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Capnocytophaga sputigena as a cause of severe orbital cellulitis and subperiosteal abscess in a child

Published:November 05, 2020DOI:https://doi.org/10.1016/j.jcjo.2020.10.012
      Orbital cellulitis is an infection of the soft tissues lying behind the orbital septum. The etiologic agents differ across age group, but gram-positive cocci colonizing the skin and the nasopharynx, such as Staphylococcus and Streptococcus species, are most commonly identified in children. Haemophilus influenzae, a gram-negative facultative anaerobe, was frequently identified before the introduction of the Haemophilus vaccine in 1985. We report the case of a 15-year-old man who presented with a severe orbital cellulitis complicated by a subperiosteal abscess (SPA) secondary to Capnocytophaga sputigena.
      A 15-year-old man presented with a 1-week history of fever and right periorbital swelling and erythema. The patient reported a general feeling of discomfort, headaches, and diplopia with both upward and downward gaze. He denied any recent travel, or orbital trauma, but had an upper respiratory tract infection preceding the onset of symptoms. His medical history was significant for an attention-deficit/hyperactivity disorder, and his immunization status was up-to-date. He had no known medication allergy and had not been treated with antibiotics before presentation.
      On examination, the patient was febrile at 38.2°C, and visual acuity was 20/60 in the right eye and 20/20 in the left eye. Pupils were round and reactive to light, and there was no afferent pupillary deficit. The right eye showed motility restriction in upgaze and downgaze, moderate chemosis, and a 9-mm proptosis. The remainder of the examination was otherwise normal. At this stage, the blurred vision in the right eye was attributed to chemosis and pooling of tears. A computed tomography scan revealed a right-sided pansinusitis involving predominantly the ethmoid and maxillary sinuses. The adjoining orbit showed soft tissue stranding and a subperiosteal gas-containing fluid collection measuring 41 mm in anteroposterior dimension and 10 mm in thickness along the floor of the orbit (Fig. 1). The patient was diagnosed with a right orbital cellulitis complicated by an SPA. The presence of gas within the abscess was suspicious for a more aggressive anaerobic infection. Other laboratory tests included a normal white blood cell count of 7.7 × 109/L (reference range 4–10.5), an elevated erythrocyte sedimentation rate of 25 mm/hour (reference range 1–10), and an elevated C-reactive protein level of 117 mg/L (reference range 0–5).
      Fig 1
      Fig. 1A computed tomography scan showing a large, gas-containing inferomedial subperiosteal abscess, associated with a right-sided pansinusitis and orbital cellulitis.
      The patient was admitted to the hospital and treated with intravenous ceftriaxone, cloxacillin, and metronidazole. Although methicillin-resistant Staphylococcus aureus is a growing concern among children presenting with orbital cellulitis, our patient presented no risk factor for such infection. He also received nasal saline and oxymetazoline sprays to promote drainage of the sinuses, but the orbital signs continued to worsen during the first 24 hours of treatment (Fig. 2). The right eye developed a complete ophthalmoplegia, and visual acuity decreased to 20/120. The patient was brought to the operative room for maxillary antrostomy and lavage and drainage of the orbital SPA via a swinging eyelid approach. A culture from the SPA showed massive growth of a beta-lactamase-producing strain of C. sputigena. Postoperatively, the patient received oral prednisone in dosages of 50 mg once daily for 3 days as well as intravenous antibiotics. He showed significant improvement during the ensuing days. He was discharged after 9 days of parenteral therapy and was prescribed oral amoxicillin-clavulanic acid for a total of 4 weeks. At the 1-month follow-up visit, visual acuity was restored to 20/20 in both eyes and extraocular movements were full.
      Fig 2
      Fig. 2Clinical photograph showing severe periorbital swelling, chemosis, and hypertropia.
      Orbital cellulitis is uncommon in children, with an estimated incidence of 1.6 cases per 100 000 individuals.
      • Murphy C
      • Livingstone I
      • Foot B
      • Murgatroyd H
      • MacEwen CJ
      Orbital cellulitis in Scotland: current incidence, aetiology, management and outcomes.
      In most cases, it is the result of direct spread of bacterial infection from periorbital structures. Paranasal sinusitis and upper respiratory tract infection are among the most important risk factors, especially when the ethmoid and maxillary sinuses are involved. Orbital cellulitis can also be caused by hematogenous spread from bacteremia or direct inoculation.
      Capnocytophaga is a genus within the family Flavobacteriaceae. It includes different species of slow-growing gram-negative bacilli that are often considered as opportunistic pathogens. These capnophilic bacteria are facultative anaerobes, and they can be further classified according to their host preference. C. sputigena, C. gingivalis, C. ochracea, C. leadbetteri, C. granulosa, and C. haemolytica are part of the normal oral flora of humans, whereas C. canimorsus, C. canis, C. stomatis, and C. cynodegmi colonize the oral cavity of dogs and cats. C. canimorsus is responsible for most cases of infection in humans, with immunosuppression and animal-related injuries being the most common risk factors. C. sputigena rarely causes infection in the general population, but it can be responsible for periodontal diseases. The involvement of this bacterium in eye infections is very sporadic, with only a few case reports of keratitis.
      • Ormerod LD
      • Foster CS
      • Paton BG
      • Haaf J
      • Baker AS
      Ocular Capnocytophaga infection in an edentulous, immunocompetent host.
      ,
      • Iimori H
      • Suzuki T
      • Inoue T
      • et al.
      A case of contact-lens-related keratitis caused by fluoroquinolone- and tobramycin-resistant Capnocytophaga sputigena.
      This report is the first to describe a case of pediatric orbital cellulitis with SPA caused by C. sputigena.
      Orbital cellulitis can usually be managed medically with a combination of IV antibiotics and nasal sprays. Adjunctive treatment with systemic corticosteroids has recently been proven safe and effective. IV dexamethasone (0.3 mg/kg/d every 6 hours for 3 days) given on admission has been shown to decrease the length of hospital stay without significant adverse effects.
      • Chen L
      • Silverman N
      • Wu A
      • Shinder R
      Intravenous steroids with antibiotics on admission for children with orbital cellulitis.
      SPA formation is a known complication of orbital cellulitis. Current guidelines recommend the consideration of surgical intervention if any of the following criteria are met: age greater than 9 years, presence of frontal sinusitis, large or nonmedial SPA, suspicion of anaerobic infection, recurrence of the SPA after previous drainage, evidence of chronic sinusitis, acute optic nerve or retinal compromise, or infection of dental origin.
      • Garcia GH
      • Harris GJ.
      Criteria for nonsurgical management of subperiosteal abscess of the orbit: analysis of outcomes 1988-1998.
      Our 15-year-old patient presented with an orbital cellulitis most likely secondary to ipsilateral ethmoid and maxillary sinusitis. The infection was initially unresponsive to medical treatment, and orbital imaging revealed an atypically large, gas-containing inferomedial SPA—features strongly suggestive of an atypical bacterial infection—that justified surgical intervention. This case highlights the importance of multidisciplinary care to achieve an optimal clinical outcome.

      Acknowledgements

      The authors would like to acknowledge the patient presented in this study, who generously granted them permission to share his story and photographs with the academic community.

      Footnotes and Disclosure

      The authors have no proprietary or commercial interest in any materials discussed in this article.

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