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1.
Medicine (Baltimore) ; 103(7): e36782, 2024 Feb 16.
Article in English | MEDLINE | ID: mdl-38363934

ABSTRACT

RATIONALE: Although patients with central disorders of hypersomnolence (CDH) exhibit characteristic symptoms of hypersomnia frequently, it takes 5 to 15 years from the onset for its diagnosis due to the lack of symptom recognition. Here, we present a case of idiopathic hypersomnia (IH), a CDH, wherein early diagnosis was aided by a video footage of a spontaneous sleep attack. PATIENT CONCERNS: A 21-year-old man lost consciousness while driving and experienced an accident. He had complained of excessive daytime sleepiness (EDS) over half a year. During his hospitalization for close monitoring of the loss of consciousness, an in-room surveillance camera captured a 14-minutes long spontaneous sleep attack, during which he experienced general muscle weakness and loss of consciousness without warnings or convulsions leading to a fall from the bed. There were no abnormalities in vital signs. DIAGNOSES: There was no significant cataplexy and less than 2 sleep-onset rapid eye movements (SOREM) in 2 sleep latency tests, with a mean sleep latency of 2.1 and 4.6 minutes. Other sleep deprivation syndromes were excluded from differential diagnosis and finally, a diagnosis of IH was confirmed according to the criteria of the Third Edition of the International Classification of Sleep Disorders. During the course of the disease, attention-deficit/hyperactive disorder (ADHD) and a gaming disorder also diagnosed. INTERVENTIONS: Pharmacological treatment with modafinil was administered for IH and methylphenidate for ADHD. Cognitive behavioral therapy was performed for the gaming disorder. OUTCOMES: The EDS improved, and sleep attacks were no longer observed. The disruption of daily life caused by the gaming disorder was also reduced. LESSONS: Video recordings of sleep attacks are beneficial for identifying the cause of loss of consciousness. Home video recordings may be helpful in the early diagnosis of IH.


Subject(s)
Disorders of Excessive Somnolence , Idiopathic Hypersomnia , Humans , Male , Young Adult , Disorders of Excessive Somnolence/diagnosis , Disorders of Excessive Somnolence/etiology , Disorders of Excessive Somnolence/drug therapy , Idiopathic Hypersomnia/diagnosis , Idiopathic Hypersomnia/drug therapy , Modafinil/therapeutic use , Sleep/physiology , Unconsciousness
2.
Nihon Ronen Igakkai Zasshi ; 45(5): 511-9, 2008 Sep.
Article in Japanese | MEDLINE | ID: mdl-19057104

ABSTRACT

AIM: Purple urine bag syndrome is a condition in which the urinary catheter bag turns purple. A tryptophan-indigo hypothesis has been proposed as the mechanism of PUBS, in which bacterial decomposition of tryptophan in gut associated with chronic constipation, bacterial overgrowth in the urinary tract and alkaline urine causes production of indigo and discoloration. We considered that further investigation of cases was needed. METHODS: We investigated 6 cases exhibiting PUBS (3 males and 3 females). RESULTS: All cases had chronic constipation. Oral ingestion was impossible in one case. PUBS disappeared after antibiotic treatment (3 cases) or spontaneously (one case). Alkaline urine and indicanuria were not found in all cases that showed the disappearance of PUBS. In bacterial culture of urine during the exhibition of PUBS, Enterococcus faecalis was isolated together with Morganella morganii (3 cases) and Pseudomonas aeruginosa (one case). Single infections by Klebsiella pneumoniae or Citrobacter species were also found. After disappearance of PUBS, infected bacterial species changed but no cases showed sterile urine. Urine and blood alpha-amino-n-butyric acid levels reduced after the disappearance of PUBS whereas tryptophan levels did not show related changes. In one case, blood protein concentration increased after the spontaneous disappearance of PUBS. Indicanuria and alkalization of urine from urinary catheter bag were more intense than of fresh urine. CONCLUSIONS: The present results generally support the 'Tryptophan-indigo hypothesis'. Furthermore, it was suggested that additional factors associated with the occurrence of PUBS are an environment that facilitates specific bacterial growth in a hospital as well as abnormal metabolism relating to alpha-amino-n-butyric acid and reduced protein synthesis in patients.


Subject(s)
Urine/chemistry , Urine/microbiology , Aged , Aged, 80 and over , Catheters, Indwelling , Color , Dementia , Enterococcus faecalis/isolation & purification , Female , Humans , Indigo Carmine , Indoles/urine , Male , Tryptophan/urine
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