Evidence map›Paper›PMID 41112086›Full record

ArticleCase reports in anesthesiology2025

Anesthetic Management of Progressive Deformity of Tracheal Cartilaginous Sleeve in a Pediatric Patient With Beare-Stevenson Syndrome: A Case Report.

Yumi Doi, Atsuko Harada, Jiro Tsugawa, Nayuta Higa, Tatsuki Oyoshi, Ryosuke Hanaya

Abstract read
In one paragraph

Article in Case reports in anesthesiology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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

What it found

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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

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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

1 citing paper in PubMed.

  1. 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

6 authors.

Yumi DoiPediatric Perioperative Center, Takatsuki General Hospital, Takatsuki, Japan.ORCID https://orcid.org/0000-0001-9069-3186
Atsuko HaradaDepartment of Pediatric Neurosurgery, Takatsuki General Hospital, Takatsuki, Japan.ORCID https://orcid.org/0000-0001-6745-4013
Jiro TsugawaDepartment of Pediatric Surgery, Takatsuki General Hospital, Takatsuki, Japan.
Nayuta HigaDepartment of Neurosurgery, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan, kagoshima-u.ac.jp.
Tatsuki OyoshiDepartment of Neurosurgery, Ichikikushikino Medical Association Neurosurgery Center, Kagoshima, Japan.
Ryosuke HanayaDepartment of Neurosurgery, Graduate School of Medical and Dental Sciences, Kagoshima University, Kagoshima, Japan, kagoshima-u.ac.jp.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Beare-Stevenson syndrome is a rare fibroblast growth factor receptor 2-related disorder characterized by craniosynostosis, midface hypoplasia, cutis gyrata, and developmental delay, with upper airway obstruction being a critical concern in early infancy. Tracheal cartilaginous sleeve is a congenital anomaly associated with fibroblast growth factor receptor 2-related syndromes, which often necessitates tracheostomy because of the potential for significant airway complications. However, serial airway imaging in patients with Beare-Stevenson syndrome with tracheal cartilaginous sleeve has not yet been documented. Our pediatric patient with Beare-Stevenson syndrome underwent airway evaluations using a rigid bronchoscope under general anesthesia at 33 days, 62 days, and then at 3 years of age. The initial rigid bronchoscopy demonstrated tracheal cartilaginous sleeve, and tracheostomy was performed. At 3 years of age, rigid bronchoscopy revealed progressive tracheal cartilage deformity with inward protrusion. This pouch-like structural change greatly affected anesthesia management because it posed a risk for airway obstruction due to potential tube misplacement. The image resolution of a rigid bronchoscope is superior to that of a flexible bronchoscope, allowing for more precise assessment. Despite the absence of abnormalities on routine flexible bronchoscopy, rigid bronchoscopy provided critical insights into airway changes. Precise imaging allowed multidisciplinary planning, and it highlighted tube malposition as a cause of intraoperative respiratory failure. Tracheal cartilaginous sleeve is a life-threatening condition that may occur in children with Beare-Stevenson syndrome. This case demonstrated progressive tracheal deformity over time, increasing the risk of sudden airway obstruction and presenting significant anesthetic challenges. Our findings highlight the clinical importance of repeated rigid bronchoscopic evaluations, which provide essential information for safe airway management.

Indexed as

airway managementBeare–Stevenson cutis gyrata syndromebronchoscopiescraniosynostosistracheal cartilaginous sleeve

Identifiers

PMID41112086
PMCPMC12531426

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