Evidence map›Paper›PMID 33114276›Full record

ArticlePediatric reports2020

Adrenocorticotropic Hormone Therapy Improved Spasms and Sleep Disturbance in Smith-Magenis Syndrome: A Case Report.

Ken Momosaki, Jun Kido, Shirou Matsumoto, Shiro Ozasa, Kimitoshi Nakamura

Abstract readCase Reports
In one paragraph

Article in Pediatric reports, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed.

  1. Deletion ofJournal of genetics · 2025
    Article
  2. Hypothalamic-Pituitary-Adrenal Axis and Epilepsy.Journal of clinical neurology (Seoul, Korea) · 2024
    Review
  3. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

5 authors.

Ken MomosakiDepartment of Pediatrics, Graduate School of Medical Sciences, Kumamoto University, Kumamoto 860-8556, Japan.
Jun KidoDepartment of Pediatrics, Graduate School of Medical Sciences, Kumamoto University, Kumamoto 860-8556, Japan.ORCID 0000-0003-1328-6696
Shirou MatsumotoDepartment of Pediatrics, Graduate School of Medical Sciences, Kumamoto University, Kumamoto 860-8556, Japan.
Shiro OzasaDepartment of Pediatrics, Graduate School of Medical Sciences, Kumamoto University, Kumamoto 860-8556, Japan.
Kimitoshi NakamuraDepartment of Pediatrics, Graduate School of Medical Sciences, Kumamoto University, Kumamoto 860-8556, Japan.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

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.

Indexed as

adrenocorticotropic hormonecorticotropin-releasing hormoneinfantile spasmssleep disturbanceSmith–Magenis syndrome

Identifiers

PMID33114276
PMCPMC7717653

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