Chitosan-associated vasculitis with eye and sensorineural symptoms following postpartum haemorrhage: a case-based review.

backgroundCogan syndrome is a rare autoimmune disorder characterised by ocular inflammation and audiovestibular dysfunction. Similar clinical phenotypes are being described in association with the haemostatic biomaterial chitosan. This "Cogan-like" entity being referred to as chitosan-associated vasculitis with eye and sensorineural symptoms (CAVES), is increasingly being reported in the literature. CASE PRESENTATION: We report the first Irish case of a 37-year-old woman presenting twenty-four hours postpartum with bilateral ocular pain, reduced visual acuity, and photophobia following management of postpartum haemorrhage using chitosan-based uterine tamponade. Ophthalmic examination demonstrated bilateral panuveitis with hypopyon, interstitial keratitis, and retinal haemorrhages. She concurrently reported vertigo and acute hearing loss. Infectious and systemic autoimmune workup revealed findings of unknown significance. DISCUSSION: The temporal relationship between chitosan exposure and the development of ocular and audiovestibular inflammation suggests a possible immune-mediated "Cogan-like" syndrome. The patient responded well to corticosteroid therapy, including subconjunctival dexamethasone and a tapering course of topical and systemic steroids. She had gradual improvement of her symptoms over several weeks though residual macular and optic nerve thinning persisted and was associated with residual deficit in best corrected visual acuity. In addition to this she experienced ongoing audiovestibular dysfunction with occasional vertigo.

conclusionThis case adds to the emerging literature of a potential novel trigger for a "Cogan-like" syndrome and adds to the cases of CAVES. This case also underscores the importance of early recognition and multidisciplinary management in postpartum patients presenting with ocular and vestibuloauditory symptoms.