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Large pulmonary arteriovenous malformation with hyperammonemia.

Teruhisa Takuwa1, Shinkichiro Yoshioka, Yoshihiro Miyata

  • 1Division of Frontier Medical Science, Department of Surgery, Programs for Biomedical Science Research, Graduate School of Biomedical Science, Hiroshima University, Hiroshima, Japan.

The Japanese Journal of Thoracic and Cardiovascular Surgery : Official Publication of the Japanese Association for Thoracic Surgery = Nihon Kyobu Geka Gakkai Zasshi
|November 24, 2004
PubMed
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Surgical resection of a large pulmonary arteriovenous malformation (PAVM) effectively treated hyperammonemia in a patient with hereditary hemorrhagic telangiectasia. This intervention improved oxygenation and ammonia levels, highlighting PAVM as a potential cause of hyperammonemia.

Area of Science:

  • Vascular Medicine
  • Hepatology
  • Pulmonology

Background:

  • Hereditary hemorrhagic telangiectasia (HHT) is a genetic disorder causing abnormal blood vessel formation.
  • Pulmonary arteriovenous malformations (PAVMs) are common in HHT and can lead to significant shunting and hypoxemia.
  • Hyperammonemia, typically associated with liver dysfunction, can present atypically in complex vascular conditions.

Observation:

  • A 45-year-old female with HHT presented with fatigue and severe anemia from gastric bleeding.
  • Investigations revealed a large PAVM, hepatic arteriovenous, and portovenous shunts.
  • The patient exhibited hypoxemia and hyperammonemia despite normal liver function tests.

Findings:

  • Surgical resection of the large PAVM was performed successfully.

Related Experiment Videos

  • Postoperatively, the patient's hyperammonemia resolved without specific hepatic treatment.
  • Improvement in hyperammonemia correlated with the resolution of the PAVM and improved oxygenation.

    • Implications:

    • Large PAVMs can cause or exacerbate hyperammonemia, even with preserved liver function.
    • Surgical management of PAVMs may be a crucial therapeutic strategy for unexplained hyperammonemia in HHT patients.
    • This case underscores the importance of considering complex vascular anomalies in the differential diagnosis of hyperammonemia.