sign in sign up
<<
Home , Necrotizing fasciitis

Yaisawang et al. Journal of Ophthalmic Inflammation and (2018) 8:5 Journal of Ophthalmic https://doi.org/10.1186/s12348-018-0147-6 Inflammation and Infection

ORIGINAL RESEARCH Open Access Ocular involvement in : a 23-year retrospective review Sasi Yaisawang1* , Somkiat Asawaphureekorn1, Ploenchan Chetchotisakd2,3, Surasakdi Wongratanacheewin3,4 and Peerapat Pakdee5

Abstract Background: Ocular involvement in melioidosis is rare and has devastating outcomes. Although there have been few reports on the condition, Khon Kaen, a city in northeast Thailand, has been called the “capital of melioidosis” due to the high prevalence of the condition in the region. We retrospectively reviewed all admitted cases of melioidosis with ocular involvement from the two largest hospitals in Khon Kaen. We reviewed cases from Srinagarind Hospital (a university hospital) of patients admitted between 1993 and 2016 and from Khon Kaen Hospital (a provincial hospital) of patients who presented from 2012 to 2016. Results: We identified 16 cases of ocular involvement. Eight of these cases were proven from positive culture, and the remaining eight were implied from high melioidosis titer. The prevalence was estimated as being from 0.49 to 1.02%. Most patients had underlying diseases (14, 88%), of which mellitus was the most prevalent (12, 75%). Nine cases (56%) were part of disseminated septicemia. Patients suffered from blindness in 11 (73%) of the 15 cases in which visual acuity was recorded. Orbital was the most common manifestation (7, 44%) followed by endophthalmitis (4, 25%). Interestingly, all patients with necrotizing (100%) developed septic as a consequence. In most of the cases, patients underwent surgery (13, 81%) including incision and drainage, , and pars plana vitrectomy. Despite appropriate management, the visual outcomes were disappointing (9, 64%). Conclusion: To summarize, ocular melioidosis is a highly destructive disease. Early detection and prompt surgical management may reduce morbidity and mortality from . Keywords: Melioidosis, Burkholderia pseudomallei, , Orbital cellulitis, Endophthalmitis

Background In northeast Thailand, there are around 2000 culture- Melioidosis is caused by a gram-negative, motile, positive melioidosis cases per year [2]. Khon Kaen, one non-spore forming facultative anaerobic bacillus of the largest cities in northeast Thailand, has been known as Burkholderia pseudomallei. The organism is called “the capital of melioidosis” due to the high preva- found in soil and surface water and is widely distrib- lence of the disease in the region. Ocular involvement in uted in Southeast Asia, especially in northeast these cases has not been investigated. The primary Thailand and northern Australia [1]. objective of this study was to estimate the prevalence Melioidosis presents with broad spectrums of clinical and investigate ocular manifestations of melioidosis in presentations and organ involvement. However, there Khon Kaen. Management and visual outcomes in these are few case reports of ocular involvement in melioid- patients were also reviewed. osis, and most of these are single-case report or small case series. Results We identified 16 cases of ocular involvement, 13 out of the 1270 melioidosis cases admitted to Srinagarind Hos- * Correspondence: [email protected]; [email protected] 1Department of Ophthalmology, Faculty of Medicine, Khon Kaen University, pital (prevalence 1.02%; 95% confidence interval from Khon Kaen, Thailand Full list of author information is available at the end of the article

© The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. Yaisawang et al. Journal of Ophthalmic Inflammation and Infection (2018) 8:5 Page 2 of 9

0.58 to 1.76%) and three out of the 607 admitted cases Discussion at Khon Kaen Hospital (prevalence 0.49%; 95% confi- Toourknowledge,thisisthefirstandlargestcaseseriesof dence interval from 0.10 to 1.51%). Overall, the esti- ocular involvement in melioidosis. A comprehensive litera- mated prevalence of ocular involvement in cases of ture review revealed only 14 cases from 12 reports [3–14], melioidosis was from 0.49 to 1.02% (Table 1). including 7 cases of orbital cellulitis (50%), 3 cases of en- Of those 16 cases, there were 8 with positive cul- dophthalmitis (21%), 3 cases of corneal ulcer (21%), and 1 tures. In the remaining eight cases, melioidosis was case of acute dacryocystitis (7%). Most of the reports were implied from the high titer for melioidosis in the single-case reports, and the largest one had only three cases. bloodstream. Clinical descriptions of all cases are In Thailand, especially in the northeast, there has been summarized in Table 2. an increase in the reported cases of melioidosis. This is Baseline characteristics of the patients were compar- likely due to increasing awareness of the condition and able to general melioidosis patients. The male to female increased sensitivity of the technology used to detect the ratio was 3 to 1 with a median age of 50.5 years old (39– organism. The mortality rate in these areas is around 70). The most common occupation was farmer (nine 40%. It is the third highest cause of mortality after ac- cases, 56%). Most patients had underlying diseases (14 quired immune deficiency syndrome and tuberculosis cases, 88%), of which diabetes mellitus was the most [2]. Although ocular involvement in melioidosis is rare, common (12 cases, 75%). Ocular involvement was part the effects on patients’ vision are devastating. Most pa- of dissemination in nine cases (56%), which were classi- tients with this condition ended up becoming legally fied as disseminated septicemic melioidosis. blind. In our series of 16 cases, there were only 5 (36%) The majority of ocular melioidosis patients (10 in which patients had improved vision after treatment. cases, 63%) presented with eye symptoms. Interest- We suspect that the number of ocular melioidosis cases ingly, the other six cases initially presented with might be underestimated. Most of the melioidosis patients or a headache. Out of the 15 cases for which there admitted to the hospital had disseminated septicemic were records of visual acuity, 11 (73%) presented with melioidosis and were treated for life-threatening symp- blindness. The ocular manifestations of melioidosis toms. Mild ocular symptoms might be easily overlooked, were classified as orbital cellulitis (seven cases, 44%), and ophthalmologists were not consulted in all cases. preseptal cellulitis (two cases, 13%), endophthalmitis The prevalence of ocular melioidosis in Srinagarind (four cases, 25%), panophthalmitis (two cases, 13%), Hospital (1.02%) was about twice that in Khon Kaen and panuveitis (one case, 6%). Hospital (0.49%). The discrepancy might be due to the In most cases, the definitive management was surgery differences between the two hospitals. Srinagarind Hos- (13 cases, 81%) including incision and drainage, debride- pital is the largest university hospital in northeast ment (eight cases, 62%), pars plana vitrectomy (three Thailand, and many severe cases of systemic melioidosis cases, 23%), and enucleation (two, 15%). There were only are referred to Srinagarind Hospital. Since more organs three cases (19%) in which the patients were able to be are affected in severe disseminated melioidosis, ocular treated without surgery. involvement is more likely in these cases. Despite adequate surgical intervention, the visual out- We suspect that the recent prevalence of ocular comes of ocular melioidosis were disappointing. Out of melioidosis in Srinagarind Hospital might be much higher the 14 cases for which there were records of final visual than what we have found. From 2007 to April 2016, there acuity, nine (64%) patients ended up legally blind. Three were 264 cases of melioidosis at Srinagarind Hospital, of of these patients (20%) presented with no light percep- which 13 had ocular involvement. According to this find- tion at the beginning, two had to be enucleated, two ing, the prevalence during this time interval was as high (14%) were stable, and two (14%) had progressive loss of as 4.9% (95% confidence interval from 2.82 to 8.32%). vision. Patients had improved vision after treatment in This study led to some interesting findings. Ten patients only five cases (36%). (63%) presented with eye symptoms, which later resulted in

Table 1 Data collection and prevalence (95%CI) calculation Tertiary hospital Srinagarind University Hospital Khon Kaen Provincial Hospital Total from all data Total from 2012 to 2016 Date January 1993 to April 2016 January 2012 to April 2016 January 1993 to April 2016 January 2012 to April 2016 Length 23 years and 4 months 4 years and 4 months 23 years and 4 months 4 years and 4 months Total melioidosis (cases) 1270 607 1877 859 Total ocular involvement (cases) 13 3 16 8 Prevalence 1.02% (0.58, 1.76%) 0.49% (0.10, 0.51%) 0.85% (0.51, 1.39%) 0.93% (0.44, 1.86%) (95% CI) Table 2 Clinical descriptions of all cases Yaisawang Year Age Sex Occupation Symptom Laterality Initial VA Ocular Ocular Risk factor Type of Primary organ Associated Investigations Treatments Outcomes positive diagnosis melioidosis symptoms finding ta.Junlo ptamcIfamto n Infection and Inflammation Ophthalmic of Journal al. et 2007 70 F None Progressive OD No LP Complete Orbital cellulitis History of Disseminated Eye Acute Hemoculture: FESS, I&D, IV VA no LP, painful ptosis, mark eye sphenoidal B. pseudomellei, ceftazidime limit EOM proptosis eyelid scratching sinusitis, LP: eosinophilic then oral 90% at with fever swelling, IOP with dirty meningitis, meningitis, MRI bactrim, lateral gaze 10 days, 21/13, hand septic arthritis, orbit: right tarsorhaphy OD, other S/P IV marked melioidosis orbital cellulitis EOM are cloxacillin chemosis, septicemia with extraconal full, normal at provincial clear cornea, latero- anterior hospital no C/F, superior aspect segment, (onset = positive pale disc, 10 days) RAPD, EOM attenuated 0% all vessel (No direction, LP at initial) pale disc with choroidal fold 2007 64 M Farmer Progressive OS CF 2 ft Lid swelling, Orbital cellulitis DM Disseminated Eye Pansinusitis, Hemoculture: I&D, FESS, IV VA 6/24, VA proptosis proptosis, without subcutaneous B. pseudomellei, ceftazidime with pinhole 10 days chemosis, DR, CKD abscess at culture: then oral 6/12, less PTA, S/P IV clear cornea, inferolateral of B. pseudomellei. bactrim, chemosis, ceftazidime, no C/F, the eye CT orbit: topical less proptosis, IV positive pansinusitis normal (2018)8:5 at provincial RAPD, EOM with severe anterior hospital 10% all orbital cellulitis segment, direction EOM limit at downgaze (onset = (improve) 10 days) 2008 61 M Farmer Progressive OS No LP Generalized Panophthalmitis DM Localized Eye Hemoculture: PPV, ECCE, VA no LP, painful visual bedewing without no growth, topical conjunctival loss 2 weeks cornea, DR, CKD melioid titer , less chemosis, hypopyon 1:5122, CT orbit: topical AC deep 2-mm, swelling of ceftazidime, with shallow AC, periorbital oral bactrim plasmoid and C/F 3+/2+, tissue hyphema, no positive record about RRAPD, EOM posterior 50% all segment direction, B (onset = scan: (No LP at 2 weeks) generalized initial) vitreous opacity, intra-op findings: dense vitreous abscess,

subretinal 9 of 3 Page abscess rupture to vitreous Table 2 Clinical descriptions of all cases (Continued) Yaisawang Year Age Sex Occupation Symptom Laterality Initial VA Ocular Ocular Risk factor Type of Primary organ Associated Investigations Treatments Outcomes positive diagnosis melioidosis symptoms finding ta.Junlo ptamcIfamto n Infection and Inflammation Ophthalmic of Journal al. et 2008 51 F Teacher Fever with OS HM, good PJ Corneal Endogenous DM Disseminated Hematogenous Pulmonary Hemoculture: PPV with VA HM poor dyspnea bedewing, endopthalmitis without DR edema no growth, silicone oil, PJ, AC deep 12 days PTA, C/F 4+/4+, melioid titer ECCE, IV with left eye positive 1:5122, MRI ceftazidime plasmoid, inflammation RRAPD, intra- orbit: preseptal then oral attach retina was found op findings: cellulitis bactrim, during attenuated topical admission vessels, vancomycin, subretinal topical (onset = NA) gliosis, ceftazidime (stable) shallow RD 2009 39 F Farmer Eye pain with OD 6/24 Marked Preseptal DM Multifocal Eye Pneumonia, Pus culture: I&D upper VA 6/9, fever 2 weeks eyelid cellulitis without DR subcutaneous B. pseudomellei, eyelid, I&D normal swelling and abscess at melioid titer right thigh, anterior and erythema, right thigh 1/640, oral bactrim posterior fluctuation, hemoculture: segment no discharge, NG, CT orbit: (onset = clear cornea, preseptal (improve) 2 weeks) no C/F, cellulitis negative RAPD, normal posterior (2018)8:5 segment 2011 46 M Labor Fever with OS CF 2 ft Conjunctival Endogenous DM Multifocal Hematogenous Liver , Hemoculture: IV ceftazidime VA 3/60, VA constitutional chemosis, endophthalmitis without DR splenic abscess no growth, then oral with pinhole symptoms corneal melioid titer bactrim 4/60, 2 weeks then stromal edema, 1:5122, CT contracted visual loss 3 hypopyon, abdomen: hypopyon, days hyphema, multiple liver vitreous C/F 4+/2+, abscesses, opacity retinal splenic grade 1 infiltration abscess (onset = 3 days) (improve) 2011 43 F Farmer Painful proptosis OS Not done Necrotizing Orbital cellulitis, First dx DM Disseminated Eye Pansinusitis, Hemoculture: Debridement Good 8 days PTA, S/P due to fasciitis at necrotizing without DR melioidosis B. pseudomellei, of necrotic wound, less IV antibiotic at alteration of left upper fasciitis septic shock pus culture: wound, IV swelling, no provincial consciousness eyelid size B. pseudomellei, ceftazidime record hospital then 1 × 8 cm, CT orbit: orbital then oral about VA alteration of purulent abscess at the bactrim consciousness discharge, superomedial 1 day ciliary wall of orbit, injection, medial rectus (onset = 8 days) clear cornea, muscle, lateral (NA) no C/F, clear rectus muscle vitreous 2011 46 M Labor Painless visual OS LP, poor PJ IOP 32, Endogenous DM Multifocal Eye, liver Liver abscess Hemoculture: PPV with Painful red loss 1 month bedewing endopthalmitis without DR NG, melioid silicone oil, eye 1 week 9 of 4 Page PTA then cornea, titer 1:640 oral bactrim after painful hypopyon discharge, proptosis 2 with VA no LP, days plasmoid in IOP 40, Table 2 Clinical descriptions of all cases (Continued) Yaisawang Year Age Sex Occupation Symptom Laterality Initial VA Ocular Ocular Risk factor Type of Primary organ Associated Investigations Treatments Outcomes positive diagnosis melioidosis symptoms finding ta.Junlo ptamcIfamto n Infection and Inflammation Ophthalmic of Journal al. et AC, negative shallow AC, RAPD, intra- iris bombe op finding: end up with subretinal enucleation, abscess intra-op finding: flank pus in the vitreous cavity (onset = (enucleated) 1month) 2012 63 M Farmer Painful OD 20/200 Marked eyelid Panuveitis, MDS, Disseminated Eye Spondylodiscitis, Hemoculture: IV ceftazidime, VA 20/200, proptosis swelling, no preseptal leukemia epidural and no growth, 1% peripheral 2 weeks discharge, cellulitis paravertebral melioid titer prednisolone synechiae conjunctival abscess 1: 5120 acetate eye 360 degrees, injection, drop RE qid vitreous keratic opacity precipitates grade 1 at the cornea, (onset = 2 weeks) peripheral (stable) synechiae

360 degrees, (2018)8:5 C/F 4+/2+, vitreous opacity grade 4 2012 54 M Farmer Fever with left OS No LP Marked eyelid Orbital cellulitis DM Disseminated Maxillary sinus Maxillary Hemoculture: I&D, orbital VA no LP, side headache swelling, without DR sinusitis, B. pseudomellei, decompression, less swelling 1 week PTA erythema and melioidosis pus culture: B. IV ceftazidime periorbital then left facial tender, septicemia pseudomellei, then oral area, edema 5 days copious pus CT orbit: bactrim conjunctival PTA then and discharge, maxillary chemosis, painful marked sinusitis normal proptosis 3 chemosis, anterior days clear cornea, segment, no C/F, limit EOM positive all direction, RRAPD, B fundus: disc scan: vitreous swelling, opacity, intra- flame shape op finding: hemorrhage pus 1 ml in (onset = the vitreous (No LP at 1 week) cavity, flame initial) shape hemorrhage, disc swelling, venous

congestion, 9 of 5 Page drusen 2012 65 M House Fever with OD 6/6 Upper eyelid Preseptal DM Disseminated Hematogenous – Hemoculture: VA 6/6, no keeper chill 3 days swelling, cellulitis without DR no growth, lid swelling, Table 2 Clinical descriptions of all cases (Continued) Yaisawang Year Age Sex Occupation Symptom Laterality Initial VA Ocular Ocular Risk factor Type of Primary organ Associated Investigations Treatments Outcomes positive diagnosis melioidosis symptoms finding ta.Junlo ptamcIfamto n Infection and Inflammation Ophthalmic of Journal al. et PTA then erythema melioid titer IV ceftazidime mild alteration of and tender, 1:640 then oral erythema, consciousness conjunctival azithromycin normal 1 day, then chemosis, anterior and right upper clear cornea, posterior eyelid swelling no C/F, segment at the negative emergency RAPD, department normal posterior (onset = segment, full (improve) < 1 day) EOM 2013 57 M Thai Pain at the left OS 20/200 Proptosis, Orbital cellulitis DM Localized Temporal Temporal Pus culture: I&D temporal VA 6/9, no massager temporal area chemosis, without DR space space abscess, B. pseudomellei, space abscess, sign of 3 week PTA clear cornea, abscess subperiosteal hemoculture: lateral and inflammation, then painful no C/F, abscess NG, CT orbit: left medial residual proptosis 3 negative panophthalmitis orbitotomy, ptosis, days, S/P IV RAPD, EOM with I&D orbital normal antibiotic at 10-20% all subperiosteal abscess, IV anterior primary care direction, abscess ceftazidime segment, no hospital, S/P fundus: then oral record about I&D temporal macular bactrim posterior space abscess striae, mild segment (2018)8:5 at provincial pale disc hospital (onset = 3 weeks) (improve) 2014 42 M Farmer Right eye OD HM at Multiple keratic Endogenous CKD, Multifocal Eye Splenic abscess Gram stain Enucleation, Good contact with provincial precipitates at endophthalmitis chronic from pus: IV ceftazidime enucleation wood particle hospital then the cornea, , gram-negative then oral wound 10 days PTA no LP C/F 4+/4+, wood rod safety bactrim then drop of positive RAPD, particle pin, pus breast milk vitreous contact, culture: no into the eye opacity grade breast milk growth, 4 days PTA 4, B scan: instillation hemoculture: then acute loculated no growth, visual loss vitreous haze, melioid titer 2 days, S/P membrane- 1:5122, IVT vancomycin, like lesion ultrasound ceftazidime attach to disc, abdomen: at provincial moderate to splenic hospital high spike, abscess intra-op (onset = finding: yellow (enucleated) 10 days) pus with blood clot 2014 45 M Officer Proptosis 4 OS CF Marked eyelid Orbital cellulitis DM Multifocal Sinus Abscess at Pus culture I&D, IV Less days PTA, S/P swelling, without DR right leg from the eye: ceftazidime swelling, B. pseudomellei FESS, orbital fluctuation, , then oral less 9 of 6 Page decompression chemosis, pus culture bactrim chemosis, at private limit EOM at from the right normal hospital upper and leg: B. anterior lateral gaze, pseudomellei, segment, Table 2 Clinical descriptions of all cases (Continued) Yaisawang Year Age Sex Occupation Symptom Laterality Initial VA Ocular Ocular Risk factor Type of Primary organ Associated Investigations Treatments Outcomes positive diagnosis melioidosis symptoms finding ta.Junlo ptamcIfamto n Infection and Inflammation Ophthalmic of Journal al. et clear cornea, hemoculture: EOM no C/F, no growth, improve, negative RAPD, ultrasound no record normal abdomen: no of VA and posterior liver or splenic posterior segment, abscess segment intra-op (onset = 4 days) finding: (NA) loculated abscess at left upper eyelid 5ml 2015 50 M Farmer Low-grade OD HM Proptosis, Panopthalmitis – Disseminated Hematogenous Liver abscess, Hemoculture: PPV, IV No LP, fever 2 weeks marked ethmoid no growth, ceftazidime normal PTA, right eye chemosis, sinusitis melioid titer globe pain 9 days AC deep 1:5122, contour, no PTA, painful with C/F vitreous record proptosis with 4+/3+, culture: no about visual loss 7 days positive RAPD, growth anterior and PTA, S/P IV peripheral posterior ceftazidime, IV synechiae, segment metronidazole vitreous at provincial opacity (2018)8:5 hospital, grade 4, EOM progressive minimal limit proptosis in this all direction, admission B scan: vitreous (onset = 9 days) opacity, (worse) subretinal abscess, intra- op finding: yellow pus 0.2 ml 2015 45 M Farmer Painful proptosis OD 1/60 at Marked eyelid Orbital cellulitis, DM Disseminated Eye Sinusitis, septic Hemoculture: I&D, FESS, VA no LP, with fever primary care and periorbital Necrotizing without DR, arthritis, splenic B. pseudomellei skin chemosis, 2 weeks, S/P IV hospital then area swelling, fasciitis psoriasis, abscess, septic x II, pus culture debridement, normal antibiotic at LP vesicle at chronic shock from the eye: IV ceftazidime anterior primary care medial alcoholism B. pseudomellei, segment, hospital canthus, pus culture RAPD marked from the right positive, no bloody knee: B. record of chemosis, pseudomellei posterior clear cornea, segment no C/F, (onset = positive RAPD, (worse) 2 weeks) EOM 0% all direction, necrotic skin at forehead 9 of 7 Page 2×3cm Yaisawang et al. Journal of Ophthalmic Inflammation and Infection (2018) 8:5 Page 8 of 9

systemic spreading. On the other hand, there were six pa- Conclusions tients (38%) whose first symptoms were not eye symptoms; In summary, ocular involvement in melioidosis was four patients (25%) presented with fever and two (13%) pre- rare, but the outcomes were devastating. The most sented with a headache. In most cases, diabetes mellitus common ocular involvements were orbital cellulitis was the underlying disease (12 cases, 75%), but none of the and endophthalmitis. The morbidity in these cases patients in those cases had diabetic retinopathy. was high, so it is critical to employ a high index of Interestingly, we found that most cases of ocular meli- suspicion. Ocular melioidosis should be considered oidosis were classified as disseminated septicemic meli- when the ocular infection does not respond to con- oidosis (nine cases, 56%) which means that there was a ventional antibiotic therapy, especially in hyperen- bloodstream infection. This is unlike other gram-positive demic regions for melioidosis. Early consultation with organisms, which usually cause orbital cellulitis and an ophthalmologist and prompt surgical intervention commonly result in a negative hemoculture. The explan- may significantly improve the final visual outcomes, ation for this finding may be attributable to the nature as well as mortality rates. of Bulkholderia pseudomallei infection, which generally presents with bloodstream infection. Methods In our study, orbital cellulitis was the most common We retrospectively reviewed all admitted cases of meli- manifestation (seven cases, 44%). Usually, orbital cellu- oidosis with ocular involvement from two tertiary hospi- litis is caused by gram-positive organisms and can be tals in Khon Kaen using electronic databases. The first is cured only by intravenous , unlike orbital Srinagarind Hospital, which is a university hospital. We cellulitis caused by melioidosis. All of these patients searched the hospital’s electronic database for cases of ended up undergoing surgical intervention (100%). The this condition from January 1993 to April 2016 (23 years abscess-forming activity of Burkholderia pseudomallei and 4 months). The second is Khon Kaen Hospital, may be the reason why intravenous antibiotics alone did which is a provincial hospital. We searched the hospital’s not work to treat the condition. electronic database for cases that presented between Moreover, there were two cases (29%) of orbital cellu- January 2012 and April 2016 (4 years and 4 months). litis that progressed to , which is un- The data were retrieved using the ICD10 code for meli- common in other types of bacterial orbital cellulitis. oidosis (all A24 codes) and all diseases of the eye and This is similar to the results of a previous case report by adnexa (code H00 to H59). Saonanon P [13]. Unfortunately, all of our patients This manuscript adheres to the guidelines and princi- (100%) with necrotizing fasciitis subsequently developed ples laid out in the Declaration of Helsinki. Institutional septic shock. Early suspicion and prompt surgical de- review board (IRB) approval was obtained from the bridement may improve mortality in these patients. Khon Kaen University and Khon Kaen Hospital, We also found that even if systemic ceftazidime was Thailand. The clinical trial was registered in Thai Clin- used, the occurrence of endogenous endophthalmitis ical Trials Registry (study ID: TCTR20160818004). caused by melioidosis was not preventable, as stated in a We only included cases in which there were posi- previous report [10]. Most of the cases diagnosed as en- tive cultures for melioidosis or high blood titer ac- dophthalmitis and panophthalmitis required surgical cording to indirect hemagglutination (IHA). The intervention (five out of six cases, 83%), including pars cutoff point for positive antibody titers has been de- plana vitrectomy (three out of five cases, 60%) and enu- termined to be 1:160 in endemic areas [15]. Irrelevant cleation (two out of five cases, 40%). ocular diagnoses, such as cataracts, glaucoma, diabetic Two cases (50%) of endophthalmitis were enucleated. retinopathy, or other underlying eye diseases, were The first case, from 2011, had a delayed presentation. The excluded. The prevalence and 95% confidence inter- patient had experienced loss of vision for 1 month prior vals (95% CI) were calculated using the modified to admission, which was the longest onset in any of the Wald method. Other results were summarized as pro- cases. In the second case, from 2014, the patient exhibited portions and percentages. two risk factors for the condition, including wood particle Abbreviations contact and breast milk instillation into the eye, as a result AC: Anterior chamber; B. pseudomellei: Bulkholderia pseudomallei; C/F: Cell/ of local traditional treatment practices. flare; CF: Counting fingers; CKD: Chronic kidney disease; DM: Diabetes There were three cases that were cured without any mellitus; DR: Diabetic retinopathy; ECCE: Extracapsular cataract extraction; EOM: Extraocular movement; F: Female; FESS: Functional endoscopic sinus surgical intervention. In one case, this was due to the surgery; HM: Hand motion; I&D: Incision and drainage; IOP: Intraocular patient seeking early treatment for endogenous endoph- pressure; LP: Light perception; LP: Lumbar puncture; M: Male; thalmitis. The other two patients had diagnoses that did MDS: Myelodysplastic syndrome; MRI: Magnetic resonance imaging; NA: Not available; OD: Right eye; OS: Left eye; PJ: Light projection; PPV: Pars plana not require an operation (namely, panuveitis and presep- vitrectomy; RAPD: Relative afferent pupillary defect; RD: Retinal detachment; tal cellulitis). RRAPD: Reverse relative afferent pupillary defect; VA: Visual acuity Yaisawang et al. Journal of Ophthalmic Inflammation and Infection (2018) 8:5 Page 9 of 9

Acknowledgements 7. Srimuang S et al (1996) Immunobiological diagnosis of tropical ocular We would like to thank our clinical colleagues at the Srinagarind hospital diseases: Toxocara, Pythium insidiosum, Pseudomonas (Burkholderia) and Khon Kaen Hospital for their expertise with regard to the detection of pseudomallei, Mycobacterium chelonei and Toxoplasma gondii. Int J Tissue ocular involvement, diagnosis, and management of the patients in this React 18(1):23–25 study. We would also like to thank our coders for the complete diagnosis 8. Abdul Rani MF, Samad Cheung H, Mohd. Shah A, Mahmood T (2002) records that lead to the discoveries described in this paper. Melioidosis presenting as orbital and parotid abscesses with intracranial extension. Int Med J Malaysia 1(2).www.e-imj.com Funding 9. Yang IH, Lee JJ, Liu JW et al (2006) Melioidosis with endophthalmitis. Arch This work was supported by the Faculty of Medicine, Khon Kaen University. Ophthalmol 124:1501–1502 10. Chen K, Sun M, Hou C, Sun C, Chen T (2007) Burkholderia pseudomallei – Authors’ contributions endophthalmitis. J Clin Microbiol 45(12):4073 4074 SY carried out the ophthalmology studies, participated in the research 11. Shawarinin J, Bakiah S, Shatriah I (2009) Successfully treated rare – design, participated in the data acquisition at the university hospital, presentation of orbital melioidosis. Int J Ophthalmol 2:90 92 participated in the data interpretation, and drafted the manuscript. SA 12. Tirakunwichcha S, Vaivanijkul J (2009) Melioidosis presenting as orbital apex – carried out the ophthalmology practices, participated in the research syndrome. Asian J Ophthalmol 11:40 42 design, participated in the statistical analysis, and helped to draft the 13. Saonanon P, Tirakunwichcha S, Chierakul W (2013) Case report of orbital manuscript. PC carried out the infectious practices, provided expertise cellulitis and necrotizing fasciitis from melioidosis. Ophthal Plast Reconstr – regarding melioidosis, participated in the research design, helped Surg 29(3):e81 e84 facilitate the coordination between two hospitals in the study, and 14. Kogilavaani J, Shatriah I, Regunath K, Helmy A (2014) Bilateral orbital drafted the manuscript. SW carried out the microbiological studies, abscesses with subdural empyema and cavernous sinus due to – provided expertise with regard to melioidosis, participated in the data melioidosis in a child. Asian Pac J Trop Dis 4:S851 S853 acquisition and coordination between departments, and helped to draft 15. Naigowit P, Maneeboonyoung W, Wongroonsub P et al (1992) the manuscript. PP carried out the ophthalmology practices at the Serosurveillance for Pseudomonas pseudomallei infection in Thailand. Jpn J – provincial hospital and participated in the data acquisition at the Med Sci Biol 45:215 230 provincial hospital. All authors read and approved the final manuscript.

Ethics approval and consent to participate The manuscript adheres to the guidelines and principles by the Declaration of Helsinki. Institutional review board (IRB) approval was obtained from Khon Kaen University, Thailand, numbered HE581497 and Khon Kaen Hospital, Thailand, numbered KE59045. The clinical trial was registered in Thai Clinical Trials Registry study ID: TCTR20160818004.

Consent for publication Not applicable

Competing interests The authors declare that they have no competing interests.

Publisher’sNote Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Author details 1Department of Ophthalmology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand. 2Department of Medicine, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand. 3Melioidosis Research Center, Khon Kaen University, Khon Kaen, Thailand. 4Department of Microbiology, Faculty of Medicine, Khon Kaen University, Khon Kaen, Thailand. 5Department of Ophthalmology, Khon Kaen Hospital, Khon Kaen, Thailand.

Received: 5 October 2017 Accepted: 6 March 2018

References 1. Wiersinga WJ, Currie BJ, Peacock SJ (2012) Melioidosis. N Engl J Med 367: 1035–1044 2. Limmathurotsakul D, Wongratanacheewin S, Teerawattanasook N et al (2010) Increasing incidence of human melioidosis in northeast Thailand. Am J Trop Med Hyg 82:1113–1117 3. Yospaiboon Y, Sangveejit J (1987) Orbital cellulitis due to Pseudomonas pseudomallei. Thai J Ophthalmol 1:51–54 4. Suwanwatana C (1990) Acute dacrocystitis due to Pseudomonas pseudomallei. Srinakarin Hosp Med J 5:56–58 5. Siripanthong S, Teerapantuwat S, Prugsanusak W et al (1991) Corneal ulcer caused by Pseudomonas pseudomallei: report of three cases. Rev Infect Dis 13:335–337 6. Wong PK, Ng PH (1996) Melioidosis presenting with orbital cellulitis. Singap Med J 37:220–221