A rare ocular presentation of melioidosis: A mimic within a mimic

GS Parvathy; Anujeet Paul; Swathi Nagarajan

doi:10.4103/ijo.IJO_1401_21

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CASE REPORT

Year : 2022 | Volume : 2 | Issue : 2 | Page : 483-484

A rare ocular presentation of melioidosis: A mimic within a mimic

GS Parvathy, Anujeet Paul, Swathi Nagarajan
Department of Ophthalmology, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth, Pondicherry, India

Date of Submission	25-May-2021
Date of Acceptance	14-Sep-2021
Date of Web Publication	13-Apr-2022

Correspondence Address:
Anujeet Paul
Department of Ophthalmology, Mahatma Gandhi Medical College and Research Institute, Pondicherry, Sri Balaji Vidyapeeth
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_1401_21

Abstract

Melioidosis, a multi-system infectious disease caused by Burkholderia pseudomallei, has varied clinical presentations. The ocular manifestations though rare are often either infectious or inflammatory sequelae. We present a case of a 49-year-old fisherman, an alcoholic and diabetic, who presented with fever, breathlessness, and altered sensorium. The pus and blood culture showed Burkholderia pseudomallei. Clinically, the patient deteriorated over a few days and succumbed to septicemia. The fundus evaluation at presentation showed multiple cotton wool spots concentric to the disk bilaterally. Subsequently, the patient developed a solitary Roth spot in the right eye coinciding with his clinical deterioration. The association of melioidosis with this fundus picture is presented for its rarity.

Keywords: Cotton wool spots, melioidosis, Roth spots

How to cite this article:
Parvathy G S, Paul A, Nagarajan S. A rare ocular presentation of melioidosis: A mimic within a mimic. Indian J Ophthalmol Case Rep 2022;2:483-4

How to cite this URL:
Parvathy G S, Paul A, Nagarajan S. A rare ocular presentation of melioidosis: A mimic within a mimic. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 24];2:483-4. Available from: https://www.ijoreports.in/text.asp?2022/2/2/483/342883

Melioidosis is a multi-system infectious disease endemic to the tropical regions of southeast Asia caused by the bacterium Burkholderia pseudomallei and commonly observed in immunocompromised males.^[1],[2],[3] The disease frequently presents as fever with pneumonia.^[4] The ocular involvement is observed in less than 1% of the cases. The common presentations include orbital cellulitis, pre-septal cellulitis, and pan ophthalmitis.^[1],[2],[5] We report an unusual occurrence of melioidosis presenting with multiple cotton wool spots concentric to the disk.

Case Report

A 49-year-old fisherman presented with altered sensorium and breathlessness warranting assisted ventilation. An alcoholic and diabetic with poor glycemic control, the patient had fever with chills and rigor for over 1-month duration for which the patient took over-the-counter medications. His blood pressure on presentation was 80/60 mmHg. The examination of the anterior segment was unremarkable bilaterally. The fundus examination revealed bilateral cotton wool spots in the posterior pole with well-defined disk margins.

The work-up for pyrexia of unknown origin revealed normocytic normochromic anemia, thrombocytopenia with a platelet count of 60,000/cumm and a total count of white blood cells of 13,000 cells/cumm with neutrophilia (differential count of neutrophils 82%). The HbA1C was 11.3% while the routine examination of urine revealed marked bacteriuria with pus cells around three to four.

The blood culture and pus culture from an anterior chest wall abscess revealed Burkholderia pseudomallei which was sensitive to ceftazidime, imipenem, meropenem, piperacillin, and cefoperazone. [Figure 1]. The patient was negative for Coronavirus disease 19 and human immunodeficiency virus. With a diagnosis of septicemia secondary to melioidosis, the patient was started on intravenous 1 g ceftazidime. He was subsequently started on triple inotrope support (noradrenaline, vasopressin, and adrenaline infusion) in view of the recurrent hypotensive spells.

Figure 1: Gram stain showing Burkholderia pseudomallei in characteristic safety pin appearance

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Over the next 5 days, the patient deteriorated and in addition to the previously observed cotton wool spots, the patient also developed a solitary Roth spot in the right eye. [Figure 2]. The patient subsequently developed lobar pneumonia, acute respiratory distress syndrome, acute kidney injury, encephalopathy, multi-organ dysfunction syndrome, and succumbed to his illness.

Figure 2: Fundus photographs taken using a BOSCH portable handheld fundus camera (a) Color fundus photo of the right eye showing cotton wool spots in the posterior pole and well-defined disk margins along with a solitary Roth spot adjacent to the disk. (b) Color fundus photo of the left eye showing cotton wool spots in the posterior pole with well-defined disk margins

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Discussion

Burkholderia pseudomallei is a gram-negative, motile, indole-negative, oxidase-positive, non-spore-forming facultative anaerobic bacillus usually present in the soil and surface water.^[1],[2] On sheep blood agar, it is typically small, smooth, cream-colored with a metallic sheen which may develop a dry or wrinkled appearance upon incubation. On MacConkey agar, colonies are lactose non-fermenting and colorless and may develop a metallic sheen with a pinkish, rugose appearance. On Ashdown agar, they are pinpoint-sized and develop into purple, flat, and wrinkled colonies. Under Gram staining, the organism is gram-negative with a safety pin appearance.^[3]

The infection is transmitted by inhalation of dust, drinking contaminated water, or contact with contaminated soil through skin abrasions.^[6] This patient was a fisherman by occupation. He was probably infected by exposure to contaminated water or soil.

With clinical presentations varying from pneumonia, skin infections, neurological deficits, internal organ abscesses, and fulminant septicemia, the disease poses a challenge in identification.^[5] In fact, it has been reported to mimic conditions like a septic abscess, cerebral abscess, subdural empyema, and cavernous sinus thrombosis.^[5],[6]

While India is not endemic to melioidosis, there have been gradually increasing reports of the disease from Tamil Nadu, Karnataka, West Bengal, and Andhra Pradesh.^[7] Factors adversely compromising the patient's immune status like diabetes mellitus, tuberculosis and alcoholism have been reported to be significantly associated with the disease.^[3] This reported patient, a chronic alcoholic and uncontrolled diabetic, was probably exposed to contaminated water.

The reported prevalence of ocular manifestations in this disease is between 0.49 and 1.02%.^[5] A retrospective study analyzing 1,270 cases of melioidosis found ocular manifestations in only 16 cases.^[1] The common presentations included orbital cellulitis (44%), endophthalmitis (25%), pre-septal cellulitis (13%), panophthalmitis (13%), and panuveitis (6%).^[1] The authors speculated that this represented the hematogenous spread of the infection to the ocular tissues in immunocompromised patients.^{[1],[2],[5],[6],[8],[9]} In the immunocompetent individuals, uveitis, periphlebitis, macular star, and disk edema have been reported. These features are probably better explained by inflammatory reactions secondary to infection.^[4],[10]

Our findings of retinal ischemia, represented by cotton wool spots in both eyes have not been reported previously. The initial picture may be confusing as it mimics post-febrile retinopathy, microangiopathy, and Purtscher-like retinopathy. With evolving Roth spots, these lesions probably depict progressive septicemia and attendant deranged coagulation profile. Poor host immunity and a prolonged period of undiagnosed disease probably contributed to the severity of the disease and the ocular manifestations observed in this patient.

Conclusion

Melioidosis is associated with a significant mortality rate attributable to severe sepsis and its complications. The cotton wool spots and Roth spots may be an unusual presentation in such a case.

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

Nil.

Conflicts of interest

There are no conflicts of interest.

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