|Year : 2023 | Volume
| Issue : 2 | Page : 375-376
Hypopyon uveitis as a presenting manifestation of biopsy-proven sarcoidosis – A case report
Kalpana Babu1, Preethi Hegde1, Govind S Mittal2
1 Department of Uvea and Ocular Inflammation, Prabha Eye Clinic and Research Centre and Vittala International Institute of Ophthalmology, Bengaluru, Karnataka, India
2 Therapeía Skin, Hair and ENT Centre, Bengaluru, Karnataka, India
|Date of Submission||17-Jun-2022|
|Date of Acceptance||11-Jan-2023|
|Date of Web Publication||28-Apr-2023|
504, 40th Cross, Jayanagar 8th Block, Bengaluru - 560 070, Karnataka
Source of Support: None, Conflict of Interest: None
A 68-year-old lady was referred for evaluation of bilateral hypopyon uveitis by her ophthalmologist. She had a history of bilateral cataract surgery 8 months ago and had received intravitreal dexamethasone implants for recurrent bouts of anterior uveitis with cystoid macular edema (CME) in both eyes 4 months ago. Systemic history was significant for diabetes mellitus and recurrent skin lesions. Aqueous tap was negative for infection on culture and polymerase chain reaction (PCR). Skin biopsy showed non-necrotizing granulomas, negative for Mycobacterium tuberculosis and fungi, suggestive of sarcoidosis. She received oral steroids and methotrexate, which resulted in resolution of ocular and skin lesions. This case highlights a rare presentation of hypopyon in ocular sarcoidosis.
Keywords: Anterior uveitis, hypopyon, non-necrotizing granulomas, ocular sarcoidosis, skin biopsy
|How to cite this article:|
Babu K, Hegde P, Mittal GS. Hypopyon uveitis as a presenting manifestation of biopsy-proven sarcoidosis – A case report. Indian J Ophthalmol Case Rep 2023;3:375-6
|How to cite this URL:|
Babu K, Hegde P, Mittal GS. Hypopyon uveitis as a presenting manifestation of biopsy-proven sarcoidosis – A case report. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 6];3:375-6. Available from: https://www.ijoreports.in/text.asp?2023/3/2/375/374897
Non-granulomatous and granulomatous anterior uveitis are recognized presentations of ocular sarcoidosis. Generally, hypopyon is not associated with ocular sarcoidosis.,, Herein, we report an unusual presentation of hypopyon uveitis as an initial manifestation of sarcoidosis.
| Case Report|| |
A 68-year-old lady was referred to us with hypopyon uveitis (right eye [OD]) by her ophthalmologist. At the time of examination, she was using topical prednisolone acetate 1% eye drops in the left eye for a documented hypopyon 10 days ago. She had a history of bilateral cataract surgery 8 months ago and had received intravitreal dexamethasone implants in both eyes for recurrent bouts of anterior uveitis with cystoid macular edema (CME) 4 months ago. Systemic history was significant for diabetes mellitus and recurrent skin lesions for a duration of 2 years. Based on a recommendation of her physician, she was using steroid-based skin creams for the skin lesions on and off.
On examination, her best corrected visual acuity was 6/12 in both eyes (OU). Slit-lamp examination showed a hypopyon in OD [Figure 1] and an anterior chamber reaction (2+) in the left eye (OS) by the standardized uveitis nomenclature (SUN) grading. Pseudophakia and few cells in the anterior hyaloid face were seen (OU). Fundus examination did not reveal any abnormality. Fluorescein angiography and indocyanine green angiography did not reveal any choroidal granulomas. Optical coherence tomography (OCT) showed an epiretinal membrane (OU). Systemic examination was significant for erythematous papulomacular lesions, both discrete and confluent, on the back and upper and lower limbs [Figure 2]a.
|Figure 2: External photograph showing erythematous papulomacular lesions in the upper limbs (a). Microphotograph of skin biopsy showing non necrotizing granulomas (b), lymphocytes, epithelioid cells, and giant cells (c) (yellow arrows)|
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Laboratory investigations showed an increase in erythrocyte sedimentation rate (ESR; 74 mm/h), random blood sugar (231 mg%), slight increase in total white blood cell (WBC) counts (13,400 cells/mm3, range: 4000–10,000 cells/mm3), and negative Mantoux, QuantiFERON TB gold test, venereal disease research laboratory (VDRL), Treponema pallidum hemagglutination (TPHA) tests, antinuclear antibody (immunofluorescence), and HLA-B27 (polymerase chain reaction [PCR]). Serum angiotensin-converting enzyme was in the normal range (13 U/L, range: 12–66 U/L). Liver function, renal function, serum calcium, and high-resolution computed tomography (HRCT) thorax were normal. An aqueous tap from OD ruled out infections on PCR and culture. Cytology showed a few lymphocytes.
Skin biopsy showed non-necrotizing granulomas with lymphocytes, macrophages, and giant cells [Figure 2]b and [Figure 2]c, negative for acid-fast bacilli (AFB) and fungi. She was started on oral steroids (0.5 mg/kg) and methotrexate 15 mg/week for a diagnosis of sarcoidosis. At 6 months, her skin and ocular inflammation had resolved well [Figure 3]. She continues to be on oral methotrexate 20 mg/week.
|Figure 3: Composite photographs showing resolution of hypopyon in the right eye (a) and resolving skin inflammation with pigmentary scars (b) at 3 months follow-up|
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| Discussion|| |
Hypopyon is layering of WBCs in the anterior chamber and signifies severe anterior segment intraocular inflammation. Hypopyon is observed in 0.86%–4.6% of cases of anterior uveitis.,, Generally, it is associated with HLA-B27 positivity, spondyloarthropathies, Bechet's disease, endophthalmitis, and drugs such as rifabutin.,, Very rarely, it has been reported in juvenile idiopathic arthritis, inflammatory bowel disease, rheumatoid arthritis, and tuberculosis.,, Our case was interesting because of the atypical presentation of hypopyon in sarcoidosis. The diagnosis of sarcoidosis was made on the basis of skin biopsy findings of non-necrotizing granulomas, which were negative for Mycobacterium tuberculosis and fungi. Sarcoidosis presenting as hypopyon uveitis and, even so, being bilateral and asymmetric, is unusual. In the SITE (Systemic Immunosuppressive Therapy For Eye Diseases Cohort Study) cohort study, sarcoidosis was a risk factor in only two cases; however' both the cases did not present with a hypopyon. Intraocular surgeries result in the breakdown of blood aqueous barrier and can cause hypopyon in systemic diseases like spondyloarthropathies and Bechet's disease. It is a speculation that a surgical insult in the form of cataract surgery in our patient may be a predisposing factor for the development of hypopyon.
| Conclusion|| |
We report a rare and unusual presentation of hypopyon uveitis in a patient with biopsy-proven sarcoidosis.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3]