Yang Guozhu,Su Yi,He Chaosheng,et al.Hereditary multiple exostosis with hemopneumothorax:a report of two case with a literature review[J].Journal of Clinical Pediatric Surgery,,():776-780.[doi:10.3760/cma.j.cn101785-202404038-013]
Hereditary multiple exostosis with hemopneumothorax:a report of two case with a literature review
- Keywords:
- Osteochondromatosis; Exostoses; Multiple Hereditary; Pneumothorax; Hemothorax; Thoracic Surgical Procedures; Treatment Outcome; Child
- Abstract:
- Objective To summarize the clinical features,diagnostics and treatments of hereditary multiple exostosis (HME) with hemopneumothorax in children. Methods From December 2022 to April 2023,the relevant clinical data were reviewed for 2 HME children with hemopneumothorax admitted into First Affiliated Hospital of Xiamen University.A systematic review of literature was performed through searching the databases of PubMed,Medline,Web of Science,China National Knowledge Infrastructure (CNKI) and Wanfang from January 1983 to January 2023.Clinical manifestations,diagnoses,treatments and outcomes were analyzed. Results Two children were discharged after thoracoscopy and had an excellent recovery during follow-ups.A total of 16 HME children with pneumothorax/hemothorax were reported.The complications were pneumothorax (n=4) and hemothorax (n=12).The affected side was left (n=6) and right (n=9).One case of bilateral pneumothorax was reported.There were 13 boys and 3 girls with an average age of 11.5(3-17) year.There were varying degrees of chest distress,shortness of breath,dyspnea or other clinical manifestations.Two cases were managed by non-surgical measures such as catheter drainage observation and the remainders operated,including thoracoscopy (n=10) and thoracotomy (n=4).All of them had a decent prognosis after treatment and postoperative pathological examination hinted at exostosis without malignant changes. Conclusions HME in children may lead to hemopneumothorax with major manifestations of anemia,chest tightness and dyspnea.A definite diagnosis is made through three-dimensional computed tomography (CT) scan.Thoracoscopic resection of rib lesion prevents the risk of thoracic organ injury and hemorrhage in early stage,As a simple and effective option,it helps to achieve a rapid recovery.
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Memo
收稿日期:2024-4-15。
基金项目:福建省自然科学基金(2019J01564)
通讯作者:李索林,Email:lisuolin@263.net