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CMAJ
CMAJ
9711805
CMAJ : Canadian Medical Association Journal
0820-3946
1488-2329
CMA Impact Inc.

39284609
10.1503/cmaj.240259
196e1038
Practice
Cases
Invasive meningococcal disease presenting with tenosynovitis
Katyukha Andriy MD
Sukhdeo Sharon MD MSc
Andany Nisha MD MPH
Lefkowitz Ariel MDCM
Temerty Faculty of Medicine and Sunnybrook Health Sciences Centre, University of Toronto, Toronto, Ont.
Correspondence to: Andriy Katyukha, a.katyukha@mail.utoronto.ca
16 9 2024
16 9 2024
196 30 E1038E1040
© 2024 CMA Impact Inc. or its licensors
2024
https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY-NC-ND 4.0) licence, which permits use, distribution and reproduction in any medium, provided that the original publication is properly cited, the use is noncommercial (i.e., research or educational use), and no modifications or adaptations are made. See: https://creativecommons.org/licenses/by-nc-nd/4.0/
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pmcKEY POINTS

Tenosynovitis and arthritis are rare manifestations of invasive infection with Neisseria meningitides; infection with Neisseria gonorrhoeae is more commonly associated with these manifestations.

Invasive meningococcal disease can occur in the absence of meningitis.

Mortality associated with invasive meningococcal disease is 8.3%–12%, which underscores the importance of prompt recognition and antimicrobial therapy.

Close contacts should receive chemoprophylaxis as guided by local public health units.

A 67-year-old man presented to the emergency department with a 5-day history of nausea, malaise, and subjective fevers. Three days before presentation, he had also developed swelling of his left wrist and ankle. He denied experiencing any neurologic symptoms, rashes, or changes to his vision.

The patient had returned from a cruise in the United States and British Virgin Islands 1 week before symptom onset. During the cruise, he did not leave the ship, did not consume any food or water from the mainland, and did not have any known sick contacts. He did not interact with any animals, did not report any insect bites, and remained well throughout the cruise. On returning home, he had 3 sexual encounters with new partners prior to the onset of his symptoms. He denied any genitourinary changes. His medical history included hypertension, remote immunoglobulin A vasculitis, and a chronically elevated prostate-specific antigen level.

In the emergency department, the patient was hypertensive, with a blood pressure reading of 148/81 mm Hg, but his vital signs including temperature were otherwise normal. On physical examination, his left ankle and wrist were erythematous and painful, limiting range of motion (Figure 1). We elicited no tenderness along the joint lines and observed no palpable joint effusions, rash, or genitourinary lesions. The remainder of the physical examination was unremarkable.

Figure 1: Tenosynovitis of the wrist and ankle in a 67-year-old man with invasive meningococcal disease.

Initial laboratory investigations showed a leukocyte count of 12.8 × 109/L (normal 4.0–11.0 × 109/L), a platelet count of 118 × 109/L (normal 150–400 × 109/L), a potassium level of 2.5 mmol/L (normal 3.5–5.0 mmol/L), and a C-reactive protein level of 277 mg/L (normal 0–5 mg/L). Radiographic images of the left hand and foot showed soft-tissue swelling with no other abnormalities. Blood culture tests were ordered, along with an infectious work-up consisting of urine and throat nucleic acid amplification testing for gonorrhea and chlamydia, as well as HIV and syphilis testing. The patient was given a dose of ceftriaxone 2 g intravenously and was admitted to hospital to help manage pain and mobility concerns. The rheumatology service was consulted for possible joint aspiration but concluded that his presentation was consistent with tenosynovitis rather than arthritis. One of 2 blood culture sets showed growth of gram-negative diplococci. The infectious diseases team was consulted for suspected disseminated gonococcal infection in the context of the patient’s sexual history and presentation. The team agreed with the presumptive diagnosis and recommended continuing antimicrobial therapy with ceftriaxone pending finalization of his blood culture results.

The blood culture test subsequently identified the gramnegative diplococci as Neisseria meningitidis, and the provincial reference laboratory confirmed this to be serogroup C. The medical team re-examined the patient, again observing no clinical suggestion of meningitis. His symptoms improved in hospital while he was receiving ceftriaxone, and he completed a 7-day course of therapy for management of invasive meningococcal disease, with no evidence of central nervous system involvement. The local public health unit was notified.

At follow-up, 11 days after discharge, the patient’s wrist and ankle swelling had improved, although he continued to have reduced range of motion. Testing for gonorrhea, chlamydia, HIV, and syphilis returned negative.

Discussion

Neisseria meningitidis is a gram-negative diplococcus with a polysaccharide capsule. Structural differences in this capsule form the basis of serogrouping; N. meningitidis has 12 different serogroups, with serogroups A, B, C, W-135, and Y most frequently associated with invasive meningococcal disease in Canada. 1 Humans are the only known reservoir, and colonization of the nasopharynx results in many asymptomatic carriers.

In Canada and many other parts of the world, vaccination for N. meningitidis has decreased the incidence of infection. In Canada, from 2017 to 2021, 522 cases of invasive meningococcal disease were reported, corresponding to a cumulative incidence rate of 1.39 cases per 100 000 people.1 In Ontario, where our patient resided, 30 cases occurred over the same period, at a cumulative incidence rate of 0.21 cases per 100 000 people. More recently in Ontario, only 8 cases occurred from 2019 to 2023, a rate of 0.05 per 100 000.2

Invasive meningococcal disease refers to the isolation of N. meningitidis from a sterile site. The clinical presentation of invasive meningococcal disease can be variable, and outside of acute meningitis, suspicion for this infection may be low. In cases of clinical infection, symptoms can appear 2–10 days after exposure. 3 Transmission occurs via respiratory droplets or exchange of oropharyngeal secretions. Host risk factors include asplenia, complement deficiencies, and untreated HIV, especially in patients with low CD4 counts and high viral loads. Other risk factors include crowded housing (e.g., postsecondary dormitories and barracks), men having sex with men, working in health care or as microbiology technicians, and travel to endemic areas. For those at increased risk owing to host risk factors or high-risk activities, meningococcal vaccination is recommended.1

The most common clinical manifestation of invasive meningococcal disease is acute bacterial meningitis, with typical symptoms of fever, headache, nuchal rigidity, and altered mental status. In patients without meningitis, invasive meningococcal disease can present as meningococcemia (isolation of N. meningitidis in blood culture) in association with septic shock, often with a characteristic purpuric rash. Less common presentations of invasive meningococcal disease (2% of all cases) include pneumonia, 4 myopericarditis,4 conjunctivitis,4 and septic arthritis.5

Whereas case reports have been published of N. meningitidis mimicking the arthritis-dermatitis syndrome that is more classically associated with disseminated gonococcal infection (due to Neisseria gonorrhoeae), tenosynovitis without purulent arthritis is an extremely rare manifestation of invasive meningococcal disease, with only a few cases documented in the literature.

With respect to articular and periarticular involvement, the largest study to date examined 522 cases of invasive meningococcal disease from a single centre in Spain.5 Over 34 years, 39 cases of meningococcal arthritis were identified; of these, only 2 involved tenosynovitis without joint involvement. In the 39 cases studied, 95% of patients had an associated rash, 79% had associated meningitis, and 15% presented with shock, further underscoring that isolated joint or tendon involvement is quite a rare presentation. This is in contrast to disseminated gonococcal infection, in which tenosynovitis is found in 67% of patients, often as part of the classic triad of polyarthralgia, tenosynovitis, and pustular dermatitis.6

Our patient had not been vaccinated before travelling and had not been vaccinated as a child. Routine vaccination with monovalent conjugate C meningococcal vaccine occurs between 12 and 23 months of age, with an additional dose when children are 12 years old.7 Though he did not travel to endemic regions, spending a week on a cruise ship in close quarters with travellers from around the world may have placed him at higher risk of infection. Moreover, his recent sexual encounters also could have exposed him to N. meningitidis, through close contact with asymptomatic carriers. Routine vaccination for travel is not recommended; however, those with risk factors or those travelling to endemic areas may be considered for meningococcal vaccination, with a booster every 3–5 years.7

In a recent review, the case fatality rate for invasive meningococcal disease was 8.3%, climbing to 12% for those older than 65 years, further emphasizing the importance of prompt recognition and antimicrobial therapy.8 Close contacts of those with invasive meningococcal disease should be treated with chemoprophylaxis as guided by local public health units. In gonococcal infection, which more commonly presents with tenosynovitis, hematogenous spread of N. gonorrhoeae occurs in only 0.5%–3% of cases.6 In a study looking at 13 states across the US, 85.6% of patients with disseminated gonococcal infection were admitted to hospital, and 2.2% died.9 Adjunct therapies, such as steroids, have been studied in invasive meningococcal disease, and currently steroids, namely dexamethasone, can be used for cases of meningitis, though the evidence is less supportive for cases of shock from meningococcemia.10 Given the importance of prompt recognition and antimicrobial therapy, this case underscores the importance of asking pertinent historical questions related to travel and sexual history, particularly in older populations for whom these questions may be asked less often. Given our patient’s presentation, sexual history was critically important in arriving at a differential of disseminated gonococcal infection as a possible cause of this presentation, which guided the initial treatment.

This case highlights the range of presentations related to invasive meningococcal disease and raises important considerations related to vaccination, treatment, and chemoprophylaxis for exposed patients, and highlights the importance of following culture test results in cases of infectious syndromes.

Competing interests: Nisha Andany has participated as a site investigator for HIV clinical trials sponsored by Gilead, Janssen, and GSK (research funds paid to institution). No other competing interests were declared.

This article has been peer reviewed.

The authors have obtained patient consent.

Contributors: All authors contributed to the conception of the case report, and drafting and revising the article. All authors gave final approval of the version to be published and agreed to be accountable for all aspects of the work.
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