Streptococcus gallolyticus prosthetic joint infection is associated with colorectal carcinoma, a case report and literature review

V.F. Zwart1 W.J.J. Bekkers1 A.M.R. Raaijmaakers1 R.C.I. van Geenen1 1 Department of Orthopaedic Surgery, Amphia Hospital, Breda, The Netherlands Corresponding author: V.F. Zwart, vfzwart@hotmail.com

We present a case of a patient with a late onset Streptococcus gallolyticus infection of a total knee arthroplasty together with endocarditis. An association between Streptococcus gallolyticus endocarditis or bacteremia and colorectal carcinoma is well described in literature. Because of a Streptococcus gallolyticus prosthetic joint infection a colorectal carcinoma was suspected, and this was proven by colonoscopy and histopathological examination. A two-stage revision of the knee was performed. Between the first and second stage of the revision the patient underwent a left sided hemicolectomy. We reviewed the literature regarding prosthetic joint infections with Streptococcus gallolyticus and its association with a malignancy of the colon. This case demonstrates the importance of accurate interpretation of unusual microbiology findings in prosthetic joint infection.

Introduction Prosthetic joint infection (PJI) with Streptococcus gallolyticus subsp. gallolyticus (Sgg) is a very rare complication and is associated with colorectal carcinoma (CRC). We present a case of a 69-year-old male patient with a late onset Sgg infection after total knee arthroplasty (TKA) together with an endocarditis in the presence of an underlying CRC.

Patient A 69-year-old patient with a right sided TKA implanted in 2009 was presented to our hospital with onset of knee pain for seven weeks. The patient was referred to our hospital because of positive synovial cultures with Streptococcus gallolyticus subs. gallolyticus, obtained from the right knee by joint aspiration. The patient had a medical history of a pancreatectomy in 2017 because of an intraductal papillary mucinous neoplasm (non-malignant) followed by type I diabetes mellitus with polyneuropathy and hypertension. Intravenous cefuroxime and tobramycin were administered before he was referred to our hospital. On physical examination the patient had cold shivers. The right knee was painful with slight effusion but did not show any redness or warmth. Range of motion was 0˚-135˚ and axial load was painful. Conventional X-rays of the knee showed no signs of loosening. Serum C-reactive protein was 86 mg/L and white blood cell count 21.1 10⁹/L. Right knee aspiration showed a leucocyte count of 31.02 10⁹/L with 94% polymorph nuclear cells.

Intervention Before intervention, one set of blood cultures was positive with Sgg. In first stage revision, the prosthesis was removed, debridement and synovectomy of the right knee was performed, and a dynamic prefabricated vancomycin and gentamycin spacer (VancogenX®-HV Tecres) was placed. One of the six tissue cultures taken during surgery was positive with Sgg. Intravenous benzyl penicillin was administered for two weeks followed by oral amoxicillin for the remainder of the three months of antibiotic treatment. Although classical symptoms of CRC such as rectal bleeding, mucus discharge or significant weight loss were absent, the gastroenterologist was consulted, because of the known association between Sgg positive cultures and CRC. A colonoscopy showed a neoplasm in the colon descendens which was suspect for CRC. Histopathological examination of biopsies confirmed a good to moderately differentiated adenocarcinoma of 1,3 cm in size. After the first stage revision of the TKA, a left sided hemicolectomy was performed with creation of a permanent end colostomy. Because of the likely hematological spread of Sgg shown by positive blood cultures and a more frequent occurrence of endocarditis in combination with underlying CRC, the cardiologist was consulted. The patient had no existing cardial complaints or signs of decompensation. Transoesophageal echocardiography was performed which showed a vegetation on the tricuspid valve, suspect for infective endocarditis with a severe tricuspid valve failure. The endocarditis was treated conservatively with antibiotics in accordance with the guidelines for endocarditis.

Comparison The patient was treated according to the recommendations for treatment methods of PJIs compiled by the Dutch Orthopaedic Association1 (NOV). In this recommendation a PJI with the Sgg is not specifically described. In the occurrence of a PJI with Sgg the gastroenterologist should be consulted because of the association with CRC. Outcome The second stage revision was performed under conventional antibiotic prophylaxis when the patient showed no signs of infection and had several negative blood cultures after an antibiotic-free interval. Intravenous benzylpenicillin was administered until cultures taken during re-implantation showed no growth of microorganisms. Six weeks after surgery the patient was seen at the orthopaedic outpatient clinic and the surgical knee wound had healed well. The function of the knee showed a restriction of 10˚ in extension which was slowly improving under supervision of a physiotherapist. Follow-up by the cardiologist showed an asymptomatic insufficient tricuspid valve. The patient has a permanent end colostomy which is monitored by the stoma care unit. Literature and discussion We conducted a PubMed search and found five other case reports and a case series of patients with PJI caused by Sgg or Streptococcus bovis in association of CRC or gastrointestinal mucosal abnormality2-7. An overview of these cases is presented in table 1. All cases, including our case, present a patient with PJI and positive cultures with Streptococcus gallolyticus or – also formerly known as – Streptococcus bovis. Sgg is well known for causing bacteremia and endocarditis and has a strong association with CRC8,9. The precise role of Sgg in the development of CRC remains partly unclear. It has been questionable for a long time whether this organism plays an active role in the development of the tumour or its presence in the body – with resulting expression through infections elsewhere – is a consequence of the tumour and colonization of the colon with Sgg. Kumar et al. recently found results that indicate a tumour-promoting role of Sgg8. Except for one case, all PJI symptoms occurred after seven weeks or later after primary implantation of the prosthesis. PJI with Sgg is typically due to hematogenous spread7. Early PJIs occur up to three months after primary arthroplasty and are commonly associated with the surgery itself. Delayed PJIs usually appear 3 to 24 months postoperatively and may be due to a delayed growth of bacteria introduced at the time of surgery or related to hematogenous spread from a distant focus10. PJI occurs in a small proportion of all arthroplasty patients (1-3%) and is thought to rise into an absolute number in the coming years as the number of patients with an arthroplasty increases11. The majority of the PJI infections are caused by coagulase-negative staphylococci and Staphylococcus aureus. These microorganisms are more commonly found in the early onset infections, suggesting the intraoperative contamination scenario3,12. The streptococci species make up for 9% of all bacterial pathogens in PJIs13. Lora-Tamayo et al. found that Streptococcus bovis was responsible in 1,5% of all streptococci PJIs14, which makes a PJI with the Streptococcus gallolyticus subsp. gallolyticus a very rare complication. The treatment of PJI is intensive, complex, costly, and pre-eminently multidisciplinary. The gastroenterologist should be consulted if this causative agent is found from cultures. It is clinically important to emphasize the association between Sgg and CRC in PJI, because it may have major consequences for the patient if this link is not known or recognized. To our knowledge, this is the first case of confirmed infection with Sgg in the unique combination of late onset PJI together with endocarditis in the presence of an underlying CRC.

Conclusion Prosthetic joint infections caused by Streptococcus gallolyticus are associated with colorectal carcinoma. Awareness can result in an early diagnosis and treatment of an otherwise asymptomatic CRC. Disclosure statement There were no conflicts in interest.

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