Pneumothorax revealing late recurrence of infantile myofibromatosis.

IF 2.1 4区 医学 Q3 CARDIAC & CARDIOVASCULAR SYSTEMS Annals of Thoracic Medicine Pub Date : 2024-10-01 Epub Date: 2024-10-03 DOI:10.4103/atm.atm_286_23
Clemence Yguel, Jean Michel Vignaud, Angelica Tiotiu
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Abstract

Adult recurrence of infantile myofibromatosis is exceptional. Here, we report the case of a 23-year-old woman with a late recurrence of infantile myofibromatosis revealed by spontaneous pneumothorax. The chest computed tomography scan found both cavitary and nodular bilateral pulmonary lesions. In infancy, she had multicentric myofibromatosis with digestive, cutaneous, and bone involvement, spontaneously regressive before the age of 15 months. Histological analysis of lung samples showed identical findings as from the cutaneous biopsy performed in infancy, confirming the recurrence of infantile myofibromatosis as pulmonary metastasis. New biopsy and long-term follow-up are highly recommended in the management of such cases.

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揭示婴儿肌纤维瘤病晚期复发的气胸。
婴儿肌纤维瘤病成年后复发的情况非常罕见。在此,我们报告了一例因自发性气胸而导致婴儿期肌纤维瘤病晚期复发的 23 岁女性病例。胸部计算机断层扫描发现双侧肺部均有空洞性和结节性病变。在婴儿时期,她曾患有多中心肌纤维瘤病,并伴有消化道、皮肤和骨骼受累,在15个月大前自发消退。肺部样本的组织学分析结果与婴儿期的皮肤活检结果相同,证实婴儿肌纤维瘤病复发为肺转移。在治疗此类病例时,强烈建议进行新的活组织检查和长期随访。
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来源期刊
Annals of Thoracic Medicine
Annals of Thoracic Medicine CARDIAC & CARDIOVASCULAR SYSTEMS-RESPIRATORY SYSTEM
CiteScore
4.10
自引率
4.30%
发文量
19
审稿时长
>12 weeks
期刊介绍: The journal will cover studies related to multidisciplinary specialties of chest medicine, such as adult and pediatrics pulmonology, thoracic surgery, critical care medicine, respiratory care, transplantation, sleep medicine, related basic medical sciences, and more. The journal also features basic science, special reports, case reports, board review , and more. Editorials and communications to the editor that explore controversial issues and encourage further discussion by physicians dealing with chest medicine.
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