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Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report.

Publication ,  Journal Article
Kiyeng, JC; Siika, A; Koech, C; Bloomfield, GS
Published in: J Med Case Rep
May 25, 2016

BACKGROUND: Hereditary hemorrhagic telangiectasia is a rare autosomal dominant inherited disease characterized by vascular dysplasia. To the best of our knowledge, we report the first case in the literature of definite hereditary hemorrhagic telangiectasia diagnosed in western Kenya, a resource-limited setting with limited treatment options. CASE PRESENTATION: A 60-year-old black Kenyan woman was admitted 1 year ago to a hospital in western Kenya with an 11-year history of recurrent spontaneous epistaxis. Her physical examination revealed that she had telangiectasias on the tongue and hard palate, severe pallor, and hepatomegaly. A chest radiograph revealed right middle lobe opacity. After a positive saline contrast echocardiography, she underwent contrast-enhanced chest computed tomography, which revealed a large pulmonary arteriovenous malformation and multiple hepatic arteriovenous malformations. Therefore, she fulfilled criteria for definite hereditary hemorrhagic telangiectasia. She was managed with nasal packing, tranexamic acid, oral ferrous sulfate, and blood transfusions, as other treatment options were unavailable in this setting. CONCLUSIONS: This rare case of hereditary hemorrhagic telangiectasia demonstrates that it occurs in an African population and that diagnostic challenges in resource-limited settings can be surmounted. Treatment options remain limited in these settings.

Duke Scholars

Published In

J Med Case Rep

DOI

EISSN

1752-1947

Publication Date

May 25, 2016

Volume

10

Issue

1

Start / End Page

126

Location

England

Related Subject Headings

  • Tranexamic Acid
  • Tomography, X-Ray Computed
  • Telangiectasia, Hereditary Hemorrhagic
  • Pulmonary Veins
  • Pulmonary Artery
  • Middle Aged
  • Liver
  • Kenya
  • Humans
  • Hepatic Veins
 

Citation

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ICMJE
MLA
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Kiyeng, J. C., Siika, A., Koech, C., & Bloomfield, G. S. (2016). Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report. J Med Case Rep, 10(1), 126. https://doi.org/10.1186/s13256-016-0909-4
Kiyeng, Joan Chepkorir, Abraham Siika, Cornelius Koech, and Gerald S. Bloomfield. “Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report.J Med Case Rep 10, no. 1 (May 25, 2016): 126. https://doi.org/10.1186/s13256-016-0909-4.
Kiyeng JC, Siika A, Koech C, Bloomfield GS. Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report. J Med Case Rep. 2016 May 25;10(1):126.
Kiyeng, Joan Chepkorir, et al. “Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report.J Med Case Rep, vol. 10, no. 1, May 2016, p. 126. Pubmed, doi:10.1186/s13256-016-0909-4.
Kiyeng JC, Siika A, Koech C, Bloomfield GS. Definite hereditary hemorrhagic telangiectasia in a 60-year-old black Kenyan woman: a case report. J Med Case Rep. 2016 May 25;10(1):126.
Journal cover image

Published In

J Med Case Rep

DOI

EISSN

1752-1947

Publication Date

May 25, 2016

Volume

10

Issue

1

Start / End Page

126

Location

England

Related Subject Headings

  • Tranexamic Acid
  • Tomography, X-Ray Computed
  • Telangiectasia, Hereditary Hemorrhagic
  • Pulmonary Veins
  • Pulmonary Artery
  • Middle Aged
  • Liver
  • Kenya
  • Humans
  • Hepatic Veins