01 October 2020
: Case report 
Short Bowel Syndrome in an Extremely Low Birth Weight Premature Infant with Wernicke Encephalopathy: A Case Report
Unusual clinical course, Challenging differential diagnosis, Diagnostic / therapeutic accidents, Management of emergency care, Rare disease, Adverse events of drug therapy, Educational Purpose (only if useful for a systematic review or synthesis)
Yanzhen Zhang1ABCFG, Bo Zhou1AF*, Lanyan Wu1AD, Hong Cao1CE, Guozhong Xie2ABG, Hua Fang2ABCDOI: 10.12659/AJCR.924830
Am J Case Rep 2020; 21:e924830
Table 1. Summary of cases with Wernicke’s encephalopathy with short bowel syndrome on total parenteral nutrition.
| Case | Age (yr/d) | Sex | Country | Year | Underlying disease | TPN | MRI finding | Thiamin level | Thiamin treatment | Outcome |
|---|---|---|---|---|---|---|---|---|---|---|
| Bian et al. []11 | 75 yr | F | China | 2016 | SBS secondary to gastrectomy | PN dependent without thiamin | High T2 intensity in both mammillary bodies and bilateral thalamus | Undetectable | NA | Improvement within 10 d and complete resolution in 29 d |
| Santarpia et al. []10 | 20 yr | F | Italy | 2009 | SBS secondary to gastrectomy | TPN dependent without thiamin | Bilateral and symmetrical hyper-intensities in the subthalamus | Undetectable | 100 mg IV daily | Improvement within 15 d and complete resolution in 40 d |
| Benidir et al. []8 | 8 yr | F | Canada | 2014 | SBS secondary to gastroschisis | TPN dependent without thiamin supplementation for 3 months | Symmetric lesions of the basal ganglia, mammillary bodies and peri-aqueductal region of the midbrain | Undetectable | 100 mg IV daily | Complete resolution within 48 h |
| Roilides et al. []9 | 3 yr | M | Greece | 2019 | SBS secondary to NEC gastrectomy for double volvulus | PN dependent with 3.1 mg thiamin | Bilateral symmetric hyper-intense signals in the peri-aqueductal area and both medial thalami | 10 μg/L (normal levels, 28–85) | 25 mg IM daily | Improvement within 48 hr and complete resolution in 96 hr |
| Present case | 3 d | M | China | 2019 | SBS secondary to gastroschisis | PN dependent with 3.0 mg thiamin | Symmetric lesions of the basal ganglia, caudate nucleus head, thalamus medial margin symmetry | 19.7 nmol/L (normal levels, 41.5–108.9 nmol/L) | 100 mg IV daily | Improvement within 72 hr and complete resolution in 96 hr |
| F – Female; IM – intramuscular; IV – intravenous; M – Male; MRI – magnetic resonance imaging; NA – not applicable; NEC – necrotizing enterocolitis; PN – parenteral nutrition; SBS – short bowel syndrome; TPN – total parenteral nutrition. | ||||||||||