- Email: [email protected]
Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica
YIJOM-3506; No of Pages 5
Int. J. Oral Maxillofac. Surg. 2016; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2016.09.008, available online at http://www.sciencedirect.com
Case Report TMJ Disorders
Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica
J. Levorova, V. Machon, A. Guha, R. Foltan Department of Oral and Maxillofacial Surgery, General Hospital and First Medicine Faculty, Prague, Czech Republic
J. Levorova, V. Machon, A. Guha, R. Foltan: Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica. Int. J. Oral Maxillofac. Surg. 2016; xxx: xxx–xxx. # 2016 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Abstract. Septic arthritis of the temporomandibular joint (TMJ) is an unusual disease in adults. Inoculation of the pathogen may occur through traumatic or iatrogenic injuries, or more often by haematogenous spread from a distant focus. The cause of infection is unknown in most cases. A case of ostensibly mild septic arthritis of the TMJ with a good response to antibiotic therapy and lavage, but that finally led to fatal destruction of the joint structures in a 38-year-old female patient, is reported herein. The infection was caused by Raoultella ornithinolytica – a rare bacterial species in humans, which has not been reported previously in any patients with joint problems. The arthritis manifested 5 weeks after an arthroscopy procedure, so the cause was not clear.
Septic arthritis of the temporomandibular joint (TMJ) is an unusual disease in adults. The aetiology is of infectious origin, and it is usually caused by bacteria (Staphylococcus, Streptococcus, Haemophilus, Neisseria, Mycobacterium) or fungi (Candida).1,2 The infection can penetrate into the joint through an open traumatic or iatrogenic wound. More often it is disseminated by haematogenous spread from some distant infectious focus in the body.2,3 Case report
A 38-year-old woman was referred to the department of oral and maxillofacial 0901-5027/000001+05
surgery of a university hospital in Prague, Czech Republic by her dentist for pain in the left pre-auricular region associated with limited mouth opening and restricted jaw movement (28 mm), lasting a week. Clinical and radiological findings led to the diagnosis of disc displacement without reduction of the left TMJ (Fig. 1 shows a panoramic radiograph; Fig. 2 shows magnetic resonance imaging findings). The patient underwent conservative treatment, namely thermotherapy, occlusal splint therapy, stretching, and physiotherapy. This was continued for 6 months with minimal effect; consequently an arthrocentesis was done. The arthrocentesis
Key words: rare infection; temporomandibular joint; septic arthritis; total alloplastic replacement. Accepted for publication 9 September 2016
was performed with the use of two 19gauge needles, in accordance with the procedure of Nitzan et al.4; 120 ml of lactated Ringer’s solution was used for irrigation, and consequently hyaluronic acid was injected slowly into the upper joint space (Hyalgan; molecular weight 500– 730 kDa, 1 ml). There was no improvement after the arthrocentesis. Therefore, the patient was indicated for arthroscopic lavage. Seven months after the diagnosis, an arthroscopy of the upper joint space of the left TMJ was performed. Arthroscopic findings showed an adhesion type column in the lateral and medial portions of the posterior recess, chronic inflammation of
# 2016 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
2
Levorova et al.
Fig. 1. Panoramic radiograph of the patient, who had pain in the left pre-auricular region and limited mouth opening. The radiograph shows no degenerative changes of the left mandibular condyle.
Fig. 2. Magnetic resonance imaging showing anterior disc displacement without reduction and even contours of the condylar head.
the retrodiscal tissue, and incipient fibrillation of the cartilage in the intermediate zone. Visualization of the anterior recess was difficult due to strong adhesions. The articular disc was found to be anteriorly displaced without reduction during movement. No degenerative changes of the disc and cartilage were noted. Lavage and lysis of the adhesions were done during the arthroscopic procedure. One week postoperatively, the patient was completely without pain, and mouth opening had improved to 42 mm (from the initial 28 mm). However, on day 43 after the arthroscopy, she suddenly presented with a complaint of swelling lasting 2 days, pain in her left pre-auricular area, and limited mouth opening. A clinical examination revealed swelling without fluctuation and redness of the left pre-auricular region. Mouth opening was limited to 32 mm, with deviation to the left. Her occlusion was normal, without discrepancies. The patient’s overall condition was normal: she was afebrile and
inflammatory markers were not increased. The wound from the previous arthroscopy was healing without any dehiscence.
A differential diagnosis of a septic complication from the previous procedure was considered, so arthroscopic lavage of the TMJ was recommended to the patient. She refused a repeat arthroscopy and general anaesthesia at that time, but agreed to arthrocentesis as a lesser invasive procedure. Arthrocentesis with two 19-gauge needles inserted in the sites of the previous insertions was performed. A translucent light yellow fluid was aspirated and it was presumed that pus was present (Fig. 3). The fluid was sent for microbiological analysis. Empirical oral antibiotics were administered (amoxicillin 500 mg, three times daily), and cultures subsequently showed the presence of Raoultella ornithinolytica. The bacterial culture was susceptible to amoxicillin–clavulanic acid, ampicillin– sulbactam, sulbactam, ciprofloxacin, cotrimoxazole, and cefuroxime, but was resistant to ampicillin. The treatment was changed to oral amoxicillin combined with clavulanic acid (the dose remained unchanged) and this was continued for 13 days. After 2 weeks, the patient was without swelling and the pre-auricular region was pain-free. No signs of inflammation were present, and the patient’s overall condition had improved considerably. Furthermore, her laboratory values had normalized. Despite the improvement, her mouth opening was still limited to 32 mm. The patient was followed up regularly and underwent rehabilitation of her mouth opening. Mouth opening improved to 41 mm with symmetrical movement of both condylar heads. Subjectively, the patient was without any complaints. Eleven months after her diagnosis, she began to complain of mild pain in the left
Fig. 3. Arthrocentesis of the left temporomandibular joint; swelling is seen in the pre-auricular region. The syringe contained a translucent yellow liquid, aspirated from the joint.
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
TMJ septic arthritis caused by rare Raoultella
3
Fig. 4. Degenerative changes in the left temporomandibular joint seen at 11 months after the diagnosis of septic arthritis.
pre-auricular region and her mouth opening gradually decreased to 24 mm with no effect of subsequent rehabilitation. Cone beam computed tomography (CBCT) scans were performed and unexpectedly showed advanced arthritic changes. Subchondral cysts and resorption of the condylar head, and resorption as well as bone apposition of the glenoid fossa were present in the left TMJ. The condition of the patient’s left TMJ is shown in Fig. 4. The patient underwent a total alloplastic joint replacement (Biomet Inc., Jacksonville, FL, USA) (Fig. 5). The only complication of the surgery was facial nerve weakness, which improved after physiotherapy. The patient is currently 12
months after reconstruction. Her mouth opening is 43 mm and she is without any complaints and is clinically stable. Discussion
Raoultella ornithinolytica is a Gram-negative aerobic bacterium belonging to the family Enterobacteriaceae. It was previously called Klebsiella ornithinolytica. This bacterium is known for its histidine decarboxylase activity, causing scombroid syndrome (a pseudoallergic food poisoning caused by the consumption of contaminated fish). R. ornithinolytica is found in fish gut, ticks, termites, and aquatic environments. Infections caused by
Fig. 5. The total joint replacement: panoramic radiograph taken on day 1 postoperative.
R. ornithinolytica are rare in humans.5 Among humans, it predominantly affects the biliary tract, and is most often associated with other previous infections, iatrogenic injuries, or malignancy.5,6 Clinical findings are various and include local symptoms such as oedema, marked rash, and pain at the site of infection, with or without bacteraemia. General symptoms include fever, hypotension, tachycardia, headache, and bronchospasm, in some cases leading to sepsis. With regard to laboratory findings, C-reactive protein, the erythrocyte sedimentation rate, and leucocyte counts may be increased.5,6 Infection of any joint, including the TMJ, has not been reported to date. General symptoms of septic arthritis of the TMJ are similar to those of any other infection: increased body temperature, fatigue, and malaise, and sometimes an increase in certain inflammatory markers. Local symptoms include pain, swelling, redness in the pre-auricular region, and limited mouth opening. An open bite on the affected side may be present due to increased joint fluid.1–3,7 Common complications of septic arthritis are osteomyelitis, fibrosis, joint dysfunction, degenerative changes of the joint structures, and ankylosis. When this
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
4
Levorova et al.
occurs in childhood, growth disorders on the affected side of the mandible may be present.2,8 The causative pathogens of septic arthritis of the TMJ are usually bacteria, the most common being Streptococcus, Staphylococcus, Haemophilus, Neisseria, and Mycobacterium. Pathogen penetration into the joint proceeds via haematogenous spread, direct extension from a nearby infection, or through an open wound.1,2 Risk factors for the development of septic arthritis include systemic and autoimmune diseases (e.g., rheumatoid arthritis, diabetes mellitus, lupus erythematosus, and immunosuppression), the long-term use of steroids, and a blunt trauma causing damage to the local vessels, with greater bacterial access into the joint.7 In the case presented herein, the patient had arterial hypertension, bronchial asthma, and fibrous mastopathy, but no other internal, oncological, or rheumatoid diseases. In the absence of subjective complaints and negative laboratory findings, it can be presumed that no other inflammation was present. Had simultaneous inflammation in another body region been identified, it would have been presumed to have been caused by the same bacterium – R. ornithinolytica. Only one case of septic arthritis as a complication of TMJ arthroscopy has been reported to date.1 In this case, McCain et al. described septic arthritis as an early complication; the symptoms became evident on day 6 postoperative (increased pain and swelling, and spontaneous drainage of pus from the puncture sites). Furthermore, even articles presenting the complications of TMJ arthroscopy for large cohorts of patients have not described cases of septic arthritis.9,10 In the case presented here, the first symptoms were noted on day 43 postoperative; and thus would not represent an early complication of surgery. The surgical wound after arthroscopy showed signs of normal healing, with no signs of infection and no exudation. This would disprove the hypothesis of penetration of the infection through the wound, except if the very small wound caused by the 1.9-mm arthroscopic cannula is considered. If haematogenous spread and spread by direct extension of another infection are excluded, then penetration could be presumed to have occurred through the wound postoperatively. Intravenous antibiotic prophylaxis was administered as recommended by the department of microbiology. The arthroscopy procedure was done under
sterile conditions. Inadequate sterilization or contamination during the period after sterilization could not be confirmed or excluded, so the possibility that the infection was introduced via the arthroscope or other materials/devices used during the procedure cannot be ruled out. Contamination with R. ornithinolytica present as part of the normal skin flora on the body surface or through cerumen has not been described previously, so it was assumed that the patient’s body surface was not colonized by this bacterium (a smear was not performed). The epidemiologist ruled out the presence of R. ornithinolytica in the patient’s area of residence (Jihlava, eastern Bohemia). The patient was asked about having ingested fish during the 2 weeks prior to the onset of the swelling (to exclude the introduction of infection with food), but she did not remember eating any fish. Scombroid syndrome takes its name from the Scombridae family. This family includes tuna fish, which are widely known and often eaten. It was considered unlikely that the patient had ingested any, but if so, she was without signs of enteritis. Septic arthritis of the TMJ is resolved by pus drainage and antibiotic therapy according to microbiological findings (with broad spectrum antibiotics in the acute stage). Arthroscopic lysis and lavage allows early elimination of degenerative irreversible changes, which may be present 7–10 days after joint infection. Arthroscopy rather than arthrocentesis is recommended for more extensive lavage and direct visualization.2,3,7 In conclusion, a rare case of R. ornithinolytica infection of the TMJ is reported here. During a period of 1 year after infection by this bacterium, progressive and destructive degenerative changes in the TMJ led to the need for a total alloplastic joint replacement. A clear route of infection was not revealed, but it is presumed that this was a late complication of arthroscopy. The presence of an acute or chronic infection could not be proven either clinically or through laboratory findings. Despite this, the pathogenic organism or inflammatory process was still present in the soft tissues of the joint. Treatment methods such as antibiotic therapy alone or in combination with arthrocentesis are not sufficient for the treatment of septic arthritis of the TMJ. The cause of this septic arthritis is not completely clear. It is alarming that previous surgery could have had such severe consequences. The treatment of such a case can be challenging, as there is currently a lack of evidence and information regarding definitive curative therapy.
Funding
There were no sources of funding for this research. Competing interests
No competing interests. Ethical approval
Institutional ethics approval was obtained (No. 1280/15 IS-IV). Patient consent
Patient consent was obtained. References 1. McCain JP, Zabiegalski NA, Levine RL. Joint infection as a complication of temporomandibular joint arthroscopy: a case report. J Oral Maxillofac Surg 1993;51:1389–92. 2. Sembronio S, Albiero AM, Robiony M, Costa F, Toro C, Politi M. Septic arthritis of the temporomandibular joint successfully treated with arthroscopic lysis and lavage: case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e1–6. 3. Murakami K, Matsumoto K, Iizuka T. Suppurative arthritis of the temporomandibular joint. Report of a case with special reference to arthroscopic observations. J Maxillofac Surg 1984;12:41–5. 4. Nitzan DW, Dolwick MF, Martinez GA. Temporomandibular joint arthrocentesis: a simplified treatment for severe, limited mouth opening. J Oral Maxillofac Surg 1991;49:1163–7. http://dx.doi.org/10.1016/ s0278-2391(91)90409-F. 5. Sandal G, Ozen M. Fatal Raoultella ornithinolytica sepsis and purpura fulminans in a preterm newborn. Indian J Paediatr Dermatol 2014;15:24–6. 6. Hadano Y, Tsukahara M, Ito K, Suzuki J, Kawamura I, Kurai H. Raoultella ornithinolytica bacteremia in cancer patients: report of three cases. Intern Med 2012;51:3193–5. 7. Leighty SM, Spach DH, Myall RW, Burns JL. Septic arthritis of the temporomandibular joint: review of the literature and report of two cases in children. Int J Oral Maxillofac Surg 1993;22:292–7. 8. Gayle EA, Young SM, McKenna SJ, McNaughton CD. Septic arthritis of the temporomandibular joint: case reports and review of the literature. J Emerg Med 2013;45:674–8. http://dx.doi.org/10.1016/ j.jemermed.2013.01.034. 9. Carls FR, Engelke W, Locher MC, Sailer HF. Complications following arthroscopy of the temporomandibular joint: analysis covering a 10-year period (451 arthroscopies). J Craniomaxillofac Surg 1996;24:12–5.
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
TMJ septic arthritis caused by rare Raoultella 10. Gonza´lez-Garcı´a R, Rodrı´guez-Campo FJ, Escorial-Herna´ndez V, Mun˜oz-Guerra MF, Sastre-Pe´rez J, Naval-Gı´as L, et al. Complications of temporomandibular joint arthroscopy: a retrospective analytic study of 670 arthroscopic procedures. J Oral Maxillofac Surg 2006;64:1587–91.
Address: Jitka Levorova Department of Oral and Maxillofacial Surgery General Hospital and First Medicine Faculty U Nemocnice 2 str.
Prague 2 120 00 Czech Republic Tel: +420 224 96 3191; Fax: +420 224 96 3193 E-mail: [email protected]
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
5
Int. J. Oral Maxillofac. Surg. 2016; xxx: xxx–xxx http://dx.doi.org/10.1016/j.ijom.2016.09.008, available online at http://www.sciencedirect.com
Case Report TMJ Disorders
Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica
J. Levorova, V. Machon, A. Guha, R. Foltan Department of Oral and Maxillofacial Surgery, General Hospital and First Medicine Faculty, Prague, Czech Republic
J. Levorova, V. Machon, A. Guha, R. Foltan: Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica. Int. J. Oral Maxillofac. Surg. 2016; xxx: xxx–xxx. # 2016 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Abstract. Septic arthritis of the temporomandibular joint (TMJ) is an unusual disease in adults. Inoculation of the pathogen may occur through traumatic or iatrogenic injuries, or more often by haematogenous spread from a distant focus. The cause of infection is unknown in most cases. A case of ostensibly mild septic arthritis of the TMJ with a good response to antibiotic therapy and lavage, but that finally led to fatal destruction of the joint structures in a 38-year-old female patient, is reported herein. The infection was caused by Raoultella ornithinolytica – a rare bacterial species in humans, which has not been reported previously in any patients with joint problems. The arthritis manifested 5 weeks after an arthroscopy procedure, so the cause was not clear.
Septic arthritis of the temporomandibular joint (TMJ) is an unusual disease in adults. The aetiology is of infectious origin, and it is usually caused by bacteria (Staphylococcus, Streptococcus, Haemophilus, Neisseria, Mycobacterium) or fungi (Candida).1,2 The infection can penetrate into the joint through an open traumatic or iatrogenic wound. More often it is disseminated by haematogenous spread from some distant infectious focus in the body.2,3 Case report
A 38-year-old woman was referred to the department of oral and maxillofacial 0901-5027/000001+05
surgery of a university hospital in Prague, Czech Republic by her dentist for pain in the left pre-auricular region associated with limited mouth opening and restricted jaw movement (28 mm), lasting a week. Clinical and radiological findings led to the diagnosis of disc displacement without reduction of the left TMJ (Fig. 1 shows a panoramic radiograph; Fig. 2 shows magnetic resonance imaging findings). The patient underwent conservative treatment, namely thermotherapy, occlusal splint therapy, stretching, and physiotherapy. This was continued for 6 months with minimal effect; consequently an arthrocentesis was done. The arthrocentesis
Key words: rare infection; temporomandibular joint; septic arthritis; total alloplastic replacement. Accepted for publication 9 September 2016
was performed with the use of two 19gauge needles, in accordance with the procedure of Nitzan et al.4; 120 ml of lactated Ringer’s solution was used for irrigation, and consequently hyaluronic acid was injected slowly into the upper joint space (Hyalgan; molecular weight 500– 730 kDa, 1 ml). There was no improvement after the arthrocentesis. Therefore, the patient was indicated for arthroscopic lavage. Seven months after the diagnosis, an arthroscopy of the upper joint space of the left TMJ was performed. Arthroscopic findings showed an adhesion type column in the lateral and medial portions of the posterior recess, chronic inflammation of
# 2016 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
2
Levorova et al.
Fig. 1. Panoramic radiograph of the patient, who had pain in the left pre-auricular region and limited mouth opening. The radiograph shows no degenerative changes of the left mandibular condyle.
Fig. 2. Magnetic resonance imaging showing anterior disc displacement without reduction and even contours of the condylar head.
the retrodiscal tissue, and incipient fibrillation of the cartilage in the intermediate zone. Visualization of the anterior recess was difficult due to strong adhesions. The articular disc was found to be anteriorly displaced without reduction during movement. No degenerative changes of the disc and cartilage were noted. Lavage and lysis of the adhesions were done during the arthroscopic procedure. One week postoperatively, the patient was completely without pain, and mouth opening had improved to 42 mm (from the initial 28 mm). However, on day 43 after the arthroscopy, she suddenly presented with a complaint of swelling lasting 2 days, pain in her left pre-auricular area, and limited mouth opening. A clinical examination revealed swelling without fluctuation and redness of the left pre-auricular region. Mouth opening was limited to 32 mm, with deviation to the left. Her occlusion was normal, without discrepancies. The patient’s overall condition was normal: she was afebrile and
inflammatory markers were not increased. The wound from the previous arthroscopy was healing without any dehiscence.
A differential diagnosis of a septic complication from the previous procedure was considered, so arthroscopic lavage of the TMJ was recommended to the patient. She refused a repeat arthroscopy and general anaesthesia at that time, but agreed to arthrocentesis as a lesser invasive procedure. Arthrocentesis with two 19-gauge needles inserted in the sites of the previous insertions was performed. A translucent light yellow fluid was aspirated and it was presumed that pus was present (Fig. 3). The fluid was sent for microbiological analysis. Empirical oral antibiotics were administered (amoxicillin 500 mg, three times daily), and cultures subsequently showed the presence of Raoultella ornithinolytica. The bacterial culture was susceptible to amoxicillin–clavulanic acid, ampicillin– sulbactam, sulbactam, ciprofloxacin, cotrimoxazole, and cefuroxime, but was resistant to ampicillin. The treatment was changed to oral amoxicillin combined with clavulanic acid (the dose remained unchanged) and this was continued for 13 days. After 2 weeks, the patient was without swelling and the pre-auricular region was pain-free. No signs of inflammation were present, and the patient’s overall condition had improved considerably. Furthermore, her laboratory values had normalized. Despite the improvement, her mouth opening was still limited to 32 mm. The patient was followed up regularly and underwent rehabilitation of her mouth opening. Mouth opening improved to 41 mm with symmetrical movement of both condylar heads. Subjectively, the patient was without any complaints. Eleven months after her diagnosis, she began to complain of mild pain in the left
Fig. 3. Arthrocentesis of the left temporomandibular joint; swelling is seen in the pre-auricular region. The syringe contained a translucent yellow liquid, aspirated from the joint.
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
TMJ septic arthritis caused by rare Raoultella
3
Fig. 4. Degenerative changes in the left temporomandibular joint seen at 11 months after the diagnosis of septic arthritis.
pre-auricular region and her mouth opening gradually decreased to 24 mm with no effect of subsequent rehabilitation. Cone beam computed tomography (CBCT) scans were performed and unexpectedly showed advanced arthritic changes. Subchondral cysts and resorption of the condylar head, and resorption as well as bone apposition of the glenoid fossa were present in the left TMJ. The condition of the patient’s left TMJ is shown in Fig. 4. The patient underwent a total alloplastic joint replacement (Biomet Inc., Jacksonville, FL, USA) (Fig. 5). The only complication of the surgery was facial nerve weakness, which improved after physiotherapy. The patient is currently 12
months after reconstruction. Her mouth opening is 43 mm and she is without any complaints and is clinically stable. Discussion
Raoultella ornithinolytica is a Gram-negative aerobic bacterium belonging to the family Enterobacteriaceae. It was previously called Klebsiella ornithinolytica. This bacterium is known for its histidine decarboxylase activity, causing scombroid syndrome (a pseudoallergic food poisoning caused by the consumption of contaminated fish). R. ornithinolytica is found in fish gut, ticks, termites, and aquatic environments. Infections caused by
Fig. 5. The total joint replacement: panoramic radiograph taken on day 1 postoperative.
R. ornithinolytica are rare in humans.5 Among humans, it predominantly affects the biliary tract, and is most often associated with other previous infections, iatrogenic injuries, or malignancy.5,6 Clinical findings are various and include local symptoms such as oedema, marked rash, and pain at the site of infection, with or without bacteraemia. General symptoms include fever, hypotension, tachycardia, headache, and bronchospasm, in some cases leading to sepsis. With regard to laboratory findings, C-reactive protein, the erythrocyte sedimentation rate, and leucocyte counts may be increased.5,6 Infection of any joint, including the TMJ, has not been reported to date. General symptoms of septic arthritis of the TMJ are similar to those of any other infection: increased body temperature, fatigue, and malaise, and sometimes an increase in certain inflammatory markers. Local symptoms include pain, swelling, redness in the pre-auricular region, and limited mouth opening. An open bite on the affected side may be present due to increased joint fluid.1–3,7 Common complications of septic arthritis are osteomyelitis, fibrosis, joint dysfunction, degenerative changes of the joint structures, and ankylosis. When this
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
YIJOM-3506; No of Pages 5
4
Levorova et al.
occurs in childhood, growth disorders on the affected side of the mandible may be present.2,8 The causative pathogens of septic arthritis of the TMJ are usually bacteria, the most common being Streptococcus, Staphylococcus, Haemophilus, Neisseria, and Mycobacterium. Pathogen penetration into the joint proceeds via haematogenous spread, direct extension from a nearby infection, or through an open wound.1,2 Risk factors for the development of septic arthritis include systemic and autoimmune diseases (e.g., rheumatoid arthritis, diabetes mellitus, lupus erythematosus, and immunosuppression), the long-term use of steroids, and a blunt trauma causing damage to the local vessels, with greater bacterial access into the joint.7 In the case presented herein, the patient had arterial hypertension, bronchial asthma, and fibrous mastopathy, but no other internal, oncological, or rheumatoid diseases. In the absence of subjective complaints and negative laboratory findings, it can be presumed that no other inflammation was present. Had simultaneous inflammation in another body region been identified, it would have been presumed to have been caused by the same bacterium – R. ornithinolytica. Only one case of septic arthritis as a complication of TMJ arthroscopy has been reported to date.1 In this case, McCain et al. described septic arthritis as an early complication; the symptoms became evident on day 6 postoperative (increased pain and swelling, and spontaneous drainage of pus from the puncture sites). Furthermore, even articles presenting the complications of TMJ arthroscopy for large cohorts of patients have not described cases of septic arthritis.9,10 In the case presented here, the first symptoms were noted on day 43 postoperative; and thus would not represent an early complication of surgery. The surgical wound after arthroscopy showed signs of normal healing, with no signs of infection and no exudation. This would disprove the hypothesis of penetration of the infection through the wound, except if the very small wound caused by the 1.9-mm arthroscopic cannula is considered. If haematogenous spread and spread by direct extension of another infection are excluded, then penetration could be presumed to have occurred through the wound postoperatively. Intravenous antibiotic prophylaxis was administered as recommended by the department of microbiology. The arthroscopy procedure was done under
sterile conditions. Inadequate sterilization or contamination during the period after sterilization could not be confirmed or excluded, so the possibility that the infection was introduced via the arthroscope or other materials/devices used during the procedure cannot be ruled out. Contamination with R. ornithinolytica present as part of the normal skin flora on the body surface or through cerumen has not been described previously, so it was assumed that the patient’s body surface was not colonized by this bacterium (a smear was not performed). The epidemiologist ruled out the presence of R. ornithinolytica in the patient’s area of residence (Jihlava, eastern Bohemia). The patient was asked about having ingested fish during the 2 weeks prior to the onset of the swelling (to exclude the introduction of infection with food), but she did not remember eating any fish. Scombroid syndrome takes its name from the Scombridae family. This family includes tuna fish, which are widely known and often eaten. It was considered unlikely that the patient had ingested any, but if so, she was without signs of enteritis. Septic arthritis of the TMJ is resolved by pus drainage and antibiotic therapy according to microbiological findings (with broad spectrum antibiotics in the acute stage). Arthroscopic lysis and lavage allows early elimination of degenerative irreversible changes, which may be present 7–10 days after joint infection. Arthroscopy rather than arthrocentesis is recommended for more extensive lavage and direct visualization.2,3,7 In conclusion, a rare case of R. ornithinolytica infection of the TMJ is reported here. During a period of 1 year after infection by this bacterium, progressive and destructive degenerative changes in the TMJ led to the need for a total alloplastic joint replacement. A clear route of infection was not revealed, but it is presumed that this was a late complication of arthroscopy. The presence of an acute or chronic infection could not be proven either clinically or through laboratory findings. Despite this, the pathogenic organism or inflammatory process was still present in the soft tissues of the joint. Treatment methods such as antibiotic therapy alone or in combination with arthrocentesis are not sufficient for the treatment of septic arthritis of the TMJ. The cause of this septic arthritis is not completely clear. It is alarming that previous surgery could have had such severe consequences. The treatment of such a case can be challenging, as there is currently a lack of evidence and information regarding definitive curative therapy.
Funding
There were no sources of funding for this research. Competing interests
No competing interests. Ethical approval
Institutional ethics approval was obtained (No. 1280/15 IS-IV). Patient consent
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Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
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TMJ septic arthritis caused by rare Raoultella 10. Gonza´lez-Garcı´a R, Rodrı´guez-Campo FJ, Escorial-Herna´ndez V, Mun˜oz-Guerra MF, Sastre-Pe´rez J, Naval-Gı´as L, et al. Complications of temporomandibular joint arthroscopy: a retrospective analytic study of 670 arthroscopic procedures. J Oral Maxillofac Surg 2006;64:1587–91.
Address: Jitka Levorova Department of Oral and Maxillofacial Surgery General Hospital and First Medicine Faculty U Nemocnice 2 str.
Prague 2 120 00 Czech Republic Tel: +420 224 96 3191; Fax: +420 224 96 3193 E-mail: [email protected]
Please cite this article in press as: Levorova J, et al. Septic arthritis of the temporomandibular joint caused by rare bacteria Raoultella ornithinolytica, Int J Oral Maxillofac Surg (2016), http://dx.doi.org/10.1016/j.ijom.2016.09.008
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