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| Content Provider | Springer Nature : BioMed Central |
|---|---|
| Author | Rossi, Alessandro Miele, Erasmo Fecarotta, Simona Veiga-da-Cunha, Maria Martinelli, Massimo Mollica, Carmine D’Armiento, Maria Mozzillo, Enza Strisciuglio, Pietro Derks, Terry G. J. Staiano, Annamaria Parenti, Giancarlo |
| Abstract | Background Besides major clinical/biochemical features, neutropenia and inflammatory bowel disease (IBD) constitute common complications of Glycogen storage disease type Ib (GSD Ib). However, their management is still challenging. Although previous reports have shown benefit of empagliflozin administration on neutropenia, no follow-up data on bowel (macro/microscopic) morphology are available. We herein present for the first time longitudinal assessment of bowel morphology in a GSD Ib child suffering from Crohn disease-like enterocolitis treated with empagliflozin. Case presentation A 14-year-old boy with GSD Ib and severe IBD was (off-label) treated with empagliflozin (20 mg/day) after informed oral and written consent was obtained from the patient’s parents. No adverse events were noted. Clinical symptoms and stool frequency improved within the first week of treatment. Pediatric Crohn disease activity index (PCDAI) normalised within the first month of treatment. Abdomen magnetic resonance imaging (MRI) performed 3 months after treatment initiation showed dramatic decrease in disease activity and length. Similar findings were reported on histology at 5.5 months. At 7.5 months hemoglobin levels normalised and fecal calprotectin almost normalised. Improved neutrophil count, metabolic control and quality of life were also noted. G-CSF dose was decreased by 33% and the patient was partly weaned from tube feeding. Conclusions This is the first report presenting extensive gastrointestinal morphology follow-up in a GSD Ib patient receiving empagliflozin. The present case suggests that empagliflozin can be safe and effective in inducing IBD remission in GSD Ib patients and can even postpone surgery. Future studies are required to confirm its effect over time and assess its benefit in various disease stages. The development of an international collaborating networks for systematic data collection is worthy. |
| Related Links | https://ijponline.biomedcentral.com/counter/pdf/10.1186/s13052-021-01100-w.pdf |
| Ending Page | 9 |
| Page Count | 9 |
| Starting Page | 1 |
| File Format | HTM / HTML |
| ISSN | 18247288 |
| DOI | 10.1186/s13052-021-01100-w |
| Journal | Italian Journal of Pediatrics |
| Issue Number | 1 |
| Volume Number | 47 |
| Language | English |
| Publisher | BioMed Central |
| Publisher Date | 2021-07-02 |
| Access Restriction | Open |
| Subject Keyword | Pediatrics Maternal and Child Health Glycogen storage disease type Ib Inflammatory bowel disease Neutropenia 1,5-anhydroglucitol Empagliflozin Continuous glucose monitoring |
| Content Type | Text |
| Resource Type | Case study |
| Subject | Pediatrics, Perinatology and Child Health |
| Journal Impact Factor | 3.2/2023 |
| 5-Year Journal Impact Factor | 3.2/2023 |
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