March hemoglobinuria progressed to acute kidney injury after kendo practice: a case report

March hemoglobinuria is a disease that presents with transient hemoglobinuria after strenuous exercise, such as a marathon, karate, and kendo. Kendo is a form of Japanese fencing characterized by repetitive stepping and is thought to result in intravascular hemolysis due to mechanical stress. Aspartate aminotransferase, creatine kinase, and lactate dehydrogenase (LDH) increased, serum haptoglobin decreased, and urinary occult blood was positive immediately after a stress test in kendo [5]. March hemoglobinuria developed not only with foot but also hand hemolysis, which was caused by playing the conga drum [6]. In the present case, hemolytic findings, such as an indirect predominant increase in bilirubin and an increase in LDH, were observed. If the urinary occult blood is positive but urinary red blood cells are not found, hemoglobinuria and myoglobinuria need to be differentiated, and paroxysmal nocturnal hemoglobinuria (PNH), rhabdomyolysis, and march hemoglobinuria should be considered.

When intravascular hemolysis persists, free hemoglobin released into the plasma by hemolysis strongly adsorbs nitric oxide, which has smooth muscle relaxation and platelet aggregation inhibition actions, resulting in vascular spasm and digestion. The function of vascular endothelial cells is often impaired, causing intravascular thrombosis [7]. In patients with PNH, 65% of the population had chronic kidney disease (CKD) and 21% were in CKD stages 3–5 [8]. It is believed that the mechanism of renal dysfunction caused by intravascular hemolysis is due to the deposition of progressive hemosiderin in the proximal tubules, which was not filtered because of its high molecular weight. Hemosiderin is thought to accumulate in the epithelium, causing tubular atrophy and interstitial fibrosis, leading to renal injury [9]. Case reports of acute kidney injury caused by march hemoglobinuria are rare because the volume of hemolyzed blood is small compared to PNH. Gilligan DR, et al. reported that the volume of hemolysis is only 6–40 mL in patients with march hemoglobinuria [10]. There were no abnormalities in urinary levels of NAG, β2MG, α1MG and L-FABP in present case (Table 1). Previous report also indicated no tubular dysfunction [11]. Thus, there are very few case reports of renal biopsy for march hemoglobinuria [11,12,13]. In the present case, hemosiderin deposition was observed in the proximal tubule, and iron granules were also taken up in lysosomes, as observed in electron microscopic findings, which led to a definitive diagnosis of march hemoglobinuria.

It is also noteworthy that the cause of march hemoglobinuria may be an abnormality in the erythrocyte membrane. Banga JP, et al. found absent low-molecular-weight protein bands of the erythrocyte membrane structure in three cases of march hemoglobinuria [14]. Although the mechanism that explained the defect causing hemolysis has not yet been elucidated, march hemoglobinuria may be induced when patients with erythrocyte membrane anomalies exercise. However, no abnormalities in the erythrocyte membrane were detected in present case. This patient did not show any abnormalities in numbers of erythrocyte as well as peripheral smear. Moreover, hyperbilirubinemia and occult blood in the urine were improved after resting and hydration. Thus, we did not measure G6PD in present case. Even in patients with no abnormalities in the erythrocyte membrane, exercise itself has the potential to cause changes in the erythrocyte fragility and impaired membrane structure, eventually promote to hemolysis [15]. Platt OS, et al. proved that dehydrated cells were more sensitive to shear stress and were turned to fragile [16]. It is suggested that severe dehydration during hard exercise enhanced intravascular hemolysis and tubular atrophy due to accumulation of hemosiderin, leading to acute kidney injuries.

It could be possible to prevent the recurrence of hemoglobinuria by wearing shoes [17], which would function to defend the feet from external stimuli. It was previously reported that hemoglobinuria after exercise caused by karate disappeared through the use of a sponge rubber cushion [18]. March hemoglobinuria is a hemolytic disease that occurs after intense exercise, and its prevalence is very rare due to a lack of a definitive diagnosis. However, it is beneficial for patients to receive lifestyle guidance via a definitive diagnosis.

In conclusion, this report serves as a reminder that march hemoglobinuria can occur in occult blood-positive cases after intense exercise.

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