pathogens Case Report Concurrent Pseudomonas Periorbital Necrotizing Fasciitis and Endophthalmitis: A Case Report and Literature Review Yu-Kuei Lee 1 and Chun-Chieh Lai 1,2,* 1 Department of Ophthalmology, National Cheng Kung University Hospital, College of Medicine, National Cheng Kung University, Tainan 704, Taiwan; [email protected] 2 Institute of Clinical Medicine, College of Medicine, National Cheng Kung University, Tainan 704, Taiwan * Correspondence: [email protected]; Tel.: +886-6-235-3535-5441 Abstract: (1) Background: Necrotizing fasciitis (NF) is an infection involving the superficial fascia and subcutaneous tissue. Endophthalmitis is an infection within the ocular ball. Herein we report a rare case of concurrent periorbital NF and endophthalmitis, caused by Pseudomonas aeruginosa (PA). We also conducted a literature review related to periorbital PA skin and soft-tissue infections. (2) Case presentation: A 62-year-old male had left upper eyelid swelling and redness; orbital cellulitis was diagnosed. During eyelid debridement, NF with the involvement of the upper Müller’s muscle and levator muscle was noted. The infection soon progressed to scleral ulcers and endophthalmitis. The eye developed phthisis bulbi, despite treatment with intravitreal antibiotics. (3) Conclusions: Immunocompromised individuals are more likely than immunocompetent hosts to be infected by PA. Although periorbital NF is uncommon due to the rich blood supply in the area, the possibility of PA infection should be considered in concurrent periorbital soft-tissue infection and endophthalmitis. Citation: Lee, Y.-K.; Lai, C.-C. Keywords: Pseudomonas aeruginosa; necrotizing fasciitis; endophthalmitis Concurrent Pseudomonas Periorbital Necrotizing Fasciitis and Endophthalmitis: A Case Report and Literature Review. Pathogens 2021, 10, 1. Introduction 854. https://doi.org/10.3390/ Necrotizing fasciitis (NF) is a severe and rapidly progressive infection of the superficial pathogens10070854 fascia and subcutaneous tissue. Endophthalmitis is a type of severe ocular inflammation that occurs due to infection within the ocular ball. Pseudomonas aeruginosa (PA), a Gram- Academic Editors: Natalia Kirienko and Carolyn Cannon negative rod and opportunistic pathogen, is commonly found naturally in waste, water and soil [1]. Although PA is regarded as a common nosocomial pathogen, it also exists Received: 30 April 2021 in the community environment and infects immunocompromised individuals [2,3]. PA Accepted: 5 July 2021 infects the human body by attaching to soft tissue via its fimbriae and producing several Published: 7 July 2021 surface-associated adherence factors or adhesins that promote attachment to epithelial cells and contribute to virulence [4]. PA then proliferates and releases enzymes such as elastase, Publisher’s Note: MDPI stays neutral alkaline protease and exotoxin A, which leads to tissue necrosis [5]. Therefore, PA may with regard to jurisdictional claims in cause periorbital NF and other related complications, especially in immunocompromised published maps and institutional affil- patients, although previous reports showed that NF is generally caused by group A strep- iations. tococci [6]. Accordingly, we report a case of concurrent periorbital NF and endophthalmitis caused by a community-acquired PA infection. As previous reports are relatively rare, we also conducted a literature review related to periorbital skin and soft-tissue infections caused by PA. Copyright: © 2021 by the authors. 2. Results Licensee MDPI, Basel, Switzerland. This article is an open access article A 62-year-old man presented with periorbital pain and erythematous eyelid swelling distributed under the terms and for two days (Figure1A). The patient’s ocular history included bilateral vitreous hem- conditions of the Creative Commons orrhage and tractional retinal detachment status after pars plana vitrectomy four years Attribution (CC BY) license (https:// prior. He had hypertension, hepatitis C and chronic kidney disease status three months creativecommons.org/licenses/by/ after kidney transplantation. After kidney transplantation, he regularly took steroids and 4.0/). Pathogens 2021, 10, 854. https://doi.org/10.3390/pathogens10070854 https://www.mdpi.com/journal/pathogens Pathogens 2021, 10, 854 2 of 6 Pathogens 2021, 10, x FOR PEER REVIEWimmunosuppressants. He had no other systemic conditions, such as diabetes mellitus,3 of 7 dyslipidemia, or a drug allergy. FigureFigure 1.1. (A) The The patient patient had had periorbital periorbital pain pain and and eyelid eyelid erythematous erythematous swelling swelling for for two two days. days. (B) (BWhitish) Whitish necrotic necrotic tissue tissue in the in theupper upper palpebral palpebral conjunctiva, conjunctiva, tarsus, tarsus, Müller’s Müller’s muscle muscle and levator and levator mus- cle was observed during surgery. (C) Slit lamp biomicroscopy revealed that the left eye injected the muscle was observed during surgery. (C) Slit lamp biomicroscopy revealed that the left eye injected conjunctiva and hypopyon 3 mm in height. (D) After four months of follow-up, he developed the conjunctiva and hypopyon 3 mm in height. (D) After four months of follow-up, he developed phthisis bulbi. phthisis bulbi. 3. Discussion The patient’s laboratory data revealed a white blood cell count of 1900/µL (normal range:NF 3400 is a− severe9500/ µinfectionL) with with an absolute a rapidly neutrophil progressive count infectious of 1041/ processµL (normal involving range: the 1300superficial−5600/ fasciaµL), C-reactive and subcutaneous protein level tissue. of Its 12.5 clinical mg/L course (normal is often range: rapidly <8.0 mg/L) progressive, and a fastingwith a bloodhigh morbidity sugar level and of mortality 99 mg/dL rate (normal and the range: infection 60−99 site mg/dL). is usually The located patient in had the regularlyextremities taken [7]. immunosuppressantsHowever, periorbital afterNF is kidney uncommon transplantation, due to the rich which blood may supply have led in the to leukopenia.area [6]. The The most blood common culture pathogens reports showed responsible no bacterial for periorbital growth. OnNF ocularbelong examination, to monomi- thecrobial best-corrected group A streptococci visual acuity [6], was whereas 20/30 causal in the microorganisms right eye and counting of NF elsewhere fingers atare one of- meterten polymicrobial, in the left eye. with The group left upper A streptococci eyelid was and extremely Enterobacteriaceae swollen, firm being and the tender most withcom- erythematousmon pathogens changes. [7]. In Theour conjunctivacase, instead was of injectedstreptococci, with chemosis.the microbial The anteriorcultures chamber,from the intraocularperiorbital lenstissue, and scleral fundus ulcers, were aqueous unremarkable. humor and vitreous all showed a PA infection. Moreover,The patient the patient was afebrile did not and have then signs administered of toxic shock 400 mg syndrome intravenous (e.g., teicoplanin fever, skin every rash otherand low day blood and 2000 pressure), mg ceftriaxone which may once cause a day. the Thehigh steroids mortality and rate immunosuppressants in patients with NF wereinfected gradually by group reduced. A streptococci However, [6]. the Notably, eyelid the swelling patient with we tendernessreported is persisted.the first case Dif- of ferentialconcurrent diagnoses, periorbital including NF and preseptalendophthalmitis cellulitis caused and orbital by PA. cellulitis, were considered. Initially,Endophthalmitis periorbital cellulitis is a type was of highlysevere ocular suspected inflammation based on thethat computed occurs due tomography to infections imageswithin showingthe ocular soft ball tissue and swellingcan be classified around the as periorbitalexogenous region.or endogenous During the based left eyelidon the incisiontransmission and drainage, route of however,the infection we noted[8]. Ex largeogenous amounts endophthalmitis of whitish necrotic occurs tissuewhen inthe the in- upperfecting palpebral organisms conjunctiva, directly enter tarsus, the Müller’seye, while muscle endogenous and levator endophthalmitis muscle (Figure results1B). Thefrom pathologyinfectious ofagent these hematogenous necrotic tissues seeding showed ofsuppurative the eye. In the inflammation literature, septic composed metastatic of dense en- neutrophilicdophthalmitis infiltration may develop with focalfrom necrosis.distant NF The of definite a lower diagnosis extremity of infected periorbital by Klebsiella NF was madepneumoniae based on[9]. the Severe surgical infections, and pathological such as liver findings. abscesses A microbial and bacteremia, culture from can the also necrotic cause endogenous endophthalmitis concurrent with orbital cellulitis or NF of the leg [10–13]. In contrast, based on the clinical manifestations and culture findings in our patient, we sur- mised that the endophthalmitis was exogenous, and was caused by direct invasion of the Pathogens 2021, 10, 854 3 of 6 tissue was performed during the operation and revealed a PA infection. The intravenous antibiotics were switched to 2000 mg ceftazidime once a day. An anterior orbitotomy with debridement was performed again with removal of necrotic tissue. He was discharged five days after the surgery against the physician’s advice. After the loss of follow-up for three weeks, he returned again due to left-eye tenderness. The left-eye best-corrected visual acuity decreased to only light perception. Left eyelid wound dehiscence