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A month prior to presentation, he had nodular lesion in lower part of left cornea and was undergoing medical management from a local ophthalmologist. After ocular and systemic examination, probable diagnosis of phlyctenular keratoconjunctivitis (PKC) was established. Since the perforation was large and not amenable to application of tissue adhesives, it was managed with a corneal patch graft. After investigations, we found that this was a case of latent tuberculosis infection. He was referred to physician and was started on appropriate anti-tubercular therapy. At the final follow-up, the perforation healed, and he regained a vision of 20/20. Even though perforation with tubercular PKC is a rare entity, this case highlights that timely diagnosis and multimodal intervention is the key to successful management." } { "@context": "http://schema.org", "@type": "BreadcrumbList", "itemListElement": [ { "@type": "ListItem", "position": "1", "item": { "@id": "https://f1000research.com/", "name": "Home" } }, { "@type": "ListItem", "position": "2", "item": { "@id": "https://f1000research.com/browse/articles", "name": "Browse" } }, { "@type": "ListItem", "position": "3", "item": { "@id": "https://f1000research.com/articles/13-10/v1", "name": "Case Report: Managing a case of Tubercular Phlycten presenting as..." } } ] } Home Browse Case Report: Managing a case of Tubercular Phlycten presenting as... ALL Metrics - Views Downloads Get PDF Get XML Cite How to cite this article Kaur A, Das S and Priyadarshini SR. Case Report: Managing a case of Tubercular Phlycten presenting as Sterile Corneal Perforation [version 1; peer review: awaiting peer review] . F1000Research 2024, 13 :10 ( https://doi.org/10.12688/f1000research.138371.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. Close Copy Citation Details Export Export Citation Sciwheel EndNote Ref. Manager Bibtex ProCite Sente EXPORT Select a format first Track Share ▬ ✚ Case Report Case Report: Managing a case of Tubercular Phlycten presenting as Sterile Corneal Perforation [version 1; peer review: awaiting peer review] Amanjot Kaur https://orcid.org/0000-0002-1259-7399 1 , Sujata Das 2 , Smruti Rekha Priyadarshini 2 Amanjot Kaur https://orcid.org/0000-0002-1259-7399 1 , Sujata Das 2 , Smruti Rekha Priyadarshini 2 PUBLISHED 03 Jan 2024 Author details Author details 1 Cornea And Anterior Segment Services, SSCI, LV Prasad Eye Institute Kode Venkatadri Chowdary Campus, Vijayawada, Andhra Pradesh, India 2 Cornea And Anterior Segment Services, SSCI, L V Prasad Eye Institute, MTC Campus, Bhubaneswar, Odisha, India Amanjot Kaur Roles: Data Curation, Formal Analysis, Visualization, Writing – Original Draft Preparation Sujata Das Roles: Funding Acquisition, Project Administration, Supervision, Writing – Review & Editing Smruti Rekha Priyadarshini Roles: Conceptualization, Data Curation, Funding Acquisition, Investigation, Methodology, Project Administration, Resources, Software, Supervision, Validation, Writing – Review & Editing OPEN PEER REVIEW REVIEWER STATUS AWAITING PEER REVIEW This article is included in the Eye Health gateway. Abstract A 22-year-old Indian male presented with a sterile perforation at 5 o’clock in the left eye, and a visual acuity of 20/80. A month prior to presentation, he had nodular lesion in lower part of left cornea and was undergoing medical management from a local ophthalmologist. After ocular and systemic examination, probable diagnosis of phlyctenular keratoconjunctivitis (PKC) was established. Since the perforation was large and not amenable to application of tissue adhesives, it was managed with a corneal patch graft. After investigations, we found that this was a case of latent tuberculosis infection. He was referred to physician and was started on appropriate anti-tubercular therapy. At the final follow-up, the perforation healed, and he regained a vision of 20/20. Even though perforation with tubercular PKC is a rare entity, this case highlights that timely diagnosis and multimodal intervention is the key to successful management. READ ALL READ LESS Keywords Phlyctenular keratoconjunctivitis, tuberculosis, perforation, corneal patch graft Corresponding Author(s) Smruti Rekha Priyadarshini ( [email protected] ) Close Corresponding author: Smruti Rekha Priyadarshini Competing interests: No competing interests were disclosed. Grant information: We received funding from the Hyderabad Eye Research Foundation. The sponsors had no role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. Copyright: © 2024 Kaur A et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. How to cite: Kaur A, Das S and Priyadarshini SR. Case Report: Managing a case of Tubercular Phlycten presenting as Sterile Corneal Perforation [version 1; peer review: awaiting peer review] . F1000Research 2024, 13 :10 ( https://doi.org/10.12688/f1000research.138371.1 ) First published: 03 Jan 2024, 13 :10 ( https://doi.org/10.12688/f1000research.138371.1 ) Latest published: 03 Jan 2024, 13 :10 ( https://doi.org/10.12688/f1000research.138371.1 ) Introduction Phlyctenular keratoconjunctivitis (PKC) is a cell-mediated allergic reaction of the conjunctiva or cornea. It might be caused by several antigens, including tubercular proteins and staphylococcal by-products. 1 – 3 Corneal phlyctens usually presents with a limbal nodule that ulcerates at the apex within 1-2 weeks. Phlyctens are acute but usually self-limiting and corneal phlyctens rarely perforate. 4 In the literature, only a few cases of perforation have been reported after non-tubercular PKC. 5 – 7 In this paper, we present a unique case of tubercular PKC, presenting with corneal perforation and a brief review of the literature. Case report A 22-year-old Indian male farmer presented with complaints of a whitish mass that grew gradually and painlessly in the left eye. He consulted locally, where he was diagnosed with a limbal nodule. There was no relevant family history. Previous medical report have described the limbal nodule to be 2×2 mm with a leash of blood vessels migrating towards the cornea from 5 to 6 o’clock position. He was started on topical prednisolone acetate 1% (4 times/day) and moxifloxacin 0.3% (8 times/day). After one month, he developed diminution of vision and presented to our clinic. He was worried as his vision has decreased suddenly. There was no history of ocular trauma. The right eye was functioning within normal limits. His uncorrected visual acuity (UCVA) was 20/20 and 20/80 in the right and left eyes, respectively. In the left eye, the conjunctiva was congested, and a 3×3 mm corneal perforation was observed at the 5 o’clock position, with iris tissue plugging the perforated site ( Figure 1a ). Both lids were healthy, with no signs of blepharitis. The fundus examination was also within normal limits. Figure 1. (a): A 3×3 mm corneal perforation on presentation. (b): Postoperative day one picture showing corneal patch graft. (c): Persistence of surrounding stromal edema due to DMD (marked with an arrow in AS-OCT). (d): Picture showing the well-integrated patch graft (marked with an arrow in AS-OCT) on the last follow-up visit. There was no infiltration or stromal cellularity around the perforated area, ruling out an infectious etiology. Peripheral ulcerative keratitis (PUK) with or without any autoimmune etiology was considered. However, the absence of an overhanging epithelium or stromal thinning in the adjacent area ruled out PUK. Considering the age, ethnic preponderance, and clinical picture, a provisional diagnosis of phlyctenular keratitis with perforation was suspected. The patient had no history of chronic cough, fever, weight loss, or night chills. He was advised investigations such as a total leukocyte count (TLC), erythrocyte sedimentation rate (ESR), and Mantoux intradermal tuberculin skin test (TST). In the meantime, he underwent a corneal patch graft to restore the anatomical integrity of the globe. The prolapsed iris tissue was abscised intraoperatively, and an unused anterior stromal cap (from the previous Descemet’s stripping keratoplasty procedure) was trephined and utilized. The 3.5 mm lamellar, circular, corneal cap was secured with six interrupted 10-0 nylon sutures. Postoperatively, the graft was in place, though mildly edematous, and the anterior chamber was well formed ( Figure 1b ). He was started on topical antibiotics (ofloxacin 0.3%, 4 times/day) and steroids (prednisolone acetate 1%, 8 times/day). In the meantime, the patient’s blood investigation showed elevated TLC (11.7×10 9 /L) and ESR (69/mm 3 in the 1 st h). TST was strongly positive, with 75 mm of induration read at 48 hours. The patient was also prescribed an interferon gamma release assay (IGRA, QuantiFERON Gold) for further confirmation. The IGRA was positive, and he was referred to a physician. The chest radiograph showed no active lesions, and a diagnosis of latent tubercular infection (LTBI) was established. The patient was started on a multi-drug anti-tubercular therapy (ATT) of oral Isoniazid (750 mg) and oral rifapentine (750 mg), once weekly for three months, and oral rifampicin (600 mg), once daily for four months for the LTBI. At the 1-week postoperative visit, the stromal edema persisted ( Figure 1c ), and Descemet’s membrane detachment (DMD) was observed in a small area adjacent to the graft, which was managed using air descemetopexy. The DMD could have resulted from the reforming of the anterior chamber postoperatively. Topical steroids (prednisolone acetate 1%, in weekly tapering doses), topical antibiotics (ofloxacin 0.3%, 4 times/day), and oral ATT were continued. Subsequently, the stromal edema subsided, and all the sutures were removed after six weeks. The graft-host junction was well apposed ( Figure 1d ), and the patient regained a UCVA of 20/25, improving to 20/20 with glasses at the final visit. The perforated site healed with scar formation, and the integrity of the globe was maintained. Since the perforation was at the perilimbal site, the patient’s visual acuity was not affected much. Discussion PKC is a cell-mediated allergic reaction to antigens, such as-tubercular proteins, staphylococcal by-products, parasites, or other microbes. 1 – 3 It is usually seen in the low socio-economic strata during the first two decades of life, with a few reports suggesting female pre-ponderance (though some reports depict no gender predilection). 8 – 10 Reports from some Asian countries show that TB accounts for approximately 77% of cases of PKC. PKC is usually seen in pulmonary and lymph node TB; however, LTBI has also been implicated as an etiology. 11 The patient in our case report was also a young Indian male from a low socio-economic background. He had no suggestive symptoms or contact with an active case of TB infection. TB is an endemic disease in the Indian subcontinent; therefore, investigations were done to rule out a tubercular infection. The clinical presentation depends on the location of the lesion and the underlying etiology. Corneal or TB-related phlyctenules are usually more symptomatic than conjunctival or staphylococcal-related phlyctenules. 4 The most common site for phlyctenules is inferiorly (4-8 o’clock area). The infection presents either as a conjunctival or corneal gelatinous nodular lesion. As the lesion progresses, it ulcerates and shows positive fluorescein staining at the apex. 4 , 12 Conjunctival phlyctens are transient nodules that rarely cause permanent scarring. In contrast, corneal phlyctenules migrate from the limbus to the cornea, leaving a leash of pannus behind. 13 In our case report, the documentation of the presence of a limbal nodule with a leash of blood vessels prior to presentation led to the suspicion of PKC. Moreover, most of the cornea was clear at presentation, ruling out an infectious cause or PUK as an etiology. No blepharitis or acne rosacea was observed in our case. In TST, a standard dose of 5 tuberculin units (0.1 mL) of purified protein derivative is injected intradermally and the results are read between 48-72 h. 14 Though it is a cheap and easy-to-perform test, the Bacille Calmette Guerin (BCG) vaccine is associated with a high risk of false positive results. 15 Therefore, in countries (like India) where BCG is a part of childhood immunization, skin induration of >15 mm is more suggestive of tuberculosis infection. 16 Moreover, IGRA increases the positive predictive value, as the BCG vaccine does not interfere with the test. 17 , 18 In our case, both TST and IGRA were strongly positive but symptoms of active TB or findings on the chest radiograph were negative. Therefore, he was started on an ATT regimen for LTBI, as per guidelines of the World Health Organization. 14 Corneal perforation is rarely reported in corneal phlyctens, as most cases heal by scar formation. Topical steroids are the first-line treatment for PKC, 3 with severe cases warranting topical immunomodulators. 19 Our patient presented with a 3×3 mm perforation, and simply applying cyanoacrylate glue would not have sufficed. Therefore, a corneal patch graft was required. 20 The advantage of corneal patch graft is rapid closure of perforation, but one needs tissue to be readily available. Apart from support from the eyebank, there could be chances of suture-related complication, such as hyphema, DMD (as seen in our case) and infections in worst case scenarios. 21 In literature, similar cases had been reported in non-tubercular PKC. The perforations had been treated with either cyanoacrylate glue, corneal patch graft, or left to heal with systemic tetracyclines or soft contact lenses based on the size of the perforation. 5 – 7 The location of the lesions is also important. Peripheral lesions close to the limbus usually heal well with scar formation. Even if a patch graft is performed, the visual acuity is mostly unaffected. Cases involving the visual axis are difficult to treat and may require an optimum-sized optical keratoplasty. In a previous case report, the corneal patch graft led to opacification in the visual axis, requiring penetrating keratoplasty later. 7 Table 1 shows the published case reports of perforations in PKC. Among the five reported cases of PKCs with perforations, none had TB as the etiology. 5 – 7 Table 1. Case reports of perforations in phlyctenular keratoconjunctivitis. Serial number Author Year Perforation/Total number of cases Etiology Management (n) 1. Ostler et al. 1975 1/1 Staphylococcal blepharitis Bandage contact lens 2. Culbertson et al. 1993 3/17 Ocular rosacea (1), Chlamydial infection (1), Unknown (1) Cyanoacrylate glue (1), Corneal patch graft (1), Tetracyclines (1) 3. Ahn et al . 2014 1/1 Staphylococcal blepharitis Corneal patch graft followed by penetrating keratoplasty * * The patient was initially managed with a patch graft; however, opacity remained in the visual axis. Subsequently, keratoplasty was done for visual rehabilitation. To the best of our knowledge, this is a unique case of tubercular PKC, presenting as a perforation. As perforations are rare in PKC, such cases can be overlooked, and other differentials, such as sterile melts in PUK, trauma, or infective perforations, may be considered. However, careful examination of the surrounding epithelium, stroma, and lids will help in reaching the correct diagnosis. In endemic countries, once the diagnosis of PKC is considered, the subject should be tested for TB. The risk of LTBI is approximately 33%. Therefore, these cases should be investigated for any active disease. Even if no active disease is present, timely ATT reduces the risk of reactivation. 22 Timely ATT is also important as the close differential is PUK. Supposedly systemic immunosuppression were started in this case (as the perforations are common in PUK than PKC and also ESR was raised), it would have wreaked havoc leading to reactivation of the latent TB infection. Therefore, in young patients from low socio-economic strata presenting with inferior conjunctival or corneal lesions, a diagnosis of PKC should always be kept in mind. In such patients, a thorough examination of the lids and laboratory analyses helps to establish the etiology. Once TB is established as an etiology, a multidisciplinary approach should be planned. An immediate referral to the nearest pulmonologist/physician can prevent systemic complications. Hence, appropriate evaluation and timely ocular and systemic intervention remains the key to successful management. This case report also emphasizes the role of an ophthalmologist in finding a serious systemic infection lurking underneath the mask of ocular pathologies. Consent Written informed consent for the publication of the case report and any associated images was obtained from the patient prior to submission. Author contributions AJK contributed to the study conceptualization, study investigation, methodology, manuscript editing, and revision. SD and SMR contributed to the original draft preparation, study methodology, and revisions. Author roles Kaur A: Data Curation, Methodology, Visualization, Writing – Original Draft Preparation. Das S and Priyadarshini SR: Conceptualization, Data Curation, Funding Acquisition, Investigation, Methodology, Project Administration, Supervision, Validation, Visualization, Writing –Review & Editing. Data availability Underlying data All data underlying the results are available as part of the article, and no additional source data are required. Acknowledgements None. References 1. Sorsby A: The aetiology of phlyctenular ophthalmia. Br. J. Ophthalmol. 1942; 26 : 159–179. PubMed Abstract | Publisher Full Text | Free Full Text 2. Thygeson P, Diaz-Bonnet V, Okumoto M: Phlyctenulosis. Attempts to produce an experimental model with BCG. Investig. Ophthalmol. 1962; 1 : 262–266. PubMed Abstract 3. Shrestha G, Gautam P, Sharma A: Phlyctenular keratoconjunctivitis among children in the tertiary eye hospital of Kathmandu, Nepal. Oman J. Ophthalmol. 2015; 8 (3): 147–150. PubMed Abstract | Publisher Full Text | Free Full Text 4. Ostler HB, Lanier JD: Phlyctenular keratoconjunctivitis with special reference to the staphylococcal type. Trans. Pac. Coast Otoophthalmol. Soc. Annu. Meet. 1974; 55 : 237–252. PubMed Abstract 5. Ostler H: Corneal Perforation in Nontuberculous (Staphylococcal) Phlyctenular Keratoconjunctivitis. Am. J. Ophthalmol. 1975; 79 : 446–448. PubMed Abstract | Publisher Full Text 6. Culbertson WW, Huang AJ, Mandelbaum SH, et al. : Effective treatment of phlyctenular keratoconjunctivitis with oral tetracycline. Ophthalmology. 1993; 100 : 1358–1366. PubMed Abstract | Publisher Full Text 7. Ahn Y, Lee J, Cho Y: Corneal Perforation in Phlyctenular Keratitis. J. Korean Ophthalmol. Soc. 2014; 55 : 298. Publisher Full Text 8. McArevey J: The social and medical problems of phlyctenular disease in Dublin. Ir. J. Med. Sci. 1944; 19 : 201–224. Publisher Full Text 9. Jones S, Weinstein J, Cumberland P, et al. : Visual Outcome and Corneal Changes in Children with Chronic Blepharokeratoconjunctivitis. Ophthalmology. 2007; 114 : 2271–2280. PubMed Abstract | Publisher Full Text 10. Rohatgi J, Dhaliwal U: Phlyctenular eye disease: a reappraisal. Jpn. J. Ophthalmol. 2000; 44 : 146–150. PubMed Abstract | Publisher Full Text 11. Ferdi A, Kopsachilis N, Parmar D: Phlyctenulosis: a systemic diagnosis made or missed in the blink of an eye. Clin. Exp. Optom. 2017; 100 : 285–287. Publisher Full Text 12. Neiberg M, Sowka J: Phlyctenular keratoconjunctivitis in a patient with Staphylococcal blepharitis and ocular rosacea. J. Am. Optom. Assoc. 2008; 79 : 133–137. PubMed Abstract | Publisher Full Text 13. Nayak S, Acharjya B: Mantoux test and its interpretation. Indian Dermatol. Online J. 2012; 3 : 2–6. PubMed Abstract | Publisher Full Text | Free Full Text 14. Wang L: A meta-analysis of the effect of Bacille Calmette Guerin vaccination on tuberculin skin test measurements. Thorax. 2002; 57 : 804–809. PubMed Abstract | Publisher Full Text | Free Full Text 15. Seddon JA, Paton J, Nademi Z, et al. : The impact of BCG vaccination on tuberculin skin test responses in children is age dependent: evidence to be considered when screening children for tuberculosis infection. Thorax. 2016; 71 (10): 932–939. PubMed Abstract | Publisher Full Text | Free Full Text 16. Manuel O, Kumar D: QuantiFERON ® -TB Gold assay for the diagnosis of latent tuberculosis infection. Expert. Rev. Mol. Diagn. 2008; 8 : 247–256. Publisher Full Text 17. Lobue P, Menzies D: Treatment of latent tuberculosis infection: An update. Respirology. 2010; 15 : 603–622. Publisher Full Text 18. World Health Organization: Latent TB infection: updated and consolidated guidelines for programmatic management. Reference Source 19. Doan S, Gabison E, Gatinel D, et al. : Topical cyclosporine A in severe steroid-dependent childhood phlyctenular keratoconjunctivitis. Am J. Ophthalmol. 2006; 141 : 62–66.e2. PubMed Abstract | Publisher Full Text 20. Jhanji V, Young A, Mehta J, et al. : Management of Corneal Perforation. Surv. Ophthalmol. 2011; 56 : 522–538. Publisher Full Text 21. Deshmukh R, Stevenson LJ, Vajpayee R: Management of corneal perforations: An update. Indian J. Ophthalmol. 2020; 68 (1): 7–14. PubMed Abstract | Publisher Full Text | Free Full Text 22. Dye C, Scheele S, Dolin P, et al. : Consensus statement. 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Publisher Full Text Comments on this article Comments (0) Version 1 VERSION 1 PUBLISHED 03 Jan 2024 ADD YOUR COMMENT Comment Author details Author details 1 Cornea And Anterior Segment Services, SSCI, LV Prasad Eye Institute Kode Venkatadri Chowdary Campus, Vijayawada, Andhra Pradesh, India 2 Cornea And Anterior Segment Services, SSCI, L V Prasad Eye Institute, MTC Campus, Bhubaneswar, Odisha, India Amanjot Kaur Roles: Data Curation, Formal Analysis, Visualization, Writing – Original Draft Preparation Sujata Das Roles: Funding Acquisition, Project Administration, Supervision, Writing – Review & Editing Smruti Rekha Priyadarshini Roles: Conceptualization, Data Curation, Funding Acquisition, Investigation, Methodology, Project Administration, Resources, Software, Supervision, Validation, Writing – Review & Editing Competing interests No competing interests were disclosed. Grant information We received funding from the Hyderabad Eye Research Foundation. The sponsors had no role in the study design, data collection and analysis, decision to publish, or preparation of the manuscript. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. Article Versions (1) version 1 Published: 03 Jan 2024, 13:10 https://doi.org/10.12688/f1000research.138371.1 Copyright © 2024 Kaur A et al . This is an open access article distributed under the terms of the Creative Commons Attribution License , which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. Download Export To Sciwheel Bibtex EndNote ProCite Ref. Manager (RIS) Sente metrics Views Downloads F1000Research - - PubMed Central info_outline Data from PMC are received and updated monthly. - - Citations open_in_new 0 open_in_new 0 open_in_new SEE MORE DETAILS CITE how to cite this article Kaur A, Das S and Priyadarshini SR. Case Report: Managing a case of Tubercular Phlycten presenting as Sterile Corneal Perforation [version 1; peer review: awaiting peer review] . F1000Research 2024, 13 :10 ( https://doi.org/10.12688/f1000research.138371.1 ) NOTE: If applicable, it is important to ensure the information in square brackets after the title is included in all citations of this article. COPY CITATION DETAILS track receive updates on this article Track an article to receive email alerts on any updates to this article. TRACK THIS ARTICLE Share Open Peer Review Current Reviewer Status: AWAITING PEER REVIEW AWAITING PEER REVIEW ? Key to Reviewer Statuses VIEW HIDE Approved The paper is scientifically sound in its current form and only minor, if any, improvements are suggested Approved with reservations A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit. 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