been in contact with TB patients. Palpation revealed painful swelling of the right pretragal region, 0.5 cm
in diameter, with no external signs. The jaw opening was 15 mm. Blood tests were normal. CT-scan revealed a partial erosion of the condylar head and pinching in the joint (figs. 1 and 2). Magnetic resonance imaging (MRI) revealed a nodular tumor of the mandibular condyle, infiltrating the peri-articular soft tissues, and intra-articular effusion with an inflammatory aspect (fig. 3). The granulomatous tissue was removed peroperatively. There was partial erosion of the temporal bone and condyloid process. The temporal process was resected.
Figure 1. Axial CT-Scan showing intra-articular lacunae of the right mandibular condyle (circle).
Figure 3. Coronal MRI showing involvement of the joint and of periarticular soft tissues, and the presence of nodular tissue on the right condyle (circle).
89
no
Author; year of publication
Sex/ age
Country of/ diagnosis
Duration of symptoms (in months)
Main early clinical signs
Side
Blood tests
Main radiological signs
Other locations
Screening for HIV
Local management
General TTT (duration)
Outcome
1
Ducci; 1951 [17]
M/9
Italy/Italy
26
Slightly painful pre-auricular swelling, fistula
L
Na
Irregularities of the mandibular condyle
NAD
Not done
streptomycin injections
2
Ducci; 1951 [17]
F/16
Italy/Italy
4
L
Na
Peri-articular granulations
NAD
Not done
Ducci; 1951 [17]
F/57
Italy/Italy
36
R
Na
Tibia, talus, cervical adenopathy
Not done
4
Dechaume et al; 1956 [15]
M/66
France/ France
1
L
Normal
Ischio-pubic
Not done
5
Rinonapoli; 1966 [16]
M/5
Italy/Italy
na
Temporal, parotid and oropharyngeal swelling painful, trismus (8 mm), cracking sound Pre-auricular swelling, trismus
Irregularities of the mandibular condyle, joint pinching Decalcification of the condyle head
excision + streptomycin injections alkaline lavage + streptomycin injections puncture
NSR at 38 months; remodeling, MO normal NSR 26 months; normal function
3
Slightly painful pre-auricular swelling Pre-auricular swelling not painful
penicillin for 48 months, then local lavages with streptomycin streptomycin, Solfone, Vitamin A and D na
na
Na
Na
Na
Not done
6
Thibault et al., 1972 [7]
M/39
Maghreb/ France
2.5
Pre-auricular swelling, cracking sound
L
Increased ESR
na
7
Ruggiero et al., 1996 [10]
F/22
Haiti/ United States
1.5
L
Hyperleukocytosis
8
Wu et al., 1998 [8]
F/59
China/ China
6
L
Na
9
Soman and Davies; 2003 [9] Prasad et al., 2007 [3]
F/37
GB/GB
48
L
F/62
India/ India
3
Coscaron Blanco et al., 2009 [11]
F/68
Taiwan/ Taiwan
nc
Pre-auricular swelling with no initial pain, trismus (10 mm), joint stiffness Not painful preauricular swelling, trismus (15 mm) Painful swelling, trismus, pain, joint stiffness Parotid swelling, trismus (20 mm), foul-smelling purulent otorrhea Painful preauricular swelling, fever, latero-cervical adenopathy
Erosion of the mandibular condyle, temporal process and glenoid fossa Erosion of the condyle and collection of pus
11
L
L
NSR 19 months; normal function
Streptomycin for 2 months
time na; NSR; normal function
na
na
Not done
puncture
streptomycin+ INH + Becilan (at least 6 months)
time na; NSR; normal function NSR 11 months; remodeling, MO: 52 mm
NAD
Negative
biopsy + drainage
NSR 18 months; MO: 40 mm, remodeling
Erosion of the condyle and temporal process
NAD
Na
curettage
Doxycycline + RIPE + PZA, then ofloxacin + E, capreomycin + PAS Rifampicin (time na)
Increased CRP, leukocytosis Increased CRP
Calcification head of condyle
NAD
Na
exploration + lavages
na
Erosion of the condyle and temporal fossa, bone sequestration
Middle ear
Na
condylectomy
antituberculous (no details)
Na
Arthrosis with involvement of the muscles and parotid gland; no erosion
Na
Na
puncture + adenectomy
EIR streptomycin, hydrazide
time na; normal function NSR at 24 months; deformation and occlusion disorder time na; NSR; normal function
Time na; NSR; normal function
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:88-93
10
C. Assouan et al.
90
Table I Main published data on TMJ tuberculosis.
Table I (Continued ) no
Author; year of publication
Sex/ age
Country of/ diagnosis
Duration of symptoms (in months)
Main early clinical signs
Side
Blood tests
Main radiological signs
Other locations
Screening for HIV
Local management
General TTT (duration)
Outcome
12
Helbling et al., 2010 [5]
F/22
Ethiopia/ Switzerland
6
Painful preauricular swelling,
L
Na
NAD
Negative
biopsy
RIPE (9 months)
NSR at 11 months; normal function
13
Patel et al., 2012 [6]
F/27
Zimbabwewe/GB
2 weeks
Trismus (10 mm), pain
L
Increased CRP
Erosion of the condyle, temporal process and glenoid fossa, subcutaneous collection of pus Erosion of the condyle, collection of pus
Spine
Negative
curettage
RIPE (8 weeks)
14
Du et al., 2012 [12]
F/68
Taiwan/ Taiwan
2 weeks
Pre-auricular swelling not painful
R
Increased CRP and ESR
NAD
Na
biopsy
Antituberculous (no details)
15
Ranganathan et al., 2012 [14]
M/45
India/ India
3 months
R
Increased ERS
NAD
Negative
puncture
16
Assouan et al.
F/37
Ivory Coast/ Ivory Coast
3 months
Pre-auricular swelling, trismus (2 finger widths), fever Pre-auricular swelling, trismus (15 mm), pain
R
Normal
Erosion of the head of condyle, calcifications, collection of pus Erosion of the condyle, enlargement of the glenoid fossa Erosion of the condyle, joint pinching, inflammatory nodules
NSR at 12 months; ankylosis; prosthetic replacement Time na; NSR; normal function
Spine + ribs
Negative
resection of the condyle
RIP+ streptomycin (3 months), then RIP (6 months) RIPE 6 months
NSR 12 months; normal function
GB: Great Britain; R: right side; L: left side; Ri: rifampicin; I: isoniazid; P: pyrazinamide; E: ethambutol; Na: not available; TTT: treatment; NSR: no sign of recurrence; MO: maximal mouth opening. NAD: nothing abnormal detected.
91
Tuberculosis of the temporomandibular joint
NSR 18 months; normal function
C. Assouan et al.
Three months after the beginning of the treatment, while waiting for the results of bacteriological tests, the patient complained of progressively increasing chest and spine pain. Myelography and cervico-thoracic myeloscan revealed major erosion at various levels of the spine and in the ribs. An osteomedullar biopsy revealed lymphocytosis, compatible with a lymphoproliferative syndrome. The results of bacteriological tests for the TMJ were obtained after 3 months and allowed diagnosing caseo-follicular tuberculosis of the right TMJ. The final diagnosis was multifocal osseous tuberculosis. Antituberculous treatment was given, including a combination of Rifampicin-Isoniazid-Pyrazinamide-Ethambutol for 2 months followed by Rifampicin-Isoniazid for 4 months. Wearing a neck brace and corset was prescribed. Cure was achieved with normal mouth opening at 35 mm, without any recurrence at 18 months of follow-up.
Review Sixteen cases (11 women and 5 men) of TMJ tuberculosis, including the one presented above, were found in the literature (table I) [5–18]. The mean age was 39.9 years (5–68). Cases occurred on every continent. Symptoms ranged from 2 weeks to 4 years before consultation (table I). Pre-auricular swelling extending to the temporal or parotid region or even the oropharynx, sometimes associated with a painful trismus, was the most frequent clinical sign. Fever and cervical adenopathy were rare. The biological tests, when reported, revealed a non-specific inflammatory syndrome (high CRP, leukocytosis) (table I). The images, when radiological examination was available, systematically showed erosion, a peri- and/or intra-articular collection of pus, and/or joint pinching (table I). Other sites were noted in 4 patients, but never the lungs (table I). The patients were always negative for HIV infection (table I). Various treatments were used (table I). The mean follow-up was 18.9 months (11 to 38 months) and there was no sign of recurrence at the last consultation for every patient. Normal TMJ function was usually restored.
Discussion Tuberculosis of the temporomandibular joint is rare even in countries where TB is endemic [1,3–18]. Female patients are most often concerned [5,6,9–14,18], but this is still unexplained. The condition usually affects patients in their forties. Cases have been reported on every continent. Immigrants and underprivileged populations are the most commonly, affected whether TB is endemic in the country or not [5,6,8,11,19,20]. The clinical signs are unspecific. A deterioration of the global health and lymph nodes are frequent in pleuro-pulmonary tuberculosis [1], whereas they are very rare in TMJ TB.
92
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:88-93
Pre-auricular swelling, of variable volume, is the most frequent sign. It sometimes comes with restricted mouth opening. In most cases it is only slightly painful [5,7–18]. Osteoarticular tuberculosis is usually considered as a secondary location of a primary lymph node or pleuro-pulmonary infection. The disease can spread via 3 pathways [1,3,17]: in most cases via blood in patients with bacillemia, less frequently via the lymphatic system, and very rarely by contiguity of an abscess with the joint. Radiological images frequently show erosion of the mandibular condyle and/or the temporal process [5,6,8,11–13,15,18]. CT-scan (or cone beam) and MRI are contributive examinations to assess intra- and peri-articular involvement. They reveal erosion associated with calcifications. MRI can also reveal synovial proliferation. The absence of lung involvement is a constant feature, even in case of multifocal disease. The intradermal tuberculin sensitivity test is weakly contributive, notably in areas where TB is endemic and for patients from these areas, since positivity is superior to 90% [1]. The biological signs are unspecific and characterize an inflammatory syndrome [3,6,8,10,11,13,15]. HIV infection is a predisposing factor for TB infection. According to the WHO, around 13% of patients with tuberculosis are HIV-positive [1]. No case of TMJ TB related to HIV has been reported since 1983 (when HIV was identified), [5,6,11,15]. However, given the epidemiological context, testing for HIV must be performed systematically. The absence of response to usual antibiotics, often initiated as an empirical treatment before obtaining bacteriological results, may guide the diagnosis. The main differential diagnoses are septic arthritis, osteosarcoma, and primary parotid tumors. The diagnosis is confirmed by direct identification of the mycobacterium (M. tuberculosis or bovis) or by histological examination (tubercular follicles or epithelioid granulomas which may be necrotic or not). In some cases the diagnosis is made only on the positive response to antituberculous therapy. It takes from 1 to 5 months to identify the bacterium [5,6,8,11] and it takes an additional 3 to 4 weeks to obtain the antibiogram. The antibiogram must be performed systematically, when possible, given the increase of multi-resistant strains [1]. Antibiotic therapy must be initiated as soon as the tuberculous granulomas have been observed, without waiting for the results of bacteriological tests. This treatment must be administered by a specialized team, as is the case for any osteoarticular or lung involvement. The standard treatment includes a combination of Isoniazid, Rifampicin, Pyrazinamide, and Ethambutol for 2 months followed by Isoniazid and Rifampicin for 4 months [1]. Local treatment (intra-articular lavage with antituberculous agents [18], surgical debridement) is no longer used. The only contribution of surgery is diagnostic. The functional prognosis depends on the degree of TMJ destruc-
Tuberculosis of the temporomandibular joint
tion at diagnosis, but is usually good [6,8–13,15–18]. Joint ankylosis is possible [6]. Surgery is used only in case of complications (abscess, bone sequestration, scar repair) [14], or to treat sequels (ankylosis, joint reconstruction) [6].
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
World Health Organization. Global Tuberculosis Control. Geneva Switzerland: WHO Report; 2011 , http://www.who. int/tb/publications/global_report/2011/en/. Burwen DR, Bloch AB, Griffin LD, Ciesielski CA, Stern HA, Onorato IM. National trends in the concurrence of tuberculosis and acquired immunodeficiency syndrome. Arch Intern Med 1995;155:1281–6. Prasad KC, Sreedharan S, Chakravarthy Y, Prasad SC. Tuberculosis in the head and neck: experience in India. J Laryngol Otol 2007;121:979–85. Nalini B, Vinayak S. Tuberculosis in ear, nose, and throat practice: its presentation and diagnosis. Am J Otolaryngol 2006;27:39–45. Helbling CA, Lieger O, Smolka W, Lizuka T, Kuttenberger J. Primary tuberculosis of the TMJ: presentation of a case and literature review. Int J Oral Maxillofac Surg 2010;39:834–8. Patel M, Scott N, Newlands C. Case of tuberculosis of the temporomandibular joint. Br J Oral Maxillofac Surg 2012; 50:e1–3. Thibault JC, Harel J, Lepoivre M. A case of primary tuberculosis osteoarthritis the temporomandibular articulation. Rev Stomatol Chir Maxillofac 1972;73:162–6.
[8]
[9] [10]
[11]
[12]
[13]
[14]
[15] [16]
[17] [18]
[19]
[20]
Wu H, Wang QZ, Jin Y. Tuberculosis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:243. Soman D, Davies SJ. A suspected case of tuberculosis of temporomandibular joint. Br Dent J 2003;194:23–4. Ruggiero SL, Hilton E, Braun TW. Trismus and pre-auricular swelling in a 20-year-old black woman. J Oral Maxillofac Surg 1996;54:1234–9. Coscaro´n Blanco E, Blanco Sancho´n JJ, Martin Garrido EP, Pe´rez Liedo MC, Garcia Garcia I. Artritis temporomandibular por Mycobacterium bovis. Un problema diagno´stico. Acta Otorrinolaringol Esp 2009;60:291–4. Du CJ, Fu CH, Ng SH, Chang KP. Radiology quiz case 1. Tuberculous arthritis of the TMJ. Arch Otolaryngol Head Neck Surg 2012;138:197–8. Prasad KC, Sreedharan S, Prasad SC, Chakravarthy Y. Tuberculosis of the temporomandibular joint and parotid secondary to tuberculous otitis media. Otolaryngol Head Neck Surg 2007;137:974–5. Ranganathan LK, Mathew GC, Gandhi S, Manohar M. Tuberculosis of temporomandibular joint presenting as swelling in the pre-auricular region. J Oral Maxillofac Surg 2012;70:e28–31. Dechaume M, Vrasse R, Descrozailles Ch. Aspects inhabituels de la tuberculose buccale. Rev Stomatol 1956;57:131–6. Rinonapoli F. Su un caso di osteoartrite tubercolare dell’ articolazione temporomandibulare. Rass Int Stomatol Prat 1966;17:41–3. Ducci L, Rarasedi di tuberculosi osteoarticulare. Tre casi di artriti temporo mandibulare. Chir Org 1951;25:377. Lillebaek T, Andersen AB, Dirksen A, Smith E, Skovgaard LT, KokJensen A. Persistent high incidence of tuberculosis in immigrants in a low-incidence country. Emerg Infect Dis 2002; 8:679–84. te Beek LA, van der Werf MJ, Richter C, Borgdorff MW. Extrapulmonary tuberculosis by nationality, the Netherlands, 1993– 2001. Emerg Infect Dis 2006;12:1375–82. Malbruny B, Le Marrec G, Courageux K, Leclercq, Cattoir V. Rapid and efficient detection of Mycobacterium tuberculosis in respiratory and non-respiratory samples. Int J Tuberc Lung Dis 2011;15:553–5.
93
Available online at
ScienceDirect www.sciencedirect.com
Review
Tuberculosis of the temporomandibular joint Tuberculose de l’articulation temporomandibulaire C. Assouana, K. Anzouana, N.D. Nguessana, M. Millogoa, K. Horob, E. Konana, N. Zwetyengac,* a
Department of Stomatology and Maxillofacial Surgery, CHU de Treichville Abidjan, Coˆte D’Ivoire, France b Department of Pneumophtisiology, CHU Cocody, Coˆte d’Ivoire, France c Department of Maxillofacial Surgery, Plastique, Aesthetic and Reconstructive Surgery, Hand Surgery, centre hospitalier universitaire, universite´ de Bourgogne, faculte´ de me´decine, boulevard de Lattre-de-Tassigny, 21000 Dijon, France
Summary Introduction. Extrapulmonary and extra-spinal tuberculosis (TB) is rare, even in countries where the disease is endemic. Ten percent of these localizations are cervico-facial. Involvement of the temporomandibular joint (TMJ) is very unusual. We present the features of such a case. Review. We looked for patients managed for TMJ TB in 2 Maxillofacial Surgery departments and in 1 Pneumology & Phthisiology Department since 1992. The second part of the study was a literature review. One case was found in our departments and 15 other cases were found in published data. Most patients were women with mean age of 39.9 years (5 to 68). Pre-auricular swelling was the predominant functional sign, often without fever or change in the health status. The biological and radiological abnormalities were nonspecific (osteolysis, joint pinching, etc.). No lung involvement was observed. The joint recovered its normal function after appropriate management. Discussion. Tuberculosis of the TMJ is difficult to diagnose given its rarity and the non-specific nature of clinical and paraclinical signs. It must be considered in the differential diagnosis for common diseases of the TMJ whether TB is endemic or not. ß 2014 Elsevier Masson SAS. All rights reserved. Keywords: Osteoarticular tuberculosis, Temporomandibular joint, HIV
Re´sume´ Introduction. Les localisations extrapulmonaires et extrarachidiennes de tuberculose sont rares meˆme dans les pays a` forte ende´mie. Dix pour cent de ces localisations sont cervico-faciales. L’atteinte de l’articulation temporomandibulaire (ATM) est exceptionnelle. Au travers d’un cas nous faisons une mise au point de cette localisation particulie`re. Mate´riel et me´thode. La premie`re partie de cette e´tude a consiste´ en une recherche des patients pris en charge dans deux services de Chirurgie maxillofaciale et un service de pneumo-phtisiologie depuis 1992. La deuxie`me partie a consiste´ en une recherche bibliographique. Re´sultats. Un cas a e´te´ trouve´ dans nos services et 15 autres dans la litte´rature. Il y avait une pre´dominance fe´minine avec un aˆge moyen de 39,9 ans (de 5 a` 68 ans). La tume´faction pre´-auriculaire e´tait le signe fonctionnel pre´dominant, souvent sans signes ge´ne´raux. Les anomalies biologiques et radiologiques (oste´olyse, pincement articulaire. . .) n’e´taient pas spe´cifiques. Aucune atteinte pulmonaire n’e´tait note´e. Apre`s une prise en charge ade´quate, l’articulation avait repris un fonctionnement normal. Discussion. L’atteinte tuberculeuse de l’ATM est difficile a` diagnostiquer du fait de sa rarete´ et de ses caracte´ristiques cliniques et paracliniques non spe´cifique. Elle doit faire partie des diagnostics diffe´rentiels des pathologies courantes de l’articulation temporomandibulaire, en milieu ende´mique ou non. ß 2014 Elsevier Masson SAS. Tous droits re´serve´s. Mots cle´s : Tuberculose oste´oarticulaire, Articulation temporomandibulaire, VIH
* Corresponding author. e-mail: [email protected] (N. Zwetyenga). 2213-6533/$ - see front matter ß 2014 Elsevier Masson SAS. All rights reserved. http://dx.doi.org/10.1016/j.revsto.2014.01.008 Rev Stomatol Chir Maxillofac Chir Orale 2014;115:88-93
88
Tuberculosis of the temporomandibular joint
Introduction Tuberculosis remains a problem of public health, in developed countries. According to the World Health Organization (WHO), there are 8.8 million new cases of tuberculosis and 1.45 million deaths related to the disease every year, worldwide [1] HIV infection [2] and the emergence of multiresistant strains of mycobacteria have aggravated this problem. The usual sites of tuberculosis are the lungs and the spine; infections in other sites are rare, even in countries where the disease is endemic [1]. Cervico-facial sites account for about 10% of extrapulmonary cases [3], with a clear predominance of cervical lymph node involvement (more than 90% of cases). Involvement of the temporomandibular joint (TMJ) is very unusual [4]. We present a case of TMJ TB and review the literature for this disease.
Figure 2. 3-D CT-Scan showing joint pinching and condyle lacunae on the right side.
Presentation of a clinical case One case of TMJ tuberculosis was found in the archives of 1 of the 2 maxillofacial surgery units in the Ivory Coast, since 1992. A 37-year-old healthy female patient consulted for pain in the right TMJ that had been increasing for the previous 3 months. The patient, who had no particular medical history, reported that the pain was associated with a progressive limitation of her mouth opening. Her vaccination schedule was up to date, and she had not been in contact with TB patients. Palpation revealed painful swelling of the right pretragal region, 0.5 cm
in diameter, with no external signs. The jaw opening was 15 mm. Blood tests were normal. CT-scan revealed a partial erosion of the condylar head and pinching in the joint (figs. 1 and 2). Magnetic resonance imaging (MRI) revealed a nodular tumor of the mandibular condyle, infiltrating the peri-articular soft tissues, and intra-articular effusion with an inflammatory aspect (fig. 3). The granulomatous tissue was removed peroperatively. There was partial erosion of the temporal bone and condyloid process. The temporal process was resected.
Figure 1. Axial CT-Scan showing intra-articular lacunae of the right mandibular condyle (circle).
Figure 3. Coronal MRI showing involvement of the joint and of periarticular soft tissues, and the presence of nodular tissue on the right condyle (circle).
89
no
Author; year of publication
Sex/ age
Country of/ diagnosis
Duration of symptoms (in months)
Main early clinical signs
Side
Blood tests
Main radiological signs
Other locations
Screening for HIV
Local management
General TTT (duration)
Outcome
1
Ducci; 1951 [17]
M/9
Italy/Italy
26
Slightly painful pre-auricular swelling, fistula
L
Na
Irregularities of the mandibular condyle
NAD
Not done
streptomycin injections
2
Ducci; 1951 [17]
F/16
Italy/Italy
4
L
Na
Peri-articular granulations
NAD
Not done
Ducci; 1951 [17]
F/57
Italy/Italy
36
R
Na
Tibia, talus, cervical adenopathy
Not done
4
Dechaume et al; 1956 [15]
M/66
France/ France
1
L
Normal
Ischio-pubic
Not done
5
Rinonapoli; 1966 [16]
M/5
Italy/Italy
na
Temporal, parotid and oropharyngeal swelling painful, trismus (8 mm), cracking sound Pre-auricular swelling, trismus
Irregularities of the mandibular condyle, joint pinching Decalcification of the condyle head
excision + streptomycin injections alkaline lavage + streptomycin injections puncture
NSR at 38 months; remodeling, MO normal NSR 26 months; normal function
3
Slightly painful pre-auricular swelling Pre-auricular swelling not painful
penicillin for 48 months, then local lavages with streptomycin streptomycin, Solfone, Vitamin A and D na
na
Na
Na
Na
Not done
6
Thibault et al., 1972 [7]
M/39
Maghreb/ France
2.5
Pre-auricular swelling, cracking sound
L
Increased ESR
na
7
Ruggiero et al., 1996 [10]
F/22
Haiti/ United States
1.5
L
Hyperleukocytosis
8
Wu et al., 1998 [8]
F/59
China/ China
6
L
Na
9
Soman and Davies; 2003 [9] Prasad et al., 2007 [3]
F/37
GB/GB
48
L
F/62
India/ India
3
Coscaron Blanco et al., 2009 [11]
F/68
Taiwan/ Taiwan
nc
Pre-auricular swelling with no initial pain, trismus (10 mm), joint stiffness Not painful preauricular swelling, trismus (15 mm) Painful swelling, trismus, pain, joint stiffness Parotid swelling, trismus (20 mm), foul-smelling purulent otorrhea Painful preauricular swelling, fever, latero-cervical adenopathy
Erosion of the mandibular condyle, temporal process and glenoid fossa Erosion of the condyle and collection of pus
11
L
L
NSR 19 months; normal function
Streptomycin for 2 months
time na; NSR; normal function
na
na
Not done
puncture
streptomycin+ INH + Becilan (at least 6 months)
time na; NSR; normal function NSR 11 months; remodeling, MO: 52 mm
NAD
Negative
biopsy + drainage
NSR 18 months; MO: 40 mm, remodeling
Erosion of the condyle and temporal process
NAD
Na
curettage
Doxycycline + RIPE + PZA, then ofloxacin + E, capreomycin + PAS Rifampicin (time na)
Increased CRP, leukocytosis Increased CRP
Calcification head of condyle
NAD
Na
exploration + lavages
na
Erosion of the condyle and temporal fossa, bone sequestration
Middle ear
Na
condylectomy
antituberculous (no details)
Na
Arthrosis with involvement of the muscles and parotid gland; no erosion
Na
Na
puncture + adenectomy
EIR streptomycin, hydrazide
time na; normal function NSR at 24 months; deformation and occlusion disorder time na; NSR; normal function
Time na; NSR; normal function
Rev Stomatol Chir Maxillofac Chir Orale 2014;115:88-93
10
C. Assouan et al.
90
Table I Main published data on TMJ tuberculosis.
Table I (Continued ) no
Author; year of publication
Sex/ age
Country of/ diagnosis
Duration of symptoms (in months)
Main early clinical signs
Side
Blood tests
Main radiological signs
Other locations
Screening for HIV
Local management
General TTT (duration)
Outcome
12
Helbling et al., 2010 [5]
F/22
Ethiopia/ Switzerland
6
Painful preauricular swelling,
L
Na
NAD
Negative
biopsy
RIPE (9 months)
NSR at 11 months; normal function
13
Patel et al., 2012 [6]
F/27
Zimbabwewe/GB
2 weeks
Trismus (10 mm), pain
L
Increased CRP
Erosion of the condyle, temporal process and glenoid fossa, subcutaneous collection of pus Erosion of the condyle, collection of pus
Spine
Negative
curettage
RIPE (8 weeks)
14
Du et al., 2012 [12]
F/68
Taiwan/ Taiwan
2 weeks
Pre-auricular swelling not painful
R
Increased CRP and ESR
NAD
Na
biopsy
Antituberculous (no details)
15
Ranganathan et al., 2012 [14]
M/45
India/ India
3 months
R
Increased ERS
NAD
Negative
puncture
16
Assouan et al.
F/37
Ivory Coast/ Ivory Coast
3 months
Pre-auricular swelling, trismus (2 finger widths), fever Pre-auricular swelling, trismus (15 mm), pain
R
Normal
Erosion of the head of condyle, calcifications, collection of pus Erosion of the condyle, enlargement of the glenoid fossa Erosion of the condyle, joint pinching, inflammatory nodules
NSR at 12 months; ankylosis; prosthetic replacement Time na; NSR; normal function
Spine + ribs
Negative
resection of the condyle
RIP+ streptomycin (3 months), then RIP (6 months) RIPE 6 months
NSR 12 months; normal function
GB: Great Britain; R: right side; L: left side; Ri: rifampicin; I: isoniazid; P: pyrazinamide; E: ethambutol; Na: not available; TTT: treatment; NSR: no sign of recurrence; MO: maximal mouth opening. NAD: nothing abnormal detected.
91
Tuberculosis of the temporomandibular joint
NSR 18 months; normal function
C. Assouan et al.
Three months after the beginning of the treatment, while waiting for the results of bacteriological tests, the patient complained of progressively increasing chest and spine pain. Myelography and cervico-thoracic myeloscan revealed major erosion at various levels of the spine and in the ribs. An osteomedullar biopsy revealed lymphocytosis, compatible with a lymphoproliferative syndrome. The results of bacteriological tests for the TMJ were obtained after 3 months and allowed diagnosing caseo-follicular tuberculosis of the right TMJ. The final diagnosis was multifocal osseous tuberculosis. Antituberculous treatment was given, including a combination of Rifampicin-Isoniazid-Pyrazinamide-Ethambutol for 2 months followed by Rifampicin-Isoniazid for 4 months. Wearing a neck brace and corset was prescribed. Cure was achieved with normal mouth opening at 35 mm, without any recurrence at 18 months of follow-up.
Review Sixteen cases (11 women and 5 men) of TMJ tuberculosis, including the one presented above, were found in the literature (table I) [5–18]. The mean age was 39.9 years (5–68). Cases occurred on every continent. Symptoms ranged from 2 weeks to 4 years before consultation (table I). Pre-auricular swelling extending to the temporal or parotid region or even the oropharynx, sometimes associated with a painful trismus, was the most frequent clinical sign. Fever and cervical adenopathy were rare. The biological tests, when reported, revealed a non-specific inflammatory syndrome (high CRP, leukocytosis) (table I). The images, when radiological examination was available, systematically showed erosion, a peri- and/or intra-articular collection of pus, and/or joint pinching (table I). Other sites were noted in 4 patients, but never the lungs (table I). The patients were always negative for HIV infection (table I). Various treatments were used (table I). The mean follow-up was 18.9 months (11 to 38 months) and there was no sign of recurrence at the last consultation for every patient. Normal TMJ function was usually restored.
Discussion Tuberculosis of the temporomandibular joint is rare even in countries where TB is endemic [1,3–18]. Female patients are most often concerned [5,6,9–14,18], but this is still unexplained. The condition usually affects patients in their forties. Cases have been reported on every continent. Immigrants and underprivileged populations are the most commonly, affected whether TB is endemic in the country or not [5,6,8,11,19,20]. The clinical signs are unspecific. A deterioration of the global health and lymph nodes are frequent in pleuro-pulmonary tuberculosis [1], whereas they are very rare in TMJ TB.
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Pre-auricular swelling, of variable volume, is the most frequent sign. It sometimes comes with restricted mouth opening. In most cases it is only slightly painful [5,7–18]. Osteoarticular tuberculosis is usually considered as a secondary location of a primary lymph node or pleuro-pulmonary infection. The disease can spread via 3 pathways [1,3,17]: in most cases via blood in patients with bacillemia, less frequently via the lymphatic system, and very rarely by contiguity of an abscess with the joint. Radiological images frequently show erosion of the mandibular condyle and/or the temporal process [5,6,8,11–13,15,18]. CT-scan (or cone beam) and MRI are contributive examinations to assess intra- and peri-articular involvement. They reveal erosion associated with calcifications. MRI can also reveal synovial proliferation. The absence of lung involvement is a constant feature, even in case of multifocal disease. The intradermal tuberculin sensitivity test is weakly contributive, notably in areas where TB is endemic and for patients from these areas, since positivity is superior to 90% [1]. The biological signs are unspecific and characterize an inflammatory syndrome [3,6,8,10,11,13,15]. HIV infection is a predisposing factor for TB infection. According to the WHO, around 13% of patients with tuberculosis are HIV-positive [1]. No case of TMJ TB related to HIV has been reported since 1983 (when HIV was identified), [5,6,11,15]. However, given the epidemiological context, testing for HIV must be performed systematically. The absence of response to usual antibiotics, often initiated as an empirical treatment before obtaining bacteriological results, may guide the diagnosis. The main differential diagnoses are septic arthritis, osteosarcoma, and primary parotid tumors. The diagnosis is confirmed by direct identification of the mycobacterium (M. tuberculosis or bovis) or by histological examination (tubercular follicles or epithelioid granulomas which may be necrotic or not). In some cases the diagnosis is made only on the positive response to antituberculous therapy. It takes from 1 to 5 months to identify the bacterium [5,6,8,11] and it takes an additional 3 to 4 weeks to obtain the antibiogram. The antibiogram must be performed systematically, when possible, given the increase of multi-resistant strains [1]. Antibiotic therapy must be initiated as soon as the tuberculous granulomas have been observed, without waiting for the results of bacteriological tests. This treatment must be administered by a specialized team, as is the case for any osteoarticular or lung involvement. The standard treatment includes a combination of Isoniazid, Rifampicin, Pyrazinamide, and Ethambutol for 2 months followed by Isoniazid and Rifampicin for 4 months [1]. Local treatment (intra-articular lavage with antituberculous agents [18], surgical debridement) is no longer used. The only contribution of surgery is diagnostic. The functional prognosis depends on the degree of TMJ destruc-
Tuberculosis of the temporomandibular joint
tion at diagnosis, but is usually good [6,8–13,15–18]. Joint ankylosis is possible [6]. Surgery is used only in case of complications (abscess, bone sequestration, scar repair) [14], or to treat sequels (ankylosis, joint reconstruction) [6].
Disclosure of interest The authors declare that they have no conflicts of interest concerning this article.
References [1]
[2]
[3]
[4]
[5]
[6]
[7]
World Health Organization. Global Tuberculosis Control. Geneva Switzerland: WHO Report; 2011 , http://www.who. int/tb/publications/global_report/2011/en/. Burwen DR, Bloch AB, Griffin LD, Ciesielski CA, Stern HA, Onorato IM. National trends in the concurrence of tuberculosis and acquired immunodeficiency syndrome. Arch Intern Med 1995;155:1281–6. Prasad KC, Sreedharan S, Chakravarthy Y, Prasad SC. Tuberculosis in the head and neck: experience in India. J Laryngol Otol 2007;121:979–85. Nalini B, Vinayak S. Tuberculosis in ear, nose, and throat practice: its presentation and diagnosis. Am J Otolaryngol 2006;27:39–45. Helbling CA, Lieger O, Smolka W, Lizuka T, Kuttenberger J. Primary tuberculosis of the TMJ: presentation of a case and literature review. Int J Oral Maxillofac Surg 2010;39:834–8. Patel M, Scott N, Newlands C. Case of tuberculosis of the temporomandibular joint. Br J Oral Maxillofac Surg 2012; 50:e1–3. Thibault JC, Harel J, Lepoivre M. A case of primary tuberculosis osteoarthritis the temporomandibular articulation. Rev Stomatol Chir Maxillofac 1972;73:162–6.
[8]
[9] [10]
[11]
[12]
[13]
[14]
[15] [16]
[17] [18]
[19]
[20]
Wu H, Wang QZ, Jin Y. Tuberculosis of the temporomandibular joint. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:243. Soman D, Davies SJ. A suspected case of tuberculosis of temporomandibular joint. Br Dent J 2003;194:23–4. Ruggiero SL, Hilton E, Braun TW. Trismus and pre-auricular swelling in a 20-year-old black woman. J Oral Maxillofac Surg 1996;54:1234–9. Coscaro´n Blanco E, Blanco Sancho´n JJ, Martin Garrido EP, Pe´rez Liedo MC, Garcia Garcia I. Artritis temporomandibular por Mycobacterium bovis. Un problema diagno´stico. Acta Otorrinolaringol Esp 2009;60:291–4. Du CJ, Fu CH, Ng SH, Chang KP. Radiology quiz case 1. Tuberculous arthritis of the TMJ. Arch Otolaryngol Head Neck Surg 2012;138:197–8. Prasad KC, Sreedharan S, Prasad SC, Chakravarthy Y. Tuberculosis of the temporomandibular joint and parotid secondary to tuberculous otitis media. Otolaryngol Head Neck Surg 2007;137:974–5. Ranganathan LK, Mathew GC, Gandhi S, Manohar M. Tuberculosis of temporomandibular joint presenting as swelling in the pre-auricular region. J Oral Maxillofac Surg 2012;70:e28–31. Dechaume M, Vrasse R, Descrozailles Ch. Aspects inhabituels de la tuberculose buccale. Rev Stomatol 1956;57:131–6. Rinonapoli F. Su un caso di osteoartrite tubercolare dell’ articolazione temporomandibulare. Rass Int Stomatol Prat 1966;17:41–3. Ducci L, Rarasedi di tuberculosi osteoarticulare. Tre casi di artriti temporo mandibulare. Chir Org 1951;25:377. Lillebaek T, Andersen AB, Dirksen A, Smith E, Skovgaard LT, KokJensen A. Persistent high incidence of tuberculosis in immigrants in a low-incidence country. Emerg Infect Dis 2002; 8:679–84. te Beek LA, van der Werf MJ, Richter C, Borgdorff MW. Extrapulmonary tuberculosis by nationality, the Netherlands, 1993– 2001. Emerg Infect Dis 2006;12:1375–82. Malbruny B, Le Marrec G, Courageux K, Leclercq, Cattoir V. Rapid and efficient detection of Mycobacterium tuberculosis in respiratory and non-respiratory samples. Int J Tuberc Lung Dis 2011;15:553–5.
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