Literature DB >> 31816104

Nocturnal enteral nutrition is therapeutic for growth failure in Fanconi-Bickel syndrome.

Alessandra Pennisi1,2,3, Bruno Maranda1,4, Jean-François Benoist1, Véronique Baudouin5, Odile Rigal1, Samia Pichard1, René Santer6, Francesca Romana Lepri7, Antonio Novelli7, Hélène Ogier de Baulny1, Carlo Dionisi-Vici2, Manuel Schiff1,8,9.   

Abstract

Fanconi-Bickel syndrome (FBS) is a rare autosomal recessive disorder characterised by impaired glucose liver homeostasis and proximal renal tubular dysfunction. It is caused by pathogenic variants in SLC2A2 coding for the glucose transporter GLUT2. Main clinical features include hepatomegaly, fasting hypoglycaemia, postprandial hyperglycaemia, Fanconi-type tubulopathy occasionally with rickets, and a severe growth disorder. While treatment for renal tubular dysfunction is well established, data regarding optimal nutritional therapy are scarce. Similarly, detailed clinical evaluation of treated FBS patients is lacking. These unmet needs were an incentive to conduct the present pilot study. We present clinical findings, laboratory parameters and molecular genetic data on 11 FBS patients with emphasis on clinical outcome under various nutritional interventions. At diagnosis, the patients' phenotypic severity could be classified into two categories: a first group with severe growth failure and rickets, and a second group with milder signs and symptoms. Three patients were diagnosed early and treated because of family history. All patients exhibited massive glucosuria at diagnosis and some in both groups had fasting hypoglycaemic episodes. Growth retardation improved drastically in all five patients treated by intensive nutritional intervention (nocturnal enteral nutrition) and uncooked cornstarch with final growth parameters in the normal range. The four severely affected patients who were treated with uncooked cornstarch alone did not catch up growth. All patients received electrolytes and l-carnitine supplementation to compensate for the tubulopathy. This is one of the largest series of FBS on therapeutic management with evidence that nocturnal enteral nutrition rescues growth failure.
© 2019 SSIEM.

Entities:  

Keywords:  Fanconi-Bickel syndrome; glucose transporter-2; glycogen storage disease type XI; nocturnal enteral nutrition; proximal renal tubular dysfunction

Year:  2020        PMID: 31816104     DOI: 10.1002/jimd.12203

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  3 in total

1.  Case Report: Fanconi-Bickel Syndrome in a Chinese Girl With Diabetes and Severe Hypokalemia.

Authors:  Hongbo Chen; Juan-Juan Lyu; Zhuo Huang; Xiao-Mei Sun; Ying Liu; Chuan-Jie Yuan; Li Ye; Dan Yu; Jin Wu
Journal:  Front Pediatr       Date:  2022-06-09       Impact factor: 3.569

2.  Fanconi-Bickel syndrome in an infant with cytomegalovirus infection: A case report and review of the literature.

Authors:  Li-Jing Xiong; Mao-Ling Jiang; Li-Na Du; Lan Yuan; Xiao-Li Xie
Journal:  World J Clin Cases       Date:  2020-11-06       Impact factor: 1.337

Review 3.  Glycogen storage diseases with liver involvement: a literature review of GSD type 0, IV, VI, IX and XI.

Authors:  Miriam Massese; Francesco Tagliaferri; Carlo Dionisi-Vici; Arianna Maiorana
Journal:  Orphanet J Rare Dis       Date:  2022-06-20       Impact factor: 4.303

  3 in total

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