Duodenal stenosis is a type of congenital intestinal atresia syndrome. Most patients are diagnosed during the neonatal period based on recurrent vomiting, but some cases develop symptoms at an older age. We report a case with an unusual pathogenic mechanism: sudden ileus due to food impaction.A 15-month-old boy was admitted to our institution with a history of recurrent vomiting and diarrhea for 7 days and fever for 2 days. Abdominal distention was found upon physical examination but other congenital anomalies were not. He was initially treated for acute gastroenteritis, but vomiting resumed after starting oral ingestion. Congenital membranous duodenal stenosis and impaction of a shimeji mushroom into the narrow section of the duodenum was recognized upon endoscopy. Dilatation was successful and restenosis was not seen. Food impaction is a rare (but important) initial event in congenital duodenal stenosis, and this disorder should be included in the differential diagnosis, especially for infants/toddlers with recurrent vomiting that develops at the time of eating solid and more diverse types of food.
Background
Various antiepileptic drugs can potentially cause psychiatric side effects in patients with epilepsy, but the precise mechanism of these actions remains unknown. In recent years, the common polymorphism C677T in the 5,10-methylenetetrahydrofolate reductase (
MTHFR
) gene has attracted attention for its role in the onset of psychiatric diseases. MTHFR and several vitamins (as cofactors) are crucial for remethylation of homocysteine via folate and homocysteine metabolism. We report a case of a Japanese patient who presented with reversible schizophrenia-like symptoms during antiepileptic drug therapy.
Case presentation
Our patient had frontal lobe epilepsy and had been treated with several antiepileptic drugs since the age of 13 years. He developed auditory hallucinations and multiple personalities at 17 years of age, several months after the initiation of phenytoin and phenobarbital, despite these antiepileptic drugs being used within the therapeutic ranges. Genetic analysis revealed that he was homozygous for the C677T polymorphism of
MTHFR
. Hyperhomocysteinemia, hypomethionemia, and multiple vitamin deficiencies, including folate, riboflavin, and pyridoxal, were identified at the age of 23 years. Vitamin supplementation and alteration of the antiepileptic drugs improved his psychotic symptoms. Multiple vitamin deficiencies with homozygous
MTHFR
C677T should be considered in patients presenting with schizophrenia-like symptoms during antiepileptic drug therapy.
Conclusions
To the best of our knowledge, this is the first report of antiepileptic drug-induced psychosis associated with homozygous C677T and multiple vitamin deficiencies. Our findings will contribute to the elucidation of the pathogenesis of the psychiatric side effects of antiepileptic drugs and lead to improved medical management for patients with epilepsy.
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