Diplopia and Vision Loss Associated With Presumed Systemic Lymphohistiocytic Disease: A Case Report

Our plain-English summary of Diplopia and Vision Loss Associated With Presumed Systemic Lymphohistiocytic Disease: A Case Report, by N. Abu-Yaghi, A. Obiedat, M. Abdaljaleel, et al (Clinical Medicine Insights. Case Reports, 2022).

The study

• Authors: N. Abu-Yaghi, A. Obiedat, M. Abdaljaleel, T. Ar'ar, M. Al-Abbadi
• Journal: Clinical Medicine Insights. Case Reports
• Published: 2022;15:11795476221137262
• Type of article: Case report
• Read it: PubMed · Publisher · Free full text

Abstract

Systemic lymphohistiocytic infiltration is a rare peculiar condition that can raise the possibility of more specific entities such as autoimmune disease, drug interaction, viral or mycobacterial infection, or malignancy. A hyper-inflammatory state can ensue leading to multi-organ failure. We report the case of a 42-year-old Jordanian male with a past history of moderate Covid-19 infection presenting with binocular diplopia and acute loss of vision in the left eye. Ophthalmic evaluation revealed limitation of extraocular motility in all directions of gaze in the left eye and a visual acuity of 6/30 with a sluggish pupil. Orbital imaging revealed a 10 mm mass at the orbital apex suspicious of malignant metastasis. A positron emission tomography CT scan showed significant pleural thickening and was highly suggestive of metastatic mesothelioma seeding to the orbit, liver, and bone. A CT guided biopsy of the right lung was negative for malignancy but had features of lymphohistiocytic pleuritis. The patient dramatically succumbed to respiratory and renal failure. Systemic lymphohistiocytic infiltration is an aggressive benign inflammatory process that may masquerade as malignancy and raise the possibility of past viral infections, autoimmune diseases, or cancer. A high index of suspicion and a multidisciplinary approach is warranted. In this particular devastating instance, a diagnostic dilemma presented to the eye clinic with diplopia, loss of vision, and an orbital mass, culminating in rapid onset respiratory and renal failure and death.

© The Author(s) 2022.

In plain English

This case report described one 42-year-old man seen at an eye clinic in Jordan with double vision and blurred vision in the left eye. About four months earlier he had moderate Covid-19. A pleural biopsy, taken because of breathlessness and pleural thickening, had shown benign inflammation. His left-eye vision was 6/30, and MRI showed a small lesion at the back of the left orbit pressing on the optic nerve. A PET-CT scan showed activity at the orbital lesion that was highly suggestive of malignancy, and right pleural thickening described as highly suspicious for mesothelioma. Liver and adrenal lesions were deemed likely metastatic. A CT-guided biopsy of the right lung then showed a benign lymphohistiocytic inflammatory process with no evidence of malignancy or specific infections. The patient died 3 weeks after first presenting, and no autopsy was done. The authors concluded that systemic lymphohistiocytic infiltration is a masquerader and that a high index of suspicion and a multidisciplinary approach is warranted. The article does not mention asbestos exposure.

We wrote this plain-English summary from the full article with the help of AI, and a second AI model checked each sentence against the article. It is not the authors' wording; the abstract and the article are their own account.

What the study did and found

• Design: case report
• Who was studied: One 42-year-old man presenting to an ophthalmology clinic; the authors are at the University of Jordan and the case was handled under Jordan University Hospital's review board
• Exposure: No asbestos, fiber or occupational exposure is described. The patient's prior history included moderate Covid-19 about 4 months before presentation; he was never vaccinated against Covid-19.
• Main results:
  • A pleural biopsy taken before the eye presentation was reported as benign inflammation with no malignancy
  • The left eye had an unaided visual acuity of 6/30 that did not improve with refraction (the right eye was 6/6)
  • MRI showed a small soft tissue lesion at the apex of the left orbit compressing the optic nerve
  • PET-CT showed 2.5 cm nodular thickening of the right pleura with intense activity, which the figure caption describes as highly suspicious for mesothelioma
  • The CT-guided right lung biopsy showed a benign lymphohistiocytic inflammatory process of the pleura with no malignancy or specific infections
  • The patient died 3 weeks after his initial presentation
  • The authors considered a Covid-19-related cause less likely because of the time gap between recovery and the onset of breathlessness
• The authors' conclusion: The authors concluded that systemic lymphohistiocytic infiltration can masquerade as other diseases and that a high index of suspicion and a multidisciplinary approach is warranted.
• Limitations the authors state:
  • No autopsy was performed to look for other possible pathology in multiple organs
  • An autoimmune cause could not be excluded

Topics

Medical terms in this study, explained in our Lexicon: Metastasis, CT Scan, Pleural Thickening, Mesothelioma, Biopsy, Multidisciplinary Team.

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