Streptococcus pneumoniae septic arthritis complicating hip osteonecrosis in adults: case report and review of the literature.

Abstract: A 42-year-old patient presented acutely with bacteremic pneumococcal pneumonia along with metastatic pneumococcal infection of the hip joint. Diagnostic evaluation revealed evidence of a pre-existing bilateral hip osteonecrosis. The osteonecrotic changes were attributed to chronic alcohol abuse and/or an old motor vehicle accident. Appropriate therapy was promptly instituted and the septic arthritis responded well, necessitating hip aspiration only once. A few months later, the patient had no permanent sequelae of the infection.

Key Words: Streptococcus pneumoniae, infectious arthritis, osteonecrosis, alcohol abuse, hip joint

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Streptococcus pneumoniae septic arthritis remains an uncommon infection in the adult population. Many physicians who treat patients with pneumococcal infections, mainly pneumonias, may not be familiar with this entity. This could result in significant morbidity related to the hip joint. We used the MEDLINE database to search the English language literature for similar cases. The occurrence of this syndrome as a complication of hip osteonecrosis has been rarely reported.

Case Report

A 42-year-old African-American man was brought to the emergency room with a 2-day history of steadily increasing right hip pain and a 3 to 4 day history of fevers with chills, cough productive of greenish sputum, generalized weakness and a recent accidental fall. Review of systems was otherwise unrevealing. He had sustained head and pelvic trauma during a motor vehicle accident (MVA) two years previously with residual left-sided spasticity and weakness. He was using a cane and, occasionally, a wheelchair. After the MVA, he had also developed seizures, necessitating antiepileptic therapy. There were no prior rheumatologic or other chronic diseases or infections. In addition, the patient gave a history of chronic alcohol abuse (40 ounces of beer per day for 25 years), as well as cocaine abuse. He denied other risk factors for human immunodeficiency virus (HIV) infection and reported a negative tuberculin skin test (PPD) 18 months before his presentation. He denied any sick contacts and was unemployed and living in a shelter house.

At admission, his temperature was 103.8[degrees]F and respiratory rate was 20 breaths/min. He was hemodynamically stable and in minimal respiratory distress with a pulse oximetry of 93% on room air. He was appropriately oriented with no signs of meningitis. Signs of consolidation were noted at the left base and a 2/6 midsystolic murmur was heard over the aortic area. He had a moderately sized ecchymosis on his anterior hip area and his right hip was tender to palpation with limited range of motion in all directions. Hip muscle group strength was 5/5 on the right and 4/5 on the left, while sensory examination was grossly normal. Left-sided spasticity was noted. All peripheral pulses were palpable.

Laboratory evaluation revealed leukocytosis with a left shift (WBC 12,400 cells/[mm.sup.3], 82% neutrophils), an erythrocyte sedimentation rate (ESR) of 103 mm in the first hour, as well as normal liver and kidney function tests. Toxicology screen was positive for cocaine and opiates while phenytoin level was suboptimal. A chest film revealed a left lower lobe infiltrate, while computed tomography of the pelvis showed bilateral hip osteonecrosis, increased synovial fluid and early right femoral head osteomyelitis. Aspiration of the hip yielded 7 [cm.sup.3] of purulent fluid with a fluid white cell count (WBC) of 42,000/[mm.sup.3] (88% polymorphonuclear cells). The fluid Gram stain revealed no organisms but the fluid culture grew S pneumoniae (SP) at 24 hours with a penicillin minimum inhibitory concentration (MIC) of 0.06 [micro]g/mL. The same organism grew from blood and sputum cultures.

The patient was originally given broad spectrum antibiotics which were subsequently changed to IV penicillin G. A transthoracic echocardiogram revealed no valve vegetation, while a repeat PPD and an HIV test were negative. He promptly defervesced while hip symptoms improved slowly. No more hip aspirations or open hip drainage were implemented. He was transferred to a rehabilitation center after 12 in-hospital days. He completed 4 weeks of IV penicillin and 2 weeks of oral penicillin therapy. Upon discharge 6 weeks later, he was ambulating with a cane, and 6 months later, he had completely recovered from his infection.

Discussion

S aureus and nongroup A [beta]-hemolytic streptococci are the most frequently involved Gram positive bacteria in adult septic arthritides. S pneumoniae septic arthritis (SPSA) has always been an uncommon infection (3-10% of septic arthritides). (1-4) Nevertheless it does occur, even in the modern antibiotic era, as concluded from several case reports and a few reviews. The joint most frequently involved in SPSA is the knee, followed by the shoulder, the elbow and multiple joints. (2-5) Spine cases are also uncommon. (6) Adults in their 5th or 6th decade seem to be more vulnerable to SPSA, with males predominating in some, but not all, series. (2,3,5-7) Several underlying articular and nonarticular illnesses and host factors have been reported to predispose to SPSA. Most of them will be mentioned during the more focused discussion on hip SPSA. Prior joint trauma is one of them. (2,6) Rarely, no underlying condition can be identified.

SPSA may involve one or more joints. Patients with rheumatoid arthritis (RA) are predisposed to polyarticular septic arthritis irrespective of etiology (up to 25%). (1) Uncommonly, prosthetic joints, usually knees and hips, are involved. (7)

SPSA is associated with pneumonia in 29 to 75% of cases, and less frequently with meningeal, endocardial or pleural space involvement (or combinations of those). (2-4,6) No extra-articular primary focus is found in 10 to 50% of patients. (3,4) Conversely, SPSA itself will seldom complicate pneumococcal pneumonia (0.3%). (3,5) Joint symptoms may appear before, during or after the primary infection has become evident, (2-4,6) and in over 50% of cases, temperature is elevated on presentation. (3,6) Symptoms and signs of arthritis are usually pronounced. A few patients present with a fulminant picture. The WBC count is often elevated with a left shift, as is the ESR (>90 mm/h in over 60% of cases). (3,6) Bacteremia occurs in 50 to 90% of SPSA cases. (3,6) The joint fluid is usually purulent, although it may look innocuous. (2,5) Positive fluid Gram stains and cultures are obtained in 70 to 85% and 60 to 85% of patients, respectively. (3,5) In most cases with negative cultures, prior antibiotics had been given. When serotyping is done, multiple serotypes are found, the majority of which are included in the 23-valent pneumococcal vaccine. (3,7)

IV penicillin has been the drug of choice in SPSA, with [3.sup.rd] generation cephalosporins and vancomycin serving as alternatives. (2-4,6) Many cases of SPSA have been treated successfully with medical treatment alone, along with one or more closed-needle aspirations. (3) Some centers use routine surgical lavage, either arthroscopic or open. (4) Prosthetic joint SPSAs have also been adequately managed with antibiotics and surgical drainage alone. (3) The prognosis in SPSA is usually favorable with appropriate antibiotics and joint drainage. (4-6) SPSA hip infections (SPSAH) are rare in adults. (4,5,7-17) Table 1 illustrates some primary features of the few SPSAH cases we found in the English medical literature. Some data were not available for all cases. Similar scattered case reports and a few case series can also be found in the non-English literature. The mean age of the 18 SPSAH cases reviewed (including ours) was 63 years, and the male to female ratio was 1:1. Several cases have involved relatively young individuals, like our patient.

Underlying conditions included rheumatoid arthritis (RA) with or without corticosteroid use (in one case the patient also received anticytokine therapy), (8,11,17) alcohol abuse. (4,5,7) degenerative joint disease, (5,12,13) chronic obstructive pulmonary disease, (4,12,13) chronic heart disease, (4,13) end-stage renal disease with secondary hyperparathyroidism, (14) malignancy, (4,9) splenectomy, (4,9,15) gastric surgery, (4,9) diabetes mellitus, (12) osteonecrosis (7) and graft-versus-host disease. (7)

We were able to identify only two more adult SPSAH cases superimposed on hip osteonecrosis. (7) Osteonecrosis usually occurs secondary to trauma but has also been associated with corticosteroid use, alcoholism, sickle cell anemia, infections, marrow infiltrating diseases, hypertriglyceridemia, HIV infection and autoimmune disorders. (14) The femoral head is most commonly affected (up to 98% bilaterally). (18) We attributed our patient's condition to prior trauma and chronic alcohol abuse.

Of note, pneumococcal arthritis seems to occur more frequently in children. In one series, S pneumoniae (SP) was the second most common isolate from children younger than 2 years with bacterial arthritis. (19) In a retrospective review of cases of osteoarticular infections in children with sickle cell anemia, SP was the most common isolate from pure arthritis cases. (20) Ischemic bone necrosis in sickle cell anemia may predispose to SP infection, while a similar pathogenetic mechanism may have occurred in our patient.

In 6 of the 18 SPSAH cases (33%), there was polyarticular involvement. Half of the SPSAH cases involved prosthetic hips. (7,10-13,16,17) There was concomitant pneumonia in 11/18 SPSAH cases (61%), meningitis in 3 and contiguous abscesses in 3 cases.

In our case, and in most SPSAH cases we found, there was a relatively short duration of symptoms, of 3 to 5 days. Interestingly, in 2 prosthetic SPSAH cases, the diagnosis was made with a delay of several months. (11,12) Temperature was always elevated on presentation for those cases where data was available and the majority presented with intense local symptoms and signs of inflammation. The WBC count was often, but not always, elevated (Table 2). The ESR was also usually elevated. Bacteremia occurred in most SPSAH cases (15/17, data missing for 1 patient). The joint fluid was usually purulent (7 of 9 cases with fluid description). The fluid WBC count was always increased with a left shift. Positive fluid Gram stains were infrequently noted (2 cases only, contrary to previous reports on SPSA) but joint fluid culture positivity was the rule. Few data on prior pneumococcal vaccinations were found. Our patient had not been vaccinated. Reports of infections by moderately or even highly resistant strains are emerging. Two SPSAH strains exhibited a penicillin MIC of 0.125 [micro]g/mL and one of them was treated with vancomycin and rifampin. (7,9) Two other strains were penicillin resistant and intermediately resistant to ceftriaxone. (7,13) Our patient's strain was not typed and was highly susceptible to penicillin.

Radiographic findings in the SPSAH cases reviewed included those of the underlying joint disease and, in addition, soft tissue swelling or effusion, joint destruction and adjacent osteomyelitis, prosthesis abnormalities and contiguous abscesses.

Most of the SPSAH patients were treated for 3 to 6 weeks of IV therapy, followed by various courses of oral therapy in some cases.

Among the 9 SPSAH cases involving natural hips, surgery (one or more procedures) was implemented in 4, and the remaining 5 were managed medically (although in one case, the patient died before the planned surgery). Among the prosthetic hip cases, surgical drainage was implemented in 7 of 9 cases and 5 of 9 prostheses were preserved. In our review, 14 of 18 patients survived and adequate mobility was preserved in 11 of the 14. Three patients died due to fulminant pneumococcal sepsis while one patient died because of miliary tuberculosis.

Conclusion

In summary, we describe an unusual case of S pneumoniae septic arthritis on top of hip osteonecrosis along with accompanying bacteremic pneumonia in an adult. Even though SPSA remains an unusual type of infection, it needs to be included in the differential diagnosis in cases of concomitant pulmonary and/or meningeal and joint infection.

Acknowledgments

Many thanks to Dr. John J. Ross for his prompt help with the literature search.

References

1. Goldenberg DL, Reed JI. Bacterial arthritis. N Engl J Med 1985;312: 764-771.

2. Torres J, Rathbun HK, Greenough WB. Pneumococcal arthritis: report of a case and review of the literature. Johns Hopkins Med J 1973;132: 234-241.

3. Ispahani P, Weston VC, Turner DP, et al. Septic arthritis due to Streptococcus pneumoniae in Nottingham, United Kingdom, 1985-1998. Clin Infect Dis 1999;29:1450-1454.

4. James PA, Thomas MG. Streptococcus pneumoniae septic arthritis in adults. Scand J Infect Dis 2000;32:491-494.

5. Kauffman CA, Watanakunakorn C, Phair JP. Pneumococcal arthritis. J Rheumatol 1976;3:409-419.

6. Turner DP, Weston VC, Ispahani P. Streptococcus pneumoniae spinal infection in Nottingham, United Kingdom: not a rare event. Clin Infect Dis 1999;28:873-881.

7. Ross JJ, Saltzman CL, Carling P, et al. Pneumococcal septic arthritis: review of 190 cases. Clin Infect Dis 2003;36:319-327.

8. Good AE, Gayes JM, Kauffman CA, et al. Multiple pneumococcal pyarthrosis complicating rheumatoid arthritis. South Med J 1978;71:502-504.

9. Merle-Melet M, Tortuyaux JM, Condette S, et al. Early postsplenectomy arthritis caused by penicillin-resistant Streptococcus pneumoniae. Eur J Clin Microbiol Infect Dis 1996;15:159-160.

10. Wollner A, Gilon D, Mattan Y, et al. Pneumococcal infection of a prosthetic hip joint. Int Orthop 1989;13:135-136.

11. Sanzen L. Spread of Streptococcus pneumoniae between artificial joints in a rheumatoid patient. Acta Orthop Scand 1991;62:597-598.

12. Maniloff G, Greenwald R, Laskin R, et al. Delayed postbacteremic prosthetic joint infection. Clin Orthop 1987;223:194-197.

13. Raad J, Peacock J. Septic arthritis in the adult caused by Streptococcus pneumoniae: a report of 4 cases and review of the literature. Semin Arthritis Rheum 2004;34:559-569.

14. Ciernik IF, Gerster JC, Burckhardt P. Destructive pneumococcal septic arthritis in end-stage renal disease. Clin Rheumatol 1997;16:477-479.

15. Rivera P, Wilson M, Rubinfeld RS. Pneumococcal keratitis, bacteremia, and septic arthritis in an asplenic patient. Cornea 1996;15:434-436.

16. Mallory TH. Sepsis in total hip replacement following pneumococcal pneumonia. A case report. J Bone Joint Surg Am 1973;55:1753-1754.

17. Baghai M, Osmon DR, Wolk DM, et al. Fatal sepsis in a patient with rheumatoid arthritis treated with etanercept. Mayo Clin Proc 2001;76: 653-656.

18. Assouline-Dayan Y, Chang C, Greenspan A, et al. Pathogenesis and natural history of osteonecrosis. Semin Arthritis Rheum 2002;32:94-124.

19. Jacobs NM. Pneumococcal osteomyelitis and arthritis in children. A hospital series and literature review. Am J Dis Child 1991;145:70-74.

20. Syrogiannopoulos GA. McCracken GH Jr, Nelson JD. Osteoarticular infections in children with sickle cell disease. Pediatrics 1986;78:1090-1096.

Ioannis G. Baraboutis, MD, Vasilios Papastamopoulos, MD, and Athanasios Skoutelis, MD

From Evaggelismos General Hospital, Athens, Greece.

Reprint requests to Ioannis G. Baraboutis, MD, Evaggelismos General Hospital, Ipsilantou 45-47, Athens, Greece, 10676. Email: [e-mail]

Accepted March 28, 2007.

RELATED ARTICLE: Key Points

* Streptococcus pneumoniae septic arthritis should be suspected in patients with arthritic symptoms and a concomitant pneumococcal infection.

* S pneumoniae septic arthritis of the hip is an uncommon infection, associated with certain predisposing factors. Osteonecrosis is an exceedingly rare predisposing factor.

* Prompt diagnosis and treatment are essential to prevent joint damage.

Table 1. Clinical features of reviewed cases of pneumococcal hip
arthritides

Patient
(ref)       Age/sex  Comorbidities          Joints involved

 1 (4)      43/M     None                   Hip
 2 (4)      70/M     Splenectomy, partial   Hip, sternoclavicular
                       gastrectomy,
                       pernicious anemia
 3 (4)      63/M     Alcohol abuse, COPD,   Hips, ankle, 1st MTP
                       lung cancer, atrial
                       fibrillation
 4 (5)      48/M     Alcohol abuse, DJD     Hip
 5 (7)      73/F     DJD, COPD              Prosthetic hip
 6 (7)      79/F     DJD, COPD, CHF         Prosthetic hip
 7 (8)      77/M     Rheumatoid arthritis   Hips, wrists, shoulders,
                                              knees, sternoclavicular,
                                              2nd PIP (total of 10
                                              joints involved)
 8 (9)      78/M     Splenectomy, total     Hip (11 days
                       gastrectomy/           postoperatively)
                       pancreatectomy,
                       gastric cancer
 9 (10)     74/F     Hip replacement 4      Prosthetic hip
                       years ago
10 (11)     70/F     Rheumatoid arthritis,  Prosthetic hip, knees, psoas
                       hip replacement 12     abscess
                       years ago
11 (12)     75/F     Diabetes mellitus,     Prosthetic hip, overlying
                       hip replacement 1      soft tissue abscess
                       year ago
12 (13)     50/F     Obesity, prior         Prosthetic hip
                       prosthetic hip
                       infection
13 (13)     47/M     ALL/BMT, GVHD,         Prosthetic hips, shoulders
                       steroid therapy,
                       b/1 hip replacement
14 (14)     67/F     ESRD/HD,               Hip
                       hyperparathyroidism
15 (15)     66/F     Splenectomy            Hip
16 (16)     69/M     Hip replacement        Prosthetic hip
17 (17)     37/F     B/1 hip replacement,   Prosthetic hip, knee, ankle
                       rheumatoid
                       arthritis, steroid
                       and etanercept
                       therapy
18 (case    42/M     Avascular hip          Hip
  patient)             necrosis, alcohol
                       abuse

                                                        Length of
                                                        antimicrobial
Patient                                       Blood     treatment
(ref)       Other sites      Fever            culture   (IV plus oral)

 1 (4)      None             NA               Negative  20
 2 (4)      Lung             NA               Positive  33
 3 (4)      Lung, meninges   NA               Positive  19
 4 (5)      Lung, meninges   Present          Positive  23-44 (a)
 5 (7)      Lung, thigh      NA               Positive  NA
              (abscess)
 6 (7)      None             NA               Positive  6 weeks IV Abx,
                                                          then oral Abx
                                                          indefinitely
 7 (8)      Lung, meninges,  40.7[degrees] C  Positive  1,5
              psoas muscle
 8 (9)      None             39[degrees] C    Positive  42
 9 (10)     Lung             38[degrees] C    NA        42
10 (11)     Lung             Present          Positive  Indefinitely
11 (12)     Lung             37.5[degrees] C  Positive  28
12 (13)     Sinuses          NA               Negative  6 weeks IV Abx,
              (clinical                                   then oral Abx
              diagnosis)                                  for several
                                                          weeks
13 (13)     None             Present          Positive  6 weeks IV Abx,
                                                          then oral Abx
                                                          indefinitely
14 (14)     Lung             Present          Positive  10 weeks IV Abx
15 (15)     Cornea           Present          Positive  Several weeks of
                                                          IV Abx, then
                                                          oral Abx
                                                          indefinitely
16 (16)     Lung             NA               Positive  IV Abx followed
                                                          by oral Abx
                                                          (total of 3
                                                          months)
17 (17)     Posterior thigh  Present          Positive  IV Abx
              (necrotizing
              fasciitis)
18 (case    Lung             Present          Positive  6 weeks
  patient)

Patient
(ref)       Joint lavage-no. of procedures  Outcome

 1 (4)      0                               Mobile
 2 (4)      1                               Mobile
 3 (4)      6                               Mobile
 4 (5)      1 (open surgery)                Died of miliary
                                              tuberculosis
 5 (7)      1 (prosthesis                   Died of sepsis
              extracted)
 6 (7)      Arthrocentesis only             Infection suppressed
 7 (8)      0 (hips explored                Died 18 hours after
              postmortem)                     admirssion
 8 (9)      0                               Mobile
 9 (10)     1 (open surgery,                Mobile
              prosthesis
              preserved)
10 (11)     1 (prosthesis                   Mobile
              extracted,
              replaced 4 weeks
              later)
11 (12)     1 (open surgery,                Mobile
              prosthesis
              preserved)
12 (13)     1 (open surgery.                Pain-free but unable
              prosthesis                      to ambulate
              preserved)
13 (13)     Arthrocenteses only             Mobile
              (prostheses
              preserved)
14 (14)     Arthrocentesis only             Mobile with crutches
15 (15)     1 (open surgery)                Mobile
16 (16)     1 (prosthesis                   Mobile with crutches
              removed)
17 (17)     1 (prosthesis                   Died within 2 days
              removed)
18 (case    Arthrocentesis only             Mobile
  patient)

Ref, reference; M, male; F, female; NA, not available; pt, patient; MTP,
metatarsophalangeal joint; PIP, proximal interphalangeal joint; COPD,
chronic obstructive pulmonary disease; DJD, degenerative joint disease;
CHF, congestive heart failure; IV, intravenous; Abx, antibioties; RA,
rheumatoid arthritis; ALL/BMT, acute lymphocytic leukemia/bone marrow
transplantation; GVHD, graft versus host disease; ESRD/HD, end-stage
renal disease/hemodialysis.
(a) Range of days of therapy in case-series where the case was included.

Table 2. Laboratory features of reviewed cases of pneumococcal hip
arthritides

Patient     Periph
(ref)       WBC (/[mm.sup.3])  % neutrophils      Character of fluid

 1 (4)      NA                 NA                 NA
 2 (4)      NA                 NA                 NA
 3 (4)      NA                 NA                 NA
 4 (5)      13900              96                 Thick, yellow
 5 (7)      Not specified (b)  Not specified (b)  Purulent
 6 (7)      Not specified (b)  Not specified (b)  Not specified (b)
 7 (8)      5100               NA                 Purulent
 8 (9)      Increased          12500/[mm.sup.3]   NA
 9 (10)     20000              80 (10% band       Purulent
                                 forms)
10 (11)     NA                 NA                 Purulent
11 (12)     10700              81                 Purulent
12 (13)     NA                 NA                 NA
13 (13)     NA                 NA                 NA
14 (14)     5200               40                 Hemorrhagic
15 (15)     12200              62                 NA
16 (16)     NA                 NA                 Purulent
17 (17)     3000               89                 NA
18 (case    12400              82                 Purulent
  patient)

Patient     Joint fluid
(ref)       WBC (/[mm.sup.3])  % neutrophils      Joint fluid smear

 1 (4)      Increased          Increased          Not specified (a)
 2 (4)      Increased          Increased          Not specified (a)
 3 (4)      Increased          Increased          Not specified (a)
 4 (5)      NA                 NA                 Negative
 5 (7)      Not specified (b)  Not specified (b)  Not specified (b)
 6 (7)      Not specified (b)  Not specified (b)  Not specified (b)
 7 (8)      Increased          Increased          Positive
 8 (9)      NA                 NA                 NA
 9 (10)     40000              99                 NA
10 (11)     Increased          Increased          NA
11 (12)     Increased          Increased          Negative
12 (13)     40750              90                 NA
13 (13)     462000             NA                 NA
14 (14)     22500              86                 Negative
15 (15)     NA                 NA                 Negative
16 (16)     NA                 NA                 Negative
17 (17)     NA                 NA                 Positive
18 (case    42000              88                 Negative
  patient)

Patient
(ref)        Joint fluid culture

 1 (4)       Not specified (a)
 2 (4)       Not specified (a)
 3 (4)       Not specified (a)
 4 (5)       Negative (had received 3 weeks of Abx)
 5 (7)       Positive
 6 (7)       Not specified (b)
 7 (8)       Positive
 8 (9)       NA
 9 (10)      Positive
10 (11)      Positive
11 (12)      Positive
12 (13)      Positive
13 (13)      Positive
14 (14)      Positive
15 (15)      Positive
16 (16)      Positive
17 (17)      Positive
18 (case     Positive
   patient)

Ref, reference; NA, not available; WBC, white blood cells.
(a) No individual data given but positive fluid smear in 8/14 and
positive fluid culture in 13/14 cases for the whole series. (4)
(b) No individual data given but serum leucocytosis in 61%, mean joint
WBC of 127,000 cells/[mm.sup.3] with 92% neutrophils, positive fluid
smear in 77% and positive fluid culture in 84% for the whole series. (7)