June 2021
Volume 62, Issue 8
Open Access
ARVO Annual Meeting Abstract  |   June 2021
Idiopathic Orbital Inflammation Refractory to Rituximab but Sensitive to Methotrexate
Author Affiliations & Notes
  • Rusdeep Mundae
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Andrew Harrison
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Ali Mokhtarzadeh
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Sandra Rocio Montezuma
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Dara Koozekanani
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Michael S Lee
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Collin McClelland
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Joshua H Hou
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Karen R Armbrust
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Justin Yamanuha
    Ophthalmology, University of Minnesota System, Minneapolis, Minnesota, United States
  • Footnotes
    Commercial Relationships   Rusdeep Mundae, None; Andrew Harrison, None; Ali Mokhtarzadeh, None; Sandra Montezuma, None; Dara Koozekanani, None; Michael Lee, None; Collin McClelland, None; Joshua Hou, None; Karen Armbrust, None; Justin Yamanuha, None
  • Footnotes
    Support  None
Investigative Ophthalmology & Visual Science June 2021, Vol.62, 1399. doi:
  • Views
  • Share
  • Tools
    • Alerts
      ×
      This feature is available to authenticated users only.
      Sign In or Create an Account ×
    • Get Citation

      Rusdeep Mundae, Andrew Harrison, Ali Mokhtarzadeh, Sandra Rocio Montezuma, Dara Koozekanani, Michael S Lee, Collin McClelland, Joshua H Hou, Karen R Armbrust, Justin Yamanuha; Idiopathic Orbital Inflammation Refractory to Rituximab but Sensitive to Methotrexate. Invest. Ophthalmol. Vis. Sci. 2021;62(8):1399.

      Download citation file:


      © ARVO (1962-2015); The Authors (2016-present)

      ×
  • Supplements
Abstract

Purpose : Idiopathic orbital inflammation (IOI) represents a spectrum of non-infectious orbital processes that are managed with corticosteroids and/or steroid sparing immunosuppressive therapies in refractory or recurrent cases. We present two patients with IOI who were resistant to rituximab infusions but subsequently responded to oral methotrexate (MTX).

Methods : Case series

Results : Patient #1: A 73-year old man with a 16-year history of biopsy-proven recurrent left IOI involving the inferior rectus (Fig. 1a) presented with diplopia and persistent optic disc edema (ODE) (Fig. 2a) first noted 5 months prior. Previous treatments included prednisone up to 80 mg daily, orbital triamcinolone injections, and two rituximab infusion courses without improvement. He was started on oral MTX 15 mg weekly with a taper of prednisone. His ODE and diplopia improved at 6 weeks; however, treatment was discontinued after six months due to his concerns related to COVID-19. All findings resolved three months after stopping MTX.

Patient #2: A 67-year old woman presented with a 3-year history of a right orbital mass involving the lacrimal gland and extraocular muscles (Fig. 1b) causing proptosis and diplopia. Orbital biopsy showed chronic inflammatory changes and was negative for IgG4 or lymphoma. Systemic inflammatory and infectious workup was unremarkable. She had minimal improvement in proptosis and diplopia after two courses of rituximab with intermittent oral prednisone. She later presented with ODE, cystoid macular edema, and an inferotemporal exudative retinal detachment (RD) (Fig. 2b). The ocular manifestations improved with a 10 day course of prednisone 40 mg/day but recurred on prednisone taper. All findings resolved after 2 months of MTX 20 mg weekly.

Conclusions : While anti-metabolites are typically used early in the course of recurrent IOI with rituximab reserved for more recalcitrant disease, this study demonstrates that MTX may provide benefit for IOI refractory to rituximab with secondary ocular manifestations. This may be due to more gradual suppression of the immune system with MTX.

This is a 2021 ARVO Annual Meeting abstract.

 

Figure 1a (left): Patient#1 CT scan showing well circumscribed orbital mass
Figure 1b (right): Patient#2 MRI scan demonstrating diffuse orbital mass

Figure 1a (left): Patient#1 CT scan showing well circumscribed orbital mass
Figure 1b (right): Patient#2 MRI scan demonstrating diffuse orbital mass

 

Fundus Photos.
Figure 2a (left): Patient #1 left eye with 360 degrees of ODE.
Figure 2b (right)Patient #2 right eye with ODE and inferotemporal exudative RD

Fundus Photos.
Figure 2a (left): Patient #1 left eye with 360 degrees of ODE.
Figure 2b (right)Patient #2 right eye with ODE and inferotemporal exudative RD

×
×

This PDF is available to Subscribers Only

Sign in or purchase a subscription to access this content. ×

You must be signed into an individual account to use this feature.

×