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Alkaptonuric ochronosis of congenital bicuspid aortic valve—a case report

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Abstract

Aortic valve diseases are most commonly caused by degenerative changes, congenital malformation and rheumatic affection of valves. A very rare cause is the deposition of foreign material in the valvular tissues as a result of alkaptonuria. In this case report, we present a case of alkaptonuric ochronosis of congenital bicuspid aortic valve, presented with severe aortic valve stenosis and treated with aortic valve replacement surgery.

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References

  1. Fisher AA, Davis MW. Alkatonuric ochronosis with aortic valve and joint replacements and femoral fracture: a case report and literature review. Clin Med Res. 2004;2:209–15.

    Article  PubMed  PubMed Central  Google Scholar 

  2. Phornphutkul C, Introne WJ, Perry MB, et al. Natural history of alkaptonuria. N Engl J Med. 2002;347:2111–21.

    Article  CAS  PubMed  Google Scholar 

  3. Atalay A, Gocen U, Basturk Y, Kozanoglu E, Yaliniz H. Ochronotic involvement of the aortic and mitral valves in a 72-year-old man. Tex Heart Inst J. 2015;42:84–6.

    Article  PubMed  PubMed Central  Google Scholar 

  4. Erek E, Casselman FR, Vanermen H. Cardiac ochronosis: valvular heart disease with dark green discoloration of the leaflets. Tex Heart Inst J. 2004;31:445–7.

    PubMed  PubMed Central  Google Scholar 

  5. Gaines Jr JJ, Pai GM. Cardiovascular ochronosis. Arch Pathol Lab Med. 1987;111:991–4.

    PubMed  Google Scholar 

  6. Hannoush H, Introne WJ, Chen MY, et al. Aortic stenosis and vascular calcifications in alkaptonuria. Mol Genet Metab. 2012;105:198–202.

    Article  CAS  PubMed  Google Scholar 

  7. Yoshikai M, Murayama J, Yamada N. Aortic valve regurgitation in alkaptonuria. J Heart Valve Dis. 2004;13:863–5.

    PubMed  Google Scholar 

  8. Kenny D, Ptacin MJ, Bamrah VS, Almagro U. Cardiovascular ochronosis: a case report and review of the medical literature. Cardiology. 1990;77:477–83.

    Article  CAS  PubMed  Google Scholar 

  9. Steger CM. Aortic valve ochronosis: a rare manifestation of alkaptonuria. BMJ Case Rep. 2011; doi:10.1136/bcr.04.2011.4119.

    Google Scholar 

  10. Ranganath LR, Jarvis JC, Gallagher JA. Recent advances in management of alkaptonuria (invited review; best practice article). J Clin Pathol. 2013;66:367–73.

    Article  CAS  PubMed  Google Scholar 

Download references

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Authors and Affiliations

  1. Department of Cardiovascular and Thoracic Surgery, Grant Medical College & Sir J.J. Group of Hospitals, 5th floor, Byculla, Mumbai, Maharashtra, 400008, India

    Hrishikesh Sukhadeo Parashi, Manoj Murlidhar Joshi, Manish Ramdhan Jadhao, Kunal Hemant Rawekar & Krishnarao Narayan Bhosle

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  1. Hrishikesh Sukhadeo Parashi
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  2. Manoj Murlidhar Joshi
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  3. Manish Ramdhan Jadhao
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  4. Kunal Hemant Rawekar
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  5. Krishnarao Narayan Bhosle
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Correspondence to Hrishikesh Sukhadeo Parashi.

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The authors declare that they have no conflict of interest.

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All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

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Informed consent was obtained from all individual participants included in the study.

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Parashi, H.S., Joshi, M.M., Jadhao, M.R. et al. Alkaptonuric ochronosis of congenital bicuspid aortic valve—a case report. Indian J Thorac Cardiovasc Surg 33, 155–158 (2017). https://doi.org/10.1007/s12055-017-0487-0

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  • DOI: https://doi.org/10.1007/s12055-017-0487-0

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