[Skip to Content]
[Skip to Content Landing]

Acute-Onset Orbital Inflammation in a Patient With Multiple Myeloma

Educational Objective
Based on this clinical scenario and the accompanying image, understand how to arrive at a correct diagnosis.
1 Credit CME

A 72-year-old male patient presented with 2 days of conjunctival injection in the right eye, chemosis, eyelid edema, and pain with eye movements. His medical history included hypothyroidism, psoriasis, and recent diagnosis of multiple myeloma, and he was taking daratumumab and zoledronic acid. He denied eye trauma, dental surgery, or sinus disease. Examination of the right eye was notable for visual acuity of 20/50, reduced ocular motility in all gazes, proptosis, and chemosis. There was no afferent pupillary defect. Examination of the left eye was unremarkable. Computed tomography of the orbits with contrast demonstrated right preseptal edema and intraorbital fat stranding of the extraconal and intraconal fat without sinus disease. Nasal endoscopy performed by the otolaryngology service showed no evidence of invasive fungal sinusitis. The patient received a single dose of intravenous vancomycin and ceftriaxone, then was transitioned to ampicillin-sulbactam for presumed orbital cellulitis. After 48 hours, motility, proptosis, and chemosis worsened (Figure 1A).

Please finish quiz first before checking answer.

You answered correctly!

Read the answer below and download your certificate.

You answered incorrectly.

Read the discussion below and retake the quiz.

Zoledronic acid-induced orbital inflammation

C. Start corticosteroids

This patient with multiple myeloma presented with acute unilateral orbital congestion and ophthalmoplegia and was initially treated for presumed orbital cellulitis. His condition worsened despite treatment with broad-spectrum intravenous antibiotics, thus raising suspicion for an alternative etiology. Although invasive fungal sinusitis was considered on the differential diagnosis due to his immunocompromised state, neither bedside nasal endoscopy nor computed tomography of the sinus was suggestive of the disease. Therefore, antifungals were not started (choice A), and sinus biopsies were not pursued (choice B).

An inflammatory etiology was considered, given the multifocal involvement observed on imaging (right orbit, left sphenoid sinus, masticator space). On further probing, the patient reported he received his first infusion of zoledronic acid, a bisphosphonate, as part of his treatment for multiple myeloma 3 days prior to presentation. Given the acute onset of symptoms within 72 hours of the first zoledronic acid infusion, a diagnosis of orbital inflammatory disease due to bisphosphonate infusion was suspected. Treatment with intravenous pulse methylprednisolone was initiated (choice C). Due to the suspected etiology and the acute nature of the presentation, an orbital biopsy would not be the best next option (choice D).

Survey Complete!

Sign in to take quiz and track your certificates

Buy This Activity
Our websites may be periodically unavailable between 12:00am CT March 25, 2023 and 4:00pm CT March 26, 2023 for regularly scheduled maintenance.

JN Learning™ is the home for CME and MOC from the JAMA Network. Search by specialty or US state and earn AMA PRA Category 1 Credit(s)™ from articles, audio, Clinical Challenges and more. Learn more about CME/MOC

CME Disclosure Statement: Unless noted, all individuals in control of content reported no relevant financial relationships. If applicable, all relevant financial relationships have been mitigated.

Article Information

Corresponding Author: George Villatoro, MD, Department of Ophthalmology, Division of Oculofacial Plastic and Reconstructive Surgery, Shiley Eye Institute, University of California, San Diego, 9415 Campus Point Dr, La Jolla, CA 92093 (gvillato@health.ucsd.edu).

Published Online: March 2, 2023. doi:10.1001/jamaophthalmol.2023.0021

Conflict of Interest Disclosures: Dr Liu reported grants from Horizon Therapeutics while serving as principal investigator for a phase IV clinical trial and royalties from Wolters Kluwers Health outside the submitted work. No other disclosures were reported.

Additional Contributions: We thank the patient for granting permission to publish this information.

References
1.
Drake  MT , Clarke  BL , Khosla  S .  Bisphosphonates: mechanism of action and role in clinical practice.   Mayo Clin Proc. 2008;83(9):1032-1045. doi:10.4065/83.9.1032PubMedGoogle ScholarCrossref
2.
Khalid  MF , Micieli  J , DeAngelis  D , Micieli  JA .  Zoledronic acid-induced orbital inflammation.   BMJ Case Rep. 2021;14(8):e245359. doi:10.1136/bcr-2021-245359PubMedGoogle ScholarCrossref
3.
Wolpert  LE , Watts  AR .  Zoledronate-induced anterior uveitis, scleritis and optic neuritis: a case report.   N Z Med J. 2021;134(1537):91-94. https://journal.nzma.org.nz/journal-articles/zoledronate-induced-anterior-uveitis-scleritis-and-optic-neuritis-a-case-reportPubMedGoogle Scholar
4.
Pirbhai  A , Rajak  SN , Goold  LA ,  et al.  Bisphosphonate-induced orbital inflammation: a case series and review.   Orbit. 2015;34(6):331-335. doi:10.3109/01676830.2015.1078380PubMedGoogle ScholarCrossref
5.
Phillips  PM , Newman  SA .  Orbital inflammatory disease after intravenous infusion of zoledronate for treatment of metastatic renal cell carcinoma.   Arch Ophthalmol. 2008;126(1):137-139. doi:10.1001/archophthalmol.2007.17PubMedGoogle ScholarCrossref
6.
Herrera  I , Kam  Y , Whittaker  TJ , Champion  M , Ajlan  RS .  Bisphosphonate-induced orbital inflammation in a patient on chronic immunosuppressive therapy.   BMC Ophthalmol. 2019;19(1):51. doi:10.1186/s12886-019-1063-8PubMedGoogle ScholarCrossref
7.
Peterson  JD , Bedrossian  EH  Jr .  Bisphosphonate-associated orbital inflammation—a case report and review.   Orbit. 2012;31(2):119-123. doi:10.3109/01676830.2011.648818PubMedGoogle ScholarCrossref
AMA CME Accreditation Information

Credit Designation Statement: The American Medical Association designates this Journal-based CME activity activity for a maximum of 1.00  AMA PRA Category 1 Credit(s)™. Physicians should claim only the credit commensurate with the extent of their participation in the activity.

Successful completion of this CME activity, which includes participation in the evaluation component, enables the participant to earn up to:

  • 1.00 Medical Knowledge MOC points in the American Board of Internal Medicine's (ABIM) Maintenance of Certification (MOC) program;;
  • 1.00 Self-Assessment points in the American Board of Otolaryngology – Head and Neck Surgery’s (ABOHNS) Continuing Certification program;
  • 1.00 MOC points in the American Board of Pediatrics’ (ABP) Maintenance of Certification (MOC) program;
  • 1.00 Lifelong Learning points in the American Board of Pathology’s (ABPath) Continuing Certification program; and
  • 1.00 CME points in the American Board of Surgery’s (ABS) Continuing Certification program

It is the CME activity provider's responsibility to submit participant completion information to ACCME for the purpose of granting MOC credit.

Close
Want full access to the AMA Ed Hub?
After you sign up for AMA Membership, make sure you sign in or create a Physician account with the AMA in order to access all learning activities on the AMA Ed Hub
Buy this activity
Close
Want full access to the AMA Ed Hub?
After you sign up for AMA Membership, make sure you sign in or create a Physician account with the AMA in order to access all learning activities on the AMA Ed Hub
Buy this activity
Close
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Education Center Collection Sign In Modal Right
Close

Name Your Search

Save Search
With a personal account, you can:
  • Access free activities and track your credits
  • Personalize content alerts
  • Customize your interests
  • Fully personalize your learning experience
Close
Close

Lookup An Activity

or

My Saved Searches

You currently have no searches saved.

Close

My Saved Courses

You currently have no courses saved.

Close