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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 762-764

Orbital cellulitis and exudative retinal detachment in a case of immune thrombocytopenic purpura treated with rituximab


Department of Ophthalmology, Institute of Medical Sciences and SUM Hospital, SOA University, Bhubaneswar, Odisha, India

Date of Submission25-Jan-2021
Date of Acceptance10-Apr-2021
Date of Web Publication09-Oct-2021

Correspondence Address:
Dr. Pradeep K Panigrahi
Department of Ophthalmology, Institute of Medical Sciences and SUM Hospital, SOA University, 8-Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_213_21

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  Abstract 


An 18-year-old female with relapsed immune thrombocytopenic purpura presented with painful swelling and loss of vision in the right eye 2 days following treatment with systemic steroids and rituximab. She was diagnosed with orbital cellulitis and exudative retinal detachment in the right eye. Following cessation of systemic therapy and institution of broad-spectrum antibiotics, there was a prompt reversal of cellulitis and exudative detachment. Ocular complications can arise following treatment with rituximab. Venous congestion and reactive edema across the sclera can lead to exudative retinal detachment in orbital cellulitis. Early diagnosis and prompt treatment are associated with a good prognosis.

Keywords: Corticosteroids, exudative retinal detachment, orbital cellulitis, rituximab


How to cite this article:
Panigrahi PK, Minj A, Satapathy J, Das S. Orbital cellulitis and exudative retinal detachment in a case of immune thrombocytopenic purpura treated with rituximab. Indian J Ophthalmol Case Rep 2021;1:762-4

How to cite this URL:
Panigrahi PK, Minj A, Satapathy J, Das S. Orbital cellulitis and exudative retinal detachment in a case of immune thrombocytopenic purpura treated with rituximab. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Oct 21];1:762-4. Available from: https://www.ijoreports.in/text.asp?2021/1/4/762/327722



Primary immune thrombocytopenic purpura (ITP) is an autoimmune disorder characterized by low platelet count and mucocutaneous bleeding.[1] Therapy with systemic corticosteroids forms the first line of therapy, but as in any autoimmune disorder, there is a high risk of relapse. Second-line therapy includes intravenous immunoglobulins, rituximab, or prolonged immunosuppression.[2] We present a rare case of a patient with relapsed ITP who presented with orbital cellulitis and exudative retinal detachment following treatment with rituximab.


  Case Report Top


An 18-year-old female presented with pain, redness, swelling, and sudden loss of vision in the right eye (RE) of 2 days duration. She was a known case of ITP. She had been previously treated with systemic steroids and intravenous (IV) immunoglobulins. She had received 1 g/kg of intravenous immunoglobulins over a period of 5 days 2 months back. She was also treated with oral prednisolone 50 mg for 1 month followed by a tapering dose of 10 mg/week. Baseline blood investigations showed a total platelet count of 1,00,000/μL of blood. Other blood parameters were within the normal limits. She had received rituximab (100 mg IV) and intravenous steroids (dexamethasone 40 mg IV once a day for 2 days) for her ITP treatment during the present admission. Two days following the treatment with rituximab, the patient complained of painful swelling and loss of vision in RE. On bedside examination, visual acuity in RE was finger counting close to face and counting fingers more than 3 m in the left eye (LE). There was significant periorbital edema, conjunctival chemosis, and mild corneal haze in RE [Figure 1]a. The anterior chamber appeared clear, and the pupillary reaction was sluggish in RE. Fundoscopy done using indirect ophthalmoscope showed bullous exudative Retinal detachment (RD) in RE. There was no evidence of intraocular inflammation. Both anterior and posterior segment examinations of LE were within normal limits. We made a provisional diagnosis of orbital cellulitis with exudative retinal detachment in RE. Routine blood investigations, blood and urine culture, and imaging of orbits were advised. The patient was apprehensive and did not want further treatment with rituximab as all her symptoms had started post rituximab therapy. Hence, further treatment cycles with rituximab were withheld. The patient was started on IV broad-spectrum antibiotics (piperacillin–tazobactum 4.5 g IV 3 times a day for 7 days), topical moxifloxacin (0.5%) 6 times a day and lubricating gel in RE. Magnetic resonance imaging (MRI) showed right axial proptosis, orbital preseptal thickening, and intra- and extraconal compartment inflammatory fat stranding, confirming orbital cellulitis [Figure 1]b. Klebsiella pneumoniae growth was obtained on blood culture, and the organism was sensitive to the antibiotics already being used. Five days following the presentation, the patient was seen in our outpatient department, and vision in RE had improved to 20/200, N18. Periorbital swelling and conjunctival chemosis had almost resolved [Figure 2]a. Exudative RD in RE had started reducing [Figure 2]b. Optical coherence tomography (OCT) of RE showed neurosensory RD with the subretinal fluid appearing turbid suggestive of high fibrin content [Figure 2]c. The patient was shifted to oral antibiotics 1 week after completion of IV antibiotics. Two weeks following the presentation Visual acuity (VA) in RE was 20/30, N8. The anterior segment was normal. OCT showed decreasing subretinal fluid. At final follow-up, 1 month after presentation, VA was 20/30, N6, with minimal subretinal fluid noted on OCT in RE [Figure 3].
Figure 1: (a) Clinical photograph of the right eye showing significant periorbital edema and conjunctival chemosis. (b) Magnetic resonance imaging confirming orbital cellulitis in the right eye

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Figure 2: (a) Clinical photograph showing almost resolved periorbital edema and conjunctival chemosis 5 days after presentation. (b) Color fundus photograph of right eye showing exudative retinal detachment in right eye 5 days after presentation. Exudative detachment had reduced considerably from the first visit when the patient had been seen at bedside. (c) Optical coherence tomography of the right eye showing neurosensory detachment involving macula

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Figure 3: (a) Color fundus photograph of right eye showing almost totally resolved exudative detachment, 1 month after presentation. (b) Optical coherence tomography of the right eye, 1 month following presentation, showing very shallow subretinal fluid

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  Discussion Top


Rituximab is a chimeric anti-CD20 antibody frequently used as a secondary treatment line in ITP. The mechanism of action involves complex immunomodulation. The recommended dose is 375 mg/m2 weekly for 4 weeks.[3] Side effects of rituximab include immediate infusion-related reactions, vasculitis, and cardiac rhythm abnormalities.[2] A comparative study by Zaja et al.[4] showed the superiority of associating dexamethasone and rituximab against the single drug therapy with dexamethasone as first-line treatment leading to better response in 6 months. Our case had initially been treated with IV immunoglobulins and systemic corticosteroids. There was a relapse when the dosage was being tapered. Following relapse, she was put on combination therapy with dexamethasone and rituximab expecting a longer disease-free period.

Our patient presented with symptoms within 2 days of initiation of the treatment. Dexamethasone and rituximab are both immunosuppressive agents. Orbital cellulitis in RE can be attributed to the transient septicemia that might have developed due to the weakened immunity. There is a possibility of a breach in sterility during the IV fluid administration that could have led to septicemia and secondary orbital cellulitis. The isolated organism was sensitive to the antibiotics administered, and there was a prompt resolution of cellulitis features. Song et al.[5] have reported a case of exudative retinal detachment derived from orbital cellulitis in Mainland China. Their case had severe orbital cellulitis in LE with exudative retinal detachment. Treatment with systemic antibiotics and topical steroids resulted in improvement of orbital cellulitis and resolution of exudative detachment. The authors have postulated that the pathogenesis of exudative retinal detachment derived from orbital cellulitis may be due to venous congestion and reactive edema across the tough fibrous sclera. A similar mechanism might have been in play in our case. The orbital cellulitis component responded to the systemic antibiotics following which there was a gradual reduction in exudative detachment. Systemic corticosteroids are known to cause exudative retinal detachment.[6],[7] Our patient had received IV dexamethasone during her present admission. However, she had also been treated with systemic steroids in the past and had never complained of vision loss following treatment. She had not developed exudative retinal detachment following steroid therapy in the past. So the chances of systemic steroids having caused the exudative detachment in this episode seem to be remote.


  Conclusion Top


To conclude, rituximab and corticosteroids are widely being used to treat a variety of autoimmune diseases. They are potent immunosuppressive agents and can lead to ocular complications. Both the primary physician and the ophthalmologist should be aware of the potential complications arising out of these therapies. Prompt recognition and early treatment of complications are associated with a good prognosis.

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.



 
  References Top

1.
Cines DB, Blanchette VS. Immune thrombocytopenic purpura. N Engl J Med 2002;346:995-1008.  Back to cited text no. 1
    
2.
Onisâi M, Vlădăreanu AM, Spînu A, Găman M, Bumbea H. Idiopathic thrombocytopenic purpura (ITP): New era for an old disease. Rom J Intern Med 2019;57:273-83.  Back to cited text no. 2
    
3.
Provan D, Butler T, Evangelista ML, Amadori S, Newland AC, Stasi R. Activity and safety profile of low-dose rituximab for the treatment of autoimmune cytopenias in adults. Haematologica 2007;92:1695-8.  Back to cited text no. 3
    
4.
Zaja F, Baccarani M, Mazza P, Bocchia M, Gugliotta L, Zaccaria A, et al. Dexamethasone plus rituximab yields higher sustained response rates than dexamethasone monotherapy in adults with primary immune thrombocytopenia. Blood 2010;115:2755-62.  Back to cited text no. 4
    
5.
Song W, Cheng D, Zhang Y. A case report of exudative retinal detachment derived from orbital cellulitis in mainland China. BMC Ophthalmol 2020;20:332.  Back to cited text no. 5
    
6.
Weng CY, Harbour JW. Serous macular detachment following a systemic corticosteroid injection. JAMA Ophthalmol 2015;133:473-4.  Back to cited text no. 6
    
7.
Desatnik HR, Gutman FA. Bilateral exudative retinal detachment complicating systemic corticosteroid therapy in presence of renal failure. Am J Ophthalmol 1996;122:432-4.  Back to cited text no. 7
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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