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Intravascular haemolysis with haemoglobinuria in a splenectomized patient with severe Plasmodium knowlesi malaria

View Article: PubMed Central - PubMed

ABSTRACT

Background: Haemoglobinuria is an uncommon complication of severe malaria, reflecting acute intravascular haemolysis and potentially leading to acute kidney injury. It can occur early in the course of infection as a consequence of a high parasite burden, or may occur following commencement of anti-malarial treatment. Treatment with quinine has been described as a risk factor; however the syndrome may also occur following treatment with intravenous artesunate. In Malaysia, Plasmodium knowlesi is the most common cause of severe malaria, often associated with high parasitaemia. Asplenic patients may be at additional increased risk of intravascular haemolysis.

Case presentation: A 61 years old asplenic man was admitted to a tertiary referral hospital in Sabah, Malaysia, with severe knowlesi malaria characterized by hyperparasitaemia (7.9 %), jaundice, respiratory distress, metabolic acidosis, and acute kidney injury. He was commenced on intravenous artesunate, but1 day later developed haemoglobinuria, associated with a 22 % reduction in admission haemoglobin. Additional investigations, including a cell-free haemoglobin of 10.2 × 105 ng/mL and an undetectable haptoglobin, confirmed intravascular haemolysis. The patient continued on intravenous artesunate for a total of 48 h prior to substitution with artemether–lumefantrine, and made a good recovery with resolution of his haemoglobinuria and improvement of his kidney function by day 3.

Conclusions: An asplenic patient with hyperparasitaemic severe knowlesi malaria developed haemoglobinuria after treatment with intravenous artesunate. There are plausible mechanisms for increased haemolysis with hyperparasitaemia, and following both splenectomy and artesunate. Although in this case the patient made a rapid recovery, knowlesi malaria patients with this unusual complication should be closely monitored for potential deterioration.

No MeSH data available.


Urine sample on day 1, after completion of two doses of intravenous artesunate
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Fig1: Urine sample on day 1, after completion of two doses of intravenous artesunate

Mentions: On examination his temperature was 38.9 °C, heart rate 93 beats per minute, blood pressure 114/79 mm Hg, respiratory rate 36 breaths/minute and oxygen saturation 88 % on room air. He was notably jaundiced and had a scar on his abdomen, but examination was otherwise unremarkable. His urine was of normal colour. Blood film was reported as P. knowlesi, with a parasite count of 7.9 %. His haemoglobin was 15.2 g/dL, white cell count 8.7 × 103/μL, platelets 24 × 103/μL, and creatinine 145 µmol/L (Table 1). He was commenced on intravenous artesunate in addition to ceftriaxone, and transferred to a tertiary referral hospital. An arterial blood gas taken the following morning on 35 % oxygen via a Venturi mask revealed metabolic acidosis with a pH of 7.31 and bicarbonate of 11 mmol/L. His chest X-ray was unremarkable. One day later, after two doses of intravenous artesunate given on admission and at 12 h, he was noted to have “coca-cola” coloured urine (Fig. 1), with urinalysis positive for haemoglobin with no intact red blood cells. Additional blood investigations revealed a bilirubin of 181 µmol/L and elevated liver transaminases (Table 1). Glucose-6 phosphate dehydrogenase (G6PD) activity was normal, thalassemia screen was negative, and dengue NS1 antigen was negative. Testing for leptospirosis was not performed. The patient received two further doses of artesunate (at 24 and 48 h) before changing to artemether–lumefantrine. He made a good clinical recovery, with improvement of his oxygen saturation, jaundice, thrombocytopaenia, and renal function (creatinine 86 µmol/L on day 3; Table 1). By day 3 he was afebrile with no malaria parasites seen on blood film, and his haemoglobinuria had largely resolved. PCR confirmed P. knowlesi mono-infection. No pathogens were isolated from blood cultures taken after commencement of antibiotics. The patient received ceftriaxone for a total of 7 days.Table 1


Intravascular haemolysis with haemoglobinuria in a splenectomized patient with severe Plasmodium knowlesi malaria
Urine sample on day 1, after completion of two doses of intravenous artesunate
© Copyright Policy - OpenAccess
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC5017000&req=5

Fig1: Urine sample on day 1, after completion of two doses of intravenous artesunate
Mentions: On examination his temperature was 38.9 °C, heart rate 93 beats per minute, blood pressure 114/79 mm Hg, respiratory rate 36 breaths/minute and oxygen saturation 88 % on room air. He was notably jaundiced and had a scar on his abdomen, but examination was otherwise unremarkable. His urine was of normal colour. Blood film was reported as P. knowlesi, with a parasite count of 7.9 %. His haemoglobin was 15.2 g/dL, white cell count 8.7 × 103/μL, platelets 24 × 103/μL, and creatinine 145 µmol/L (Table 1). He was commenced on intravenous artesunate in addition to ceftriaxone, and transferred to a tertiary referral hospital. An arterial blood gas taken the following morning on 35 % oxygen via a Venturi mask revealed metabolic acidosis with a pH of 7.31 and bicarbonate of 11 mmol/L. His chest X-ray was unremarkable. One day later, after two doses of intravenous artesunate given on admission and at 12 h, he was noted to have “coca-cola” coloured urine (Fig. 1), with urinalysis positive for haemoglobin with no intact red blood cells. Additional blood investigations revealed a bilirubin of 181 µmol/L and elevated liver transaminases (Table 1). Glucose-6 phosphate dehydrogenase (G6PD) activity was normal, thalassemia screen was negative, and dengue NS1 antigen was negative. Testing for leptospirosis was not performed. The patient received two further doses of artesunate (at 24 and 48 h) before changing to artemether–lumefantrine. He made a good clinical recovery, with improvement of his oxygen saturation, jaundice, thrombocytopaenia, and renal function (creatinine 86 µmol/L on day 3; Table 1). By day 3 he was afebrile with no malaria parasites seen on blood film, and his haemoglobinuria had largely resolved. PCR confirmed P. knowlesi mono-infection. No pathogens were isolated from blood cultures taken after commencement of antibiotics. The patient received ceftriaxone for a total of 7 days.Table 1

View Article: PubMed Central - PubMed

ABSTRACT

Background: Haemoglobinuria is an uncommon complication of severe malaria, reflecting acute intravascular haemolysis and potentially leading to acute kidney injury. It can occur early in the course of infection as a consequence of a high parasite burden, or may occur following commencement of anti-malarial treatment. Treatment with quinine has been described as a risk factor; however the syndrome may also occur following treatment with intravenous artesunate. In Malaysia, Plasmodium knowlesi is the most common cause of severe malaria, often associated with high parasitaemia. Asplenic patients may be at additional increased risk of intravascular haemolysis.

Case presentation: A 61 years old asplenic man was admitted to a tertiary referral hospital in Sabah, Malaysia, with severe knowlesi malaria characterized by hyperparasitaemia (7.9 %), jaundice, respiratory distress, metabolic acidosis, and acute kidney injury. He was commenced on intravenous artesunate, but1 day later developed haemoglobinuria, associated with a 22 % reduction in admission haemoglobin. Additional investigations, including a cell-free haemoglobin of 10.2 × 105 ng/mL and an undetectable haptoglobin, confirmed intravascular haemolysis. The patient continued on intravenous artesunate for a total of 48 h prior to substitution with artemether–lumefantrine, and made a good recovery with resolution of his haemoglobinuria and improvement of his kidney function by day 3.

Conclusions: An asplenic patient with hyperparasitaemic severe knowlesi malaria developed haemoglobinuria after treatment with intravenous artesunate. There are plausible mechanisms for increased haemolysis with hyperparasitaemia, and following both splenectomy and artesunate. Although in this case the patient made a rapid recovery, knowlesi malaria patients with this unusual complication should be closely monitored for potential deterioration.

No MeSH data available.