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Tominaga T, Sato T, Ichihashi Y, et al. Transient Fanconi syndrome in two preterm infants with hydronephrosis and urinary tract infection. CEN Case Rep. 2017;6(1):88-90doi: 10.1007/s13730-017-0250-9.
Tominaga, T., Sato, T., Ichihashi, Y., Amano, N., Kobayashi, Y., & Awazu, M. (2017). Transient Fanconi syndrome in two preterm infants with hydronephrosis and urinary tract infection. CEN case reports, 6(1), 88-90. https://doi.org/10.1007/s13730-017-0250-9
Tominaga, Takahiro, et al. "Transient Fanconi syndrome in two preterm infants with hydronephrosis and urinary tract infection." CEN case reports vol. 6,1 (2017): 88-90. doi: https://doi.org/10.1007/s13730-017-0250-9
Tominaga T, Sato T, Ichihashi Y, Amano N, Kobayashi Y, Awazu M. Transient Fanconi syndrome in two preterm infants with hydronephrosis and urinary tract infection. CEN Case Rep. 2017 May;6(1):88-90. doi: 10.1007/s13730-017-0250-9. Epub 2017 Feb 16. PMID: 28509132; PMCID: PMC5438814.
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CEN Case Rep. 2017 May;6(1):88-90. doi: 10.1007/s13730-017-0250-9. Epub 2017 Feb 16.

Transient Fanconi syndrome in two preterm infants with hydronephrosis and urinary tract infection.

CEN case reports

Takahiro Tominaga, Takeshi Sato, Yosuke Ichihashi, Naoko Amano, Yasuaki Kobayashi, Midori Awazu

Affiliations

  1. Department of Pediatrics, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan.
  2. Department of Pediatrics, Ashikaga Red Cross Hospital, Tochigi, Japan.
  3. Department of Pediatrics, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan. [email protected].

PMID: 28509132 PMCID: PMC5438814 DOI: 10.1007/s13730-017-0250-9

Abstract

Type IV renal tubular acidosis is known to occur in obstructive uropathy with urinary tract infection. Fanconi syndrome, however, has not been described in these settings. We report two preterm infants who developed Fanconi syndrome associated with hydronephrosis and urinary tract infection. Patient 1 is a boy with 21 trisomy, bilateral renal hypoplasia and bilateral vesicoureteral reflux delivered at 35 weeks' gestation. At postnatal day 42, he developed Fanconi syndrome after urinary tract infection, which persisted until the surgical correction of vesicoureteral reflux. Patient 2 was delivered at 35 weeks' gestation. At postnatal day 9, he was admitted for severe dehydration. He had phimosis and ultrasonography showed left pelviectasis. Laboratory data were compatible with Fanconi syndrome, which resolved spontaneously after fluid therapy. Subsequently urine culture grew bacteria and treatment for infection and topical corticosteroid for phimosis were performed. DMSA scintigraphy performed later showed left renal scar. Tubular cell stretch, due to vesicoureteral reflux in Patient 1 and phimosis in Patient 2, and urinary tract infection in association with immaturity of tubules are thought to have caused Fanconi syndrome.

Keywords: Cell stretch; Immaturity; Proximal tubule; Renal tubular acidosis; Urinary tract obstruction

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