ArticleJIMD reports2014
Reversible keratopathy due to hypertyrosinaemia following intermittent low-dose nitisinone in alkaptonuria: a case report.
Article in JIMD reports, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
What it found
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The trial behind it
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Who cites it
17 citing papers in PubMed, 34 citations in OpenAlex.
- Trial
- Managing Alkaptonuria in Absence of Appropriate Medication: A Case Report and Review of Literature.Clinical medicine & research · 2024Review
- Article
- Article
- Association of alkaptonuria and low dose nitisinone therapy with cataract formation in a large cohort of patients.JIMD reports · 2022Article
- Impact of Nitisinone on the Cerebrospinal Fluid Metabolome of a Murine Model of Alkaptonuria.Metabolites · 2022Article
- Long-term low dose nitisinone therapy in adults with alkaptonuria shows no cognitive decline or increased severity of depression.JIMD reports · 2022Article
- Characterization of changes in the tyrosine pathway by 24-h profiling during nitisinone treatment in alkaptonuria.Molecular genetics and metabolism reports · 2022Article
- Vitiligo, alkaptonuria, and nitisinone-A report of three families and review of the literature.JIMD reports · 2021Article
- Article
- Activation of Tyrosine Metabolism in CD13+ Cancer Stem Cells Drives Relapse in Hepatocellular Carcinoma.Cancer research and treatment · 2020Article
- Dietary restriction of tyrosine and phenylalanine lowers tyrosinemia associated with nitisinone therapy of alkaptonuria.Journal of inherited metabolic disease · 2020Observational
- Asymptomatic Corneal Keratopathy Secondary to Hypertyrosinaemia Following Low Dose Nitisinone and a Literature Review of Tyrosine Keratopathy in Alkaptonuria.JIMD reports · 2018Article
- Nitisinone-Induced Keratopathy in Alkaptonuria: A Challenging Diagnosis Despite Clinical Suspicion.JIMD reports · 2018Article
- Acute fatal metabolic complications in alkaptonuria.Journal of inherited metabolic disease · 2016Review
- Osteoarticular cells tolerate short-term exposure to nitisinone-implications in alkaptonuria.Clinical rheumatology · 2016Article
- The role of nitisinone in tyrosine pathway disorders.Current rheumatology reports · 2014Review
Corrections and comments
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Authors and funding
5 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
We describe a patient with ultra-rare disease, alkaptonuria, who developed tyrosine keratopathy following nitisinone therapy of 2 mg on alternate days. His vision became impaired approximately 7 weeks following the commencement of nitisinone and ophthalmological examination at week nine showed characteristic dendritic keratopathy associated with tyrosinaemia. The corneal lesion as well as his visual symptoms normalized completely following discontinuation of nitisinone. This is the first documented report of keratopathy due to acquired tyrosinaemia due to very low-dose nitisinone.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.