|Year : 2018 | Volume
| Issue : 2 | Page : 108-110
Bilateral endogenous endophthalmitis in disseminated histoplasmosis secondary to immunosuppression: A rare case report
Surpriya Hawaibam1, Dipankar Das2, Ronel Soibam3, Harsha Bhattacharjee3, Saurabh Deshmukh4, Richa Shrivastava1, Krati Gupta4
1 Department of Community, Comprehensive and Oculoplasty Services, Guwahati, Assam, India
2 Department of Ocular Pathology, Uveitis and Neuro-Ophthalmology Services, Guwahati, Assam, India
3 Vitreo-Retina Services, Sri Sankaradeva Nethralaya, Guwahati, Assam, India
4 Department of Ophthalmology, Sri Sankaradeva Nethralaya, Guwahati, Assam, India
|Date of Web Publication||6-Aug-2018|
Dr. Saurabh Deshmukh
Sri Sankaradeva Nethralaya, 96, Basistha Road, Beltola, Guwahati - 781 028, Assam
Source of Support: None, Conflict of Interest: None
We report the clinical course of a 47-year-old diabetic and hypertensive male who presented to us with diminution of vision in both eyes (OU) for 4 months. He was diagnosed with granulomatosis with polyangiitis and was treated with cyclophosphamide and steroids elsewhere. Before this, he was on steroids and methotrexate for probable sarcoidosis based on cavitary lesions in the lungs. During tapering of the steroids and so in relatively immunosuppressed status, he developed fever and maculopapulo-nodular skin lesions. Skin lesion biopsy confirmed Histoplasma capsulatum and he was started on systemic antifungals. Later, he developed bilateral endogenous endophthalmitis and was managed with intravitreal medications followed by vitrectomy with lensectomy OU. The patient regained vision and is maintaining the same until 1 year of follow-up. Thus, in spite of its rare occurrence, H. capsulatum should be considered as a cause of endogenous endophthalmitis in an immunosuppressed host with systemic histoplasmosis.
Keywords: Endophthalmitis, granulomatosis with polyangiitis, histoplasmosis, immunosuppression, Wegener's granulomatosis
|How to cite this article:|
Hawaibam S, Das D, Soibam R, Bhattacharjee H, Deshmukh S, Shrivastava R, Gupta K. Bilateral endogenous endophthalmitis in disseminated histoplasmosis secondary to immunosuppression: A rare case report. TNOA J Ophthalmic Sci Res 2018;56:108-10
|How to cite this URL:|
Hawaibam S, Das D, Soibam R, Bhattacharjee H, Deshmukh S, Shrivastava R, Gupta K. Bilateral endogenous endophthalmitis in disseminated histoplasmosis secondary to immunosuppression: A rare case report. TNOA J Ophthalmic Sci Res [serial online] 2018 [cited 2019 Jul 20];56:108-10. Available from: http://www.tnoajosr.com/text.asp?2018/56/2/108/238498
| Introduction|| |
Histoplasma capsulatum is an unencapsulated dimorphic fungus and the infection caused by it is called histoplasmosis. It was first described by Darling in 1906, and hence called Darling's disease. It is endemic in the United States and various regions of Africa and Asia. The fungus has two variants, namely, H. capsulatum var. capsulatum and H. capsulatum var. duboisii. It is a saprophytic fungus commonly isolated from the soil contaminated with bird or bat droppings. Systemically, it causes two different types of infection on the basis of the immune status of the host. In the immunocompetent individuals, inhalation of this contaminated soil may lead to the development of mild flu-like respiratory symptoms. Lungs may be affected leading to the development of calcified pulmonary nodules secondary to the localized granulomatous reaction., In the immunocompromised individuals, it leads to disseminated infection leading to the formation of granulomas in the liver, spleen, lymph nodes, and adrenals, manifesting as a malignant systemic disease with fever, generalized lymphadenopathy, hepatosplenomegaly, bone marrow suppression, and adrenal suppression.,,,, In the eyes, it causes two distinct varieties of infections. The common variety is the one in which there is a formation of granulomas in choroid, later leading to the formation of punched-out chorioretinal scars also known as the “histo-spots.” This variety is called presumed ocular histoplasmosis syndrome. The second variety is the one in which there is endogenous intraocular infection secondary to disseminated histoplasmosis.,
A detailed literature search using PubMed revealed that only seven cases of endogenous endophthalmitis from H. capsulatum are reported in the literature.,,,,,, Here we present a rare case of a 47-year-old patient, post immunosuppressive therapy for Granulomatosis with polyangiitis (Wegener's granulomatosis), with maculopapulonodular lesion over the face, neck and the trunk with ocular involvement.
| Case Report|| |
A 47-year-old male, resident of Assam, known diabetic and hypertensive, presented to us in October 2014 with the chief complaint of diminution of vision in both eyes (OU) for 4 months. On careful history taking, it was found that he first consulted a physician 6 months back in May 2014 for the complaints of fever, weight loss, and worsening of diabetes and was started on empirical antitubercular therapy (ATT) for cavitary lesions in lungs seen on chest X-ray for 6 months. Three weeks later, he developed an oral ulcer and gastrointestinal symptoms. On the basis of skin biopsy, a probable diagnosis of sarcoidosis was made and so he was started on steroids and methotrexate along with ATT. One month later, in June 2014, the patient then consulted elsewhere and considering the possibility of GPA (Wegener's granulomatosis), he was advised antineutrophil cytoplasmic antibody test and anti-proteinase 3 test. Both the tests came out to be positive and the diagnosis of GPA was confirmed and he was continued on immunosuppressive therapy; this time, he was treated with intravenous cyclophosphamide and methylprednisolone followed by oral prednisolone till October 2014. During tapering of the steroids in October 2014, he developed fever; lesions in the oral cavity; maculopapulo-nodular lesions over the face, neck, and trunk; and worsening of diabetes [Figure 1]a. Biopsy of these skin lesions done elsewhere confirmed the presence of H. capsulatum and was treated with intravenous liposomal amphotericin B (200 mg once daily for 30 days) followed by oral itraconazole (400 mg twice daily for 1 year). After taking antifungal treatment, the patient's symptoms improved, but he developed decreased vision in OU which on evaluation elsewhere showed bilateral uveitis and was prescribed topical steroids and cycloplegics and referred to a tertiary eye care center elsewhere where he was administered one dose of intravitreal injection in OU (ceftazidime 2.25 mg and voriconazole 100 μg each in 0.1 ml).
|Figure 1: (a) Clinical photograph of the patient showing maculopapulo-nodular lesions over the face. (b) Posttreatment clinical photograph showing resolution of facial lesions with eventual scarring|
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He came to us for further management. On examination, his visual acuity in OU was perception of light positive with accurate projection of rays in all the four quadrants. Slit-lamp examination showed fibrinous exudates, 360° posterior synechiae, complicated cataract, and small nodules all over the iris surface in OU [Figure 2]a. Intraocular pressure (IOP) was found to be 4 mmHg and 3 mmHg in the right eye (OD) and left eye (OS), respectively. Ultrasonography B-scan of the OU revealed low-reflective membrane echoes in vitreous cavity and increased retinochoroidal thickness [Figure 3]. The patient was systemically stable with blood sugar levels within normal limits.
|Figure 2: (a) Slit-lamp photograph of the right eye showing fibrinous exudates, 360° posterior synechiae, complicated cataract, and small nodules all over the iris surface. (b) Slit-lamp photograph postvitrectomy and lensectomy treatment showing resolution of exudates and clearing of visual axis|
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|Figure 3: (a) Ultrasonography B-scan of the right eye showing low-reflective intragel membranous echoes in the vitreous cavity with retinochoroidal thickness of 2.1 mm. (b) Ultrasonography B-scan of the left eye showing low-reflective intragel membranous echoes in the vitreous cavity with retinochoroidal thickness of 3 mm.|
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The patient was diagnosed to have bilateral endogenous endophthalmitis. Considering the deteriorating ocular condition of the patient and need for prompt treatment, a provisional diagnosis of H. capsulatum endophthalmitis, based on the skin biopsy report in an immunosuppressed state, was made. Accordingly, the patient was posted for intravitreal injection of voriconazole and ceftazidime and later to plan for lensectomy + vitrectomy in OU. Accordingly, the patient was given intravitreal injections in OU (ceftazidime 2.25 mg and voriconazole 100 μg each in 0.1 ml) in October 2014. An aqueous sample was simultaneously collected, but the sample was found to be inadequate for microbiological examination. A rapidly deteriorating ocular condition of the patient disallowed further resampling. Thus, to salvage the eyes and to prevent further delay in treatment, a provisional diagnosis of Histoplasma endophthalmitis was made based on biopsy-proven systemic histoplasmosis. The patient then underwent lensectomy + vitrectomy + silicone oil implantation and intravitreal voriconazole without intraocular lens implantation in OU elsewhere.
The patient was followed up monthly and the vision gradually improved to 6/18 OD and 6/60 OS with the resolution of facial lesions with minimal scarring in May 2015 [Figure 1]b.
In June 2015, the patient's best-corrected visual acuity was 6/18 OD and 6/36 OS, with the resolution of exudates and endothelial deposits and the clearing of visual axis [Figure 2]b. Due to continued instillation of steroid, there was a rise in IOP to 34 mmHg OD and 24 mmHg OS, which was treated with antiglaucoma medication.
In August 2015, emulsification of the silicone oil was noted in OU and the patient underwent silicone oil removal in OU. Subsequently, the patient developed hypotony and the vision decreased to 6/18 OD and counting fingers at 2 m OS with IOP of 6 mmHg OD and 7 mmHg OS. His vision was maintained till 1 year of follow-up.
| Discussion|| |
H. capsulatum is a dimorphic fungus since it grows in mycelial form at room temperature in the soil or in Sabouraud's agar and, at body temperature, it grows like yeast.
It can be diagnosed microscopically and through serological testing. On microscopy, at room temperature, it grows as septate hyphae with round- or pear-shaped conidia and, at body temperature, it grows like yeast. Serological tests include complement fixation, immunodiffusion, and mycelial antigen identification.
The severity of infection caused by H. capsulatum depends on the basis of the immune status of the patient. Our patient was a known diabetic and, in addition, was taking immunosuppressive drugs for a prolonged period of time. Drug-induced immunosuppression probably led to the opportunistic infection by H. capsulatum.
To our knowledge, only seven cases of endogenous endophthalmitis by H. capsulatum have been reported in the literature.,,,,,, On the basis of immune status, of the seven cases reported, five were immunocompetent,,,,, one was an HIV positive, and, in one case, immunologic status was unknown. In one case, infection developed post cataract extraction. Of all the seven cases, six cases had poor visual outcome,,,, but one case showed improvement.
Due to its slow rate of growth, the treatment of intraocular histoplasmosis is difficult. We treated our patient aggressively with multiple intravitreal injections of voriconazole and ceftazidime followed by pars plana vitrectomy with lensectomy.
Although a rare entity, H. capsulatum should be considered as a possible etiology of endophthalmitis in an immunosuppressed host. Thus, in this case report, the authors want to emphasize that the systemic condition of the patient should be taken into account while formulating the provisional diagnosis. This case is of endogenous endophthalmitis since there is no history of trauma or any surgery, and the only systemic cause that could be pointed out is histoplasmosis secondary to immunosuppression.
We would like to thank Sri Kanchi Sankara Health and Educational Foundation, Guwahati, Assam, India, for their support.
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.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3]