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Pediatrics International (2014) 56, e68–e71
doi: 10.1111/ped.12412
Patient Report
Transient renal dysfunction with reversible splenial lesion Toru Watanabe, Tomoka Matsuda, Ryoichi Kitagata, Iwao Tajima, Hiroyuki Ono, Keiko Hirano, Masami Shirai, Akira Endoh and Teruaki Hongo Department of Pediatrics, Iwata City Hospital, Iwata, Japan Abstract
We report the case of a 6-month-old boy with transient renal dysfunction who had an intensified signal in the splenium of the corpus callosum on magnetic resonance imaging. He presented to hospital with fever and sudden disturbance of consciousness. Cerebrospinal fluid analysis did not show pleocytosis. The mild consciousness disturbance disappeared after 30 min, but the splenial signal persisted even after 8 days. Further, renal glucosuria, increased excretion of select amino acids, and abnormal fractional excretion of electrolytes were observed, indicating renal tubular dysfunction. The abnormal urinary findings spontaneously resolved by day 9 of hospitalization. The splenial lesion took 21 days to normalize. There were no signs of neurological complications 2 months later. This case suggests the possibility of renal involvement in splenial lesions.
Key words apparent life-threatening event, corpus callosum, central nervous system symptom, Fanconi syndrome, mild encephalitis/encephalopathy with reversible splenial lesion.
Reversible splenial lesions are often found in acute encephalopathy, which is considered clinically mild encephalitis/ encephalopathy with a reversible splenial lesion (MERS).1,2 We encountered the case of an infant boy with a reversible splenial lesion and renal dysfunction similar to that observed in transient Fanconi syndrome, which is often drug induced.3 The patient had transient brain, renal, and hepatic damage. Here, we present the pathophysiology of his condition and review the literature. To our knowledge, this is the first report of a case of this kind without encephalitis.
Case report A 6-month-old boy was admitted to hospital for disturbance of consciousness. His medical history was unremarkable. He had not received any medication, and his development had been normal, but his birthweight had been low, at 2410 g. Review of family history showed that his grandfather had epilepsy. On the day of admission, the infant was pale with lip cyanosis and profuse sweating in the prone position; he did not respond to stimulation by his mother, showing disturbance of consciousness and weak muscle tone. We suspected that he had apparent lifethreatening events (ALTE). On examination in the emergency department, heart rate was 193 beats/min with a regular beat; blood pressure, 80/51 mmHg; respiratory rate, 32/min; SpO2, 98% (room air); body temperature, 39.2°C; and Glasgow coma scale, E4V5M6. There was no neck stiffness or any remarkable Correspondence: Toru Watanabe, Department of Pediatrics, Iwata City Hospital, 512-3, Iwata City, Shizuoka 438-8550, Japan. Email: [email protected] Received 2 October 2013; revised 24 March 2014; accepted 12 May 2014.
© 2014 Japan Pediatric Society
chest and abdominal findings. The pupils were 3/3 mm with normal light reflexes and tendon reflexes. Complete blood count was unremarkable, but laboratory findings showed elevated liver enzymes, renal dysfunction, hyperglycemia, and coagulation disorder (Table 1). Venous blood gas analysis showed metabolic acidosis (Table 1). On urinalysis, alkalinuria, glucosuria, proteinuria and β2-microglobulin (β2-MG) 15 448 μg/L were noted. Urinary tract infection was not suspected because no leukocyturia or inflammation was noted. Interleukin (IL)-6 was 43.6 pg/mL (normal,
Pediatrics International (2014) 56, e68–e71
doi: 10.1111/ped.12412
Patient Report
Transient renal dysfunction with reversible splenial lesion Toru Watanabe, Tomoka Matsuda, Ryoichi Kitagata, Iwao Tajima, Hiroyuki Ono, Keiko Hirano, Masami Shirai, Akira Endoh and Teruaki Hongo Department of Pediatrics, Iwata City Hospital, Iwata, Japan Abstract
We report the case of a 6-month-old boy with transient renal dysfunction who had an intensified signal in the splenium of the corpus callosum on magnetic resonance imaging. He presented to hospital with fever and sudden disturbance of consciousness. Cerebrospinal fluid analysis did not show pleocytosis. The mild consciousness disturbance disappeared after 30 min, but the splenial signal persisted even after 8 days. Further, renal glucosuria, increased excretion of select amino acids, and abnormal fractional excretion of electrolytes were observed, indicating renal tubular dysfunction. The abnormal urinary findings spontaneously resolved by day 9 of hospitalization. The splenial lesion took 21 days to normalize. There were no signs of neurological complications 2 months later. This case suggests the possibility of renal involvement in splenial lesions.
Key words apparent life-threatening event, corpus callosum, central nervous system symptom, Fanconi syndrome, mild encephalitis/encephalopathy with reversible splenial lesion.
Reversible splenial lesions are often found in acute encephalopathy, which is considered clinically mild encephalitis/ encephalopathy with a reversible splenial lesion (MERS).1,2 We encountered the case of an infant boy with a reversible splenial lesion and renal dysfunction similar to that observed in transient Fanconi syndrome, which is often drug induced.3 The patient had transient brain, renal, and hepatic damage. Here, we present the pathophysiology of his condition and review the literature. To our knowledge, this is the first report of a case of this kind without encephalitis.
Case report A 6-month-old boy was admitted to hospital for disturbance of consciousness. His medical history was unremarkable. He had not received any medication, and his development had been normal, but his birthweight had been low, at 2410 g. Review of family history showed that his grandfather had epilepsy. On the day of admission, the infant was pale with lip cyanosis and profuse sweating in the prone position; he did not respond to stimulation by his mother, showing disturbance of consciousness and weak muscle tone. We suspected that he had apparent lifethreatening events (ALTE). On examination in the emergency department, heart rate was 193 beats/min with a regular beat; blood pressure, 80/51 mmHg; respiratory rate, 32/min; SpO2, 98% (room air); body temperature, 39.2°C; and Glasgow coma scale, E4V5M6. There was no neck stiffness or any remarkable Correspondence: Toru Watanabe, Department of Pediatrics, Iwata City Hospital, 512-3, Iwata City, Shizuoka 438-8550, Japan. Email: [email protected] Received 2 October 2013; revised 24 March 2014; accepted 12 May 2014.
© 2014 Japan Pediatric Society
chest and abdominal findings. The pupils were 3/3 mm with normal light reflexes and tendon reflexes. Complete blood count was unremarkable, but laboratory findings showed elevated liver enzymes, renal dysfunction, hyperglycemia, and coagulation disorder (Table 1). Venous blood gas analysis showed metabolic acidosis (Table 1). On urinalysis, alkalinuria, glucosuria, proteinuria and β2-microglobulin (β2-MG) 15 448 μg/L were noted. Urinary tract infection was not suspected because no leukocyturia or inflammation was noted. Interleukin (IL)-6 was 43.6 pg/mL (normal,
