Adrenocorticotropic Hormone Therapy Improved Spasms and Sleep Disturbance in Smith–Magenis Syndrome: A Case Report
| Autor: | Shiro Ozasa, Jun Kido, Kimitoshi Nakamura, Shirou Matsumoto, Ken Momosaki |
|---|---|
| Rok vydání: | 2020 |
| Předmět: |
Pediatrics
medicine.medical_specialty adrenocorticotropic hormone Case Report Adrenocorticotropic hormone corticotropin-releasing hormone Electroencephalography 03 medical and health sciences Corticotropin-releasing hormone 0302 clinical medicine Medicine Ictal Craniofacial 030304 developmental biology 0303 health sciences Sleep disorder medicine.diagnostic_test business.industry sleep disturbance medicine.disease Smith–Magenis syndrome Sleep in non-human animals business 030217 neurology & neurosurgery infantile spasms |
| Zdroj: | Pediatric Reports |
| ISSN: | 2036-7503 |
| Popis: | Smith–Magenis syndrome (SMS) is a complex disorder characterized by variable mental retardation, sleep disturbances, craniofacial and skeletal anomalies, self-injurious and attention-seeking behaviors, and speech and motor delays. The case of a 14-month-old girl with SMS who was experiencing spasm clusters and sleep disturbances with sleep–wake intervals of 1.5 to 2 h persisting from the neonatal period was examined. The patient’s spasms stopped and interictal electroencephalography did not show epileptic discharges after undergoing a high-dose adrenocorticotropic hormone (ACTH) therapy. Moreover, the patient’s sleep cycle stabilized 1 month after receiving the ACTH therapy. Dramatic reductions in the patient’s self-injurious behaviors were also noted. At 1 year following ACTH treatment, the patient’s improved sleep was maintained. High-dose ACTH treatment was considered to contribute to the normal adaptation of the hypothalamic–pituitary–adrenal axis by regulating the release of corticotropin-releasing hormone, resulting in improvement of the patient’s infantile spasms and sleep disturbances. |
| Databáze: | OpenAIRE |
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