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01 October 2020 : Case report  China (mainland)

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 Fang2ABC

DOI: 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.

CaseAge (yr/d)SexCountryYearUnderlying diseaseTPNMRI findingThiamin levelThiamin treatmentOutcome
Bian et al. []11 75 yrFChina2016SBS secondary to gastrectomyPN dependent without thiaminHigh T2 intensity in both mammillary bodies and bilateral thalamusUndetectableNAImprovement within 10 d and complete resolution in 29 d
Santarpia et al. []10 20 yrFItaly2009SBS secondary to gastrectomyTPN dependent without thiaminBilateral and symmetrical hyper-intensities in the subthalamusUndetectable100 mg IV dailyImprovement within 15 d and complete resolution in 40 d
Benidir et al. []8 8 yrFCanada2014SBS secondary to gastroschisisTPN dependent without thiamin supplementation for 3 monthsSymmetric lesions of the basal ganglia, mammillary bodies and peri-aqueductal region of the midbrainUndetectable100 mg IV dailyComplete resolution within 48 h
Roilides et al. []9 3 yrMGreece2019SBS secondary to NEC gastrectomy for double volvulusPN dependent with 3.1 mg thiaminBilateral symmetric hyper-intense signals in the peri-aqueductal area and both medial thalami10 μg/L (normal levels, 28–85)25 mg IM dailyImprovement within 48 hr and complete resolution in 96 hr
Present case3 dMChina2019SBS secondary to gastroschisisPN dependent with 3.0 mg thiaminSymmetric lesions of the basal ganglia, caudate nucleus head, thalamus medial margin symmetry19.7 nmol/L (normal levels, 41.5–108.9 nmol/L)100 mg IV dailyImprovement 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.

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923