Advertisement

Canalicular melt secondary to Pseudomonas aeruginosa infection in a pediatric patient

Published:October 18, 2018DOI:https://doi.org/10.1016/j.jcjo.2018.07.009
      Pseudomonas aeruginosa can cause ocular or periocular infections, such as keratitis, conjunctivitis, and dacryocystitis.
      • Lattman J
      • Massry GG
      • Hornblass A
      Pseudomonal eyelid necrosis: clinical characteristics and review of the literature.
      There have been reports of periocular and eyelid necrotizing fasciitis secondary to Pseudomonas infection and these cases are usually accompanied by systemic illness, malnutrition, alcoholism, or minor ocular trauma.
      • Comaish I
      • Thaller V
      • Newman P
      Necrosis of the lid due to Pseudomonas aeruginosa.
      Typically, bilateral ocular involvement and neutropenia is seen. This is the first documented case of unilateral, localized canalicular melting caused by Pseudomonas microbial infection.

      Case Report

      A 14-year-old girl presented with fever, swelling, redness, and pain over the right medial eyelid. She complained of epiphora, foreign body sensation, and mild blurred vision without a history of trauma to the right eye. On examination, she had a visual acuity of 20/20 bilaterally without limitations of extraocular movements. Eye examination was remarkable for congested conjunctiva and tenderness with palpation of the medial right eyelid with purulent discharge from the puncta noted.
      Unlabelled image
      Panel A. Right upper eyelid with probed canalicular disruption and associated purulent drainage and chemosis.
      Unlabelled image
      Panel C. Intraoperative silicone intubation of the upper and lower canaliculi of the right eye.
      Unlabelled image
      Panel D. Post-operative photo of reconstructed and healed right eyelid canalicular system.
      Fig 1
      Fig. 1A. right upper eyelid with probed canalicular disruption and associated purulent drainage and chemosis. B, right lower eyelid with probed canalicular disruption and associated purulent drainage and chemosis. C, intraoperative silicone intubation of the upper and lower canaliculi of the right eye. D, post-operative photo of reconstructed and healed right eyelid canalicular system.
      The patient was treated with cefepime and piperacillin and was taken to the operating room for debridement, repair, and silicone intubation of the upper and lower canaliculi (Fig. 1C). Three months after the operation, the silicone tubes were removed. The upper and lower canalicular structures were normal on exam (Fig. 1D) and the lacrimal drainage system was patent on syringe irrigation test at a 1-year follow-up.

      Discussion

      Eyelid or periocular necrosis due to infection has been described in several reports, most of which are secondary to trauma such as animal bites or avulsive lacerations.
      • Lattman J
      • Massry GG
      • Hornblass A
      Pseudomonal eyelid necrosis: clinical characteristics and review of the literature.
      • Comaish I
      • Thaller V
      • Newman P
      Necrosis of the lid due to Pseudomonas aeruginosa.
      • Kreger AS
      Pathogenesis of Pseudomonas aeruginosa ocular diseases.
      • Kronish JW
      • McLeish WM
      Eyelid necrosis and periorbital necrotizing fasciitis.
      • Lazzeri D
      • Lazzeri S
      • Figus M
      • et al.
      Periorbital necrotising fasciitis.
      • Rodriguez-Gonzalez F
      • Marrero-Saavedra D
      • Rutllan-Civit J
      • Cabrera-Vargas E
      • Martinez-Qintana E
      Ocular necrotizing fasciitis due to pseudomonas aeruginosa in a non-neutropenic patient.
      • Prendiville KJ
      • Bath PE
      Lateral cantholysis and eyelid necrosis secondary to pseudomonas aeruginosa.
      Risks increase in those with systemic diseases such as diabetes, alcoholism, or any immunocompromised condition.
      • Prendiville KJ
      • Bath PE
      Lateral cantholysis and eyelid necrosis secondary to pseudomonas aeruginosa.
      The most common etiology of periorbital necrotizing fasciitis is Streptococcus pyogenes or Staphylococcus aureus, but rarely Pseudomonas aeruginosa have been identified.
      • Lattman J
      • Massry GG
      • Hornblass A
      Pseudomonal eyelid necrosis: clinical characteristics and review of the literature.
      • Comaish I
      • Thaller V
      • Newman P
      Necrosis of the lid due to Pseudomonas aeruginosa.
      • Kreger AS
      Pathogenesis of Pseudomonas aeruginosa ocular diseases.
      • Kronish JW
      • McLeish WM
      Eyelid necrosis and periorbital necrotizing fasciitis.
      • Lazzeri D
      • Lazzeri S
      • Figus M
      • et al.
      Periorbital necrotising fasciitis.
      • Rodriguez-Gonzalez F
      • Marrero-Saavedra D
      • Rutllan-Civit J
      • Cabrera-Vargas E
      • Martinez-Qintana E
      Ocular necrotizing fasciitis due to pseudomonas aeruginosa in a non-neutropenic patient.
      • Prendiville KJ
      • Bath PE
      Lateral cantholysis and eyelid necrosis secondary to pseudomonas aeruginosa.
      Non-infectious lid destruction has been described from the bite of the brown recluse spider
      • Kronish JW
      • McLeish WM
      Eyelid necrosis and periorbital necrotizing fasciitis.
      • Zeligowski AA
      • Peled U
      • Wexier MR
      Eyelid necrosis after spider bites.
      or neoplastic processes like sebaceous gland carcinoma.
      • Kronish JW
      • McLeish WM
      Eyelid necrosis and periorbital necrotizing fasciitis.
      Tissue destruction such as necrotizing fasciitis is uncommon in the eyelids due to a rich blood supply; that being said, it can still be fatal.
      • Rodriguez-Gonzalez F
      • Marrero-Saavedra D
      • Rutllan-Civit J
      • Cabrera-Vargas E
      • Martinez-Qintana E
      Ocular necrotizing fasciitis due to pseudomonas aeruginosa in a non-neutropenic patient.
      Pseudomonas aeruginosa attaches to sialic acid receptors by attaching to the fimbriae; the bacteria then proliferates and releases enzymes such as elastase, alkaline protease, and exotoxin A, resulting in tissue necrosis.
      • Kreger AS
      Pathogenesis of Pseudomonas aeruginosa ocular diseases.
      In a study of canalicular laceration, 55% of cases were caused by direct penetrating injury. Injury to the lacrimal duct system can lead to scarring, stenosis, and epiphora if not repaired in a timely fashion. In traumatic disruption of the canaliculus, it is common to repair using silicone tubing as a lacrimal stent.
      • Jordan DR
      • Ziai S
      • Gilberg SM
      • Mawn LA
      Pathogenesis of canalicular laceration.
      • Reifler DM
      Management of canalicular laceration.
      In this case, the patient was neutropenic secondary to an SLE flare. Most cases of Pseudomonas-related eyelid necrosis have shown association with neutropenia secondary to systemic conditions such as septicemia, malnutrition, cancer, and other types of immunocompromised conditions. There has not been a documented case of such localized canalicular injury as seen in our patient.
      Vasculitis has been documented in up to 36% of SLE patients; of those patients, cutaneous manifestations (petechiae, palpable purpura, cutaneous infarction, superficial ulceration, etc.) were the most common and indicate small vessel involvement.
      • Kenneth TC
      • Balabanova M
      Vasculitis in systemic lupus erythematosus.
      The inflammation of these vessels can lead to ischemia of downstream tissue via antibody complex and complement aggregation and deposition. This ischemia combined with low complement levels predisposes the area to infection—in this case, likely predisposing the patient to the localized Pseudomonas infection and destruction of the pericanalicular structures. That being said, typically cutaneous presentations of vasculitis in SLE patients are much more widespread and not so localized.
      Early diagnosis of the patient's underlying condition was important in this case as resolution of neutropenia has been associated with improved clinical improvement. We were fortunate to isolate the Pseudomonas culture early, which may have prevented progression from tissue and muscle necrosis to true necrotizing fasciitis and also allowed for timely repair of the canalicular disruption. If left untreated, the patient may have had devastating, irreparable necrosis and destruction of the lacrimal drainage system.

      Conclusions

      Although it is rare, it is important to rule out tissue necrosis secondary to Pseudomonas in periocular infection. Early diagnosis and recognition of a systemic medical illness in the setting of eyelid necrosis caused by Pseudomonas aeruginosa is an important step to prevent possible progression to necrotizing fasciitis and key to a good clinical outcome after treatment and therapy. Thorough evaluation of the eyelid and examining for canalicular disruption helps with initiation of repair as quickly as possible to prevent scarring and stenosis of the lacrimal drainage system.

      Disclosure

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

      Appendix. Supplementary materials

      References

        • Lattman J
        • Massry GG
        • Hornblass A
        Pseudomonal eyelid necrosis: clinical characteristics and review of the literature.
        Ophthal Plast Reconstr Surg. 1998; 14: 290-294
        • Comaish I
        • Thaller V
        • Newman P
        Necrosis of the lid due to Pseudomonas aeruginosa.
        Eye. 2000; 14: 387-389
        • Kreger AS
        Pathogenesis of Pseudomonas aeruginosa ocular diseases.
        Rev Infect Dis. 1983; 5: S931-S935
        • Kronish JW
        • McLeish WM
        Eyelid necrosis and periorbital necrotizing fasciitis.
        Ophthalmology. 1991; 98: 92-98
        • Lazzeri D
        • Lazzeri S
        • Figus M
        • et al.
        Periorbital necrotising fasciitis.
        Br J Ophthalmol. 2010; 94: 1577-1585
        • Rodriguez-Gonzalez F
        • Marrero-Saavedra D
        • Rutllan-Civit J
        • Cabrera-Vargas E
        • Martinez-Qintana E
        Ocular necrotizing fasciitis due to pseudomonas aeruginosa in a non-neutropenic patient.
        Saudi J Ophthalmol. 2013; 27: 281-282
        • Prendiville KJ
        • Bath PE
        Lateral cantholysis and eyelid necrosis secondary to pseudomonas aeruginosa.
        Ann Ophthalmol. 1988; 20: 193-195
        • Zeligowski AA
        • Peled U
        • Wexier MR
        Eyelid necrosis after spider bites.
        Am J Ophthalmol. 1986; 101: 254-255
        • Jordan DR
        • Ziai S
        • Gilberg SM
        • Mawn LA
        Pathogenesis of canalicular laceration.
        Ophthal Plast Reconstr Surg. 2008; 24: 394-398
        • Reifler DM
        Management of canalicular laceration.
        Surv Ophthalmol. 1991; 36: 113-132
        • Kenneth TC
        • Balabanova M
        Vasculitis in systemic lupus erythematosus.
        Clin Dermatol. 2004; 22: 148-156