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ARCHIVOS DE LA SOCIEDAD ESPAÑOLA DE OFTALMOLOGÍA www.elsevier.es/oftalmologia
Short communication
Bilateral enucleation due to multi-bacterial fulminant endogenous panophthalmitis夽 T.A. Chaparro Tapias a , C.M. Rangel Gualdron a , H.A. Rodriguez a,b , ˜ c L.M. Rodriguez a , L. Flores de los Reyes c,∗ , J.C. Sánchez Espana a b c
Departamento de Cirugía Plástica Ocular, Oncológica y Órbita, Fundación Oftalmológica de Santander FOSCAL, Santander, Colombia Departamento de Oftalmología, Universidad Industrial de Santander, Bucaramanga, Santander, Colombia Departamento de Oftalmología, Hospital General de Granollers, Granollers, Barcelona, Spain
a r t i c l e
i n f o
a b s t r a c t
Article history:
The case is presented of a 62 year-old woman with a rapid, progressive bilateral decrease in
Received 24 July 2019
visual acuity and panuveitis with orbital cellulitis. She was also in poor general condition,
Accepted 10 October 2019
with emesis and fever. Septicaemia due to Klebsiella pneumoniae and bilateral endogenous
Available online 12 December 2019
panophthalmitis were diagnosed. The ocular infection quickly progressed to sclerokeratitis and bilateral perforation despite broad spectrum systemic antibiotic management, and
Keywords:
eventually the patient required bilateral enucleation. Microbiological cultures of the surgical
Panophthalmitis
pieces identified Klebsiella pneumoniae and Candida magnoliae.
Eye enucleation
To our knowledge, this is the third published case that required bilateral enucleation or
Bacteraemia
evisceration due to endogenous panophthalmitis, and the first case of endogenous ocular
Klebsiella pneumoniae
infection caused by Candida magnoliae.
Candida magnolia
˜ ˜ S.L.U. All rights de Oftalmolog´ıa. Published by Elsevier Espana, © 2019 Sociedad Espanola reserved.
Enucleación bilateral por panoftalmitis endógena polimicrobiana fulminante r e s u m e n Palabras clave:
˜ Mujer de 62 anos con disminución de la agudeza visual bilateral rápidamente progre-
Panoftalmitis
siva y panuveítis con celulitis orbitaria, asociado a mal estado general, emesis y fiebre.
Enucleación
Se diagnosticó septicemia por Klebsiella pneumoniae y panoftalmitis endógena bilateral. La
Bacteriemia
afectación ocular progresó rápidamente a escleroqueratitis y perforación en ambos ojos pese
Klebsiella pneumoniae
a recibir manejo antibiótico sistémico de amplio espectro. Finalmente, la paciente requirió
Candida magnoliae
enucleación bilateral. Los cultivos microbiológicos de las piezas quirúrgicas identificaron Klebsiella Pneumoniae y Candida Magnoliae.
夽 ˜ Please cite this article as: Chaparro Tapias TA, Rangel Gualdron CM, Rodriguez HA, Rodriguez LM, Flores de los Reyes L, Sánchez Espana JC. Enucleación bilateral por panoftalmitis endógena polimicrobiana fulminante. Arch Soc Esp Oftalmol. 2020;95:34–37. ∗ Corresponding author. E-mail address: fl
[email protected] (L. Flores de los Reyes). ˜ ˜ S.L.U. All rights reserved. 2173-5794/© 2019 Sociedad Espanola de Oftalmolog´ıa. Published by Elsevier Espana,
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De acuerdo con nuestro conocimiento, es el tercer caso publicado que haya requerido enucleación o evisceración bilateral por panoftalmitis endógena y el primer caso de infección ocular endógena causada por Candida Magnoliae. ˜ ˜ S.L.U. Todos de Oftalmolog´ıa. Publicado por Elsevier Espana, © 2019 Sociedad Espanola los derechos reservados.
Introduction Endogenous endophthalmitis refers to intraocular infectious compromise secondary to hematogenous dissemination from a different origin.1–7 In severe cases, it could extend to the orbital cavity and evolve to panophthalmitis.5,6 Treatment combines the use of intravitreal and periocular antibiotics and corticoids, in some cases associated to early therapeutic vitrectomy.1,7 Despite said treatments, a high percentage of cases evolve rapidly with poor visual prognosis. Frequently, endogenous endophthalmitis is associated to severe systemic infections that endanger the life of the patient, reaching mortality rates between 4 and 50 % according to different series.1–7 The case of a patient without known comorbidity is presented. The patient exhibited bilateral endogenous panophthalmitis in which, despite systemic antibiotic management, ocular and orbital infection progressed rapidly and evolved to ocular perforation requiring enucleation of both eyes.
Clinic case Female, 62, who consulted the Ophthalmology Dept. due to fast and progressive visual acuity reduction with 4 days evolution associated to fever, emesis and drowsiness. The patient did not refer remarkable ophthalmological or pathological history, traumatism or previous surgery. Visual acuity was perception of light in both eyes. Examination showed signs of anterior orbital cellulitis associated to chemosis, 2 mm hyperopia and marked vitritis (Fig. 1a). Due to the poor overall condition of the patient and to the bilateral ocular compromise, she was assessed by the Internal Medicine Dept. that found sepsis secondary to bacteremia by Klebsiella with liver loci. The ophthalmological diagnostic was endogenous bilateral panophthalmitis. The patient was admitted to the Intensive Care Unit for antibiotic treatment with 2 g of intravenous cefepime every 8 h as well as to complete the systemic study, which discarded parenteral consumption of drugs, human immunodeficiency virus infection and immunodepression for other reasons, meningitis, selfimmune diseases, diabetes and cardiac disease among others. Chest-abdominal tomography showed labor abscesses and multiple pulmonary lesions. Antibiotic treatment produced general improvement within 48 h but the ophthalmological condition worsened, with increased chemosis and orbital cellulitis, giving rise to
Fig. 1 – Hospitalization day 1, evidencing bilateral orbital cellulitis and hemorrhagic chemosis (a). The condition worsened 48 h later (b, c). Despite the change of antibiotic treatment, ocular compromise worsened to necrosis and bilateral scleral perforation (d–f).
extreme proptosis (Fig. 1b, c). It was decided to initiate management with broad range systemic antibiotics, meropenem and vancomycin. On the 5th day of hospitalization, the patient exhibited scleral necrosis with perforation and bilateral loss of intraocular content (Fig. 1d–f). Visual acuity worsened to no perception of light. Due to the persistence of the active infectious loci and ocular perforation, urgent surgery was decided for enucleation of both eyes without implant. The anatomopathological analysis of the surgical pieces reported the presence of necrotizing keratitis and scleritis, acute purulent iridocyclitis and purulent endophthalmitis (Fig. 2a–c). The microbiological study of both eyes reported the presence of Klebsiella pneumoniae and Candida magnoliae-type fungii (Fig. 2d, e).
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Fig. 2 – Photograph of surgical parts, deformed ocular globes covered by hemorrhagic inflammatory exudate (a). Pigmented uvea fragments, replaced by inflammatory exudate (b). Pale pink sclera interrupted and infiltrated by a fibrin-purulent exudate which also occupies the vitreous (c). Filamented variable gram bacillus (d, e); culture identified Klebsiella pneumoniae. Staining with calcofluor white evidencing the presence of yeasts (f); culture identified Candida magnoliae.
Discussion In endogenous panophthalmitis, microorganisms penetrate the eye from another infectious location through the hematogenous pathway. Their association to septicemia signifies a condition with very poor visual and in some cases ocular and systemic prognosis.1–7 The literature describes infections caused by gram-positive and gram-negative germs, including species of Streptococcus, Staphylococcus, Bacillus cereus, Propionibacterium acnes, Escherichia coli, Klebsiella, Neisseria and Pseudomonas.1–7 In contrast, only 2 cases were found in which the Candida magnoliae yeast generated disease in humans, although none with ocular compromise as in the present patient.8,9 In the present case, even though the life of the patient was saved, it was necessary to perform an extreme surgical procedure such as bilateral enucleation in order to achieve local control of the infection. Only 2 other cases were found in the literature describing this mutilation surgery as a necessity due to bilateral endogenous panophthalmitis, i.e., patient 22 in the series reported by Yang et al. in 20073, that required evisceration of one eye and enucleation of the other one, and NFK, a patient in the series recently reported by Chung et al.7 who required bilateral evisceration. Other cases of bilateral ocular perforation with autoevisceration have been reported, in which patients had rejected ocular extraction or were in premortem condition and were not candidates for surgery (Chee SP, Personal Communication, 2017). It has been proposed that the early use of intravitreal antibiotics and posterior pars plana vitrectomy or multiple intravitreal and periocular applications of antibiotics combined with steroids could reduce the possibility of requiring enucleation or evisceration.1,2,7 The use of systemic antibiotics also appears to be useful to reduce local infection and accordingly diminish the risk of enucleation.1 In the present case it was not possible to perform early surgery or more aggressive conservative treatment from onset due to the critical condition of the patient during several days
that posed a danger to her life. This could have been a factor in the final necessity of enucleation. In the absence of references and large studies recommending enucleation as opposed to evisceration or the other way round in patients with orbital or intraocular infection, the decision must be taken individually.10 In the present case, scleral perforation that indicated poor tissue condition and the persistence of the orbital infection justified the choice of enucleation in both eyes. In addition, the necessity of longer surgery time and higher risk in a patient in critical general condition influenced the decision of not placing an implant in the first surgery. The frequency of bilateral endogenous panophthalmitis is low. Even so, it is important to be familiar with said entity and obtain an in-depth knowledge of its management due to the poor prognosis it entails. Early establishment of intravitreal and periocular treatment, associated to vitrectomy in some cases, could improve results and diminish the necessity of performing enucleation or evisceration in these patients.
Conflict of interests No conflict of interests was declared by the authors.
references
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8. Lane J, Lee M, Stephens J. Tenosynovitis secondary to Candida magnoliae in an immunocompetent host: Candida magnoliae tenosynovitis. Internet J Infect Dis. 2000;1:1–5. 9. Cascio Lo G, Carbonare D, Maccacaro L, Caliari F, Ligozzi V, Cascio Lo R. First Case of Bloodstream Infection Due to Candida magnoliae in a Chinese Oncological Patient. J Clin Microbiol. 2007;45:3470–3. 10. Hui JI. Outcomes of orbital implants after evisceration and enucleation in patients with endopthalmitis. Curr Opin Ophthalmol. 2010;21:375–9.