Bilateral Peripheral Ulcerative Keratitis with Corneal Perforation Revealing Acute Myeloid Leukaemia in a Young Adult: A Case Report
Lotfi Chaabani *
Department of Ophthalmology, Badr al-Din al-Alawi University Hospital, Kasserine, Tunisia; Faculty of Medicine of Sousse, University of Sousse, Sousse, Tunisia.
Ines Bouallegui
Department of Ophthalmology, Badr al-Din al-Alawi University Hospital, Kasserine, Tunisia; Faculty of Medicine of Sousse, University of Sousse, Sousse, Tunisia.
Mohamed Said
Department of Ophthalmology, Badr al-Din al-Alawi University Hospital, Kasserine, Tunisia; Faculty of Medicine of Sousse, University of Sousse, Sousse, Tunisia.
Yosra Hamdi
Department of Ophthalmology, Badr al-Din al-Alawi University Hospital, Kasserine, Tunisia; Faculty of Medicine of Sousse, University of Sousse, Sousse, Tunisia.
*Author to whom correspondence should be addressed.
Abstract
Background: Peripheral ulcerative keratitis (PUK) is a destructive corneal disorder that may progress rapidly to perforation and can occasionally indicate serious systemic disease.
Aim: To report a rare and clinically important case of bilateral peripheral ulcerative keratitis (PUK), complicated by unilateral corneal perforation, as the initial ophthalmic presentation that led to the diagnosis of acute myeloid leukaemia (AML) in a young adult.
Presentation of Case: A 31-year-old man with no previous ocular or systemic history presented with severe asthenia, dyspnoea, diffuse arthromyalgia, and bilateral ocular pain, redness, tearing, photophobia and decreased vision of approximately three weeks' duration. Best-corrected visual acuity was counting fingers in the right eye and light perception in the left eye. Slit-lamp examination showed bilateral conjunctival hyperaemia, palpebral conjunctival fibrosis, a superior temporal peripheral corneal ulcer in the right eye, and an inferior temporal corneal perforation sealed by iris tissue in the left eye. Microbiological and autoimmune investigations were non-contributory. Haematological assessment, followed by bone marrow examination, confirmed AML. Detailed AML subtype-defining investigations, the coagulation profile, and complete treatment-outcome data were not available in the ophthalmic record and are acknowledged as limitations. Multidisciplinary management included ocular surface stabilisation, planned tectonic corneal surgery, and systemic antileukaemic treatment. The patient died before completion of treatment.
Discussion: PUK is usually immune-mediated or infectious, but it may rarely reveal an occult haematological malignancy. In this patient, bilateral, rapidly progressive PUK with corneal perforation, systemic symptoms, negative microbiology and non-contributory autoimmune testing supported a clinical association with AML after exclusion of more common causes. Potential mechanisms include immune-mediated stromal melt, paraneoplastic inflammation, leukostasis-related microvascular injury and AML-associated coagulopathy, although direct ocular tissue infiltration, hyperleukocytosis and disseminated intravascular coagulation were not documented.
Conclusion: Severe, bilateral, or perforating PUK should prompt urgent systemic assessment, including complete blood count and haematological evaluation. Early recognition is important because this ocular emergency may reveal a life-threatening systemic disease requiring immediate multidisciplinary management.
Keywords: Acute myeloid leukaemia, peripheral ulcerative keratitis, corneal perforation, ocular manifestations, haematological malignancy
How to Cite
Downloads
References
Background: Peripheral ulcerative keratitis (PUK) is a destructive corneal disorder that may progress rapidly to perforation and can occasionally indicate serious systemic disease.
Aim: To report a rare and clinically important case of bilateral peripheral ulcerative keratitis (PUK), complicated by unilateral corneal perforation, as the initial ophthalmic presentation that led to the diagnosis of acute myeloid leukaemia (AML) in a young adult.
Presentation of Case: A 31-year-old man with no previous ocular or systemic history presented with severe asthenia, dyspnoea, diffuse arthromyalgia, and bilateral ocular pain, redness, tearing, photophobia and decreased vision of approximately three weeks' duration. Best-corrected visual acuity was counting fingers in the right eye and light perception in the left eye. Slit-lamp examination showed bilateral conjunctival hyperaemia, palpebral conjunctival fibrosis, a superior temporal peripheral corneal ulcer in the right eye, and an inferior temporal corneal perforation sealed by iris tissue in the left eye. Microbiological and autoimmune investigations were non-contributory. Haematological assessment, followed by bone marrow examination, confirmed AML. Detailed AML subtype-defining investigations, the coagulation profile, and complete treatment-outcome data were not available in the ophthalmic record and are acknowledged as limitations. Multidisciplinary management included ocular surface stabilisation, planned tectonic corneal surgery, and systemic antileukaemic treatment. The patient died before completion of treatment.
Discussion: PUK is usually immune-mediated or infectious, but it may rarely reveal an occult haematological malignancy. In this patient, bilateral, rapidly progressive PUK with corneal perforation, systemic symptoms, negative microbiology and non-contributory autoimmune testing supported a clinical association with AML after exclusion of more common causes. Potential mechanisms include immune-mediated stromal melt, paraneoplastic inflammation, leukostasis-related microvascular injury and AML-associated coagulopathy, although direct ocular tissue infiltration, hyperleukocytosis and disseminated intravascular coagulation were not documented.
Conclusion: Severe, bilateral, or perforating PUK should prompt urgent systemic assessment, including complete blood count and haematological evaluation. Early recognition is important because this ocular emergency may reveal a life-threatening systemic disease requiring immediate multidisciplinary management.