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肺移植候选患者经皮肤活检确诊为朗格汉斯细胞组织细胞增多症的延迟诊断

Late Diagnosis of Langerhans Cell Histiocytosis by Skin Biopsy in a Lung Transplant Candidate Patient.

作者信息

Klein Francisco R, Klein Julia, Otalora Lozano Diego, Vigliano Carlos

机构信息

Critical Care Medicine, Favaloro University, Faculty of Medical Sciences, Buenos Aires, ARG.

Critical Care Medicine, Favaloro Foundation University Hospital, Buenos Aires, ARG.

出版信息

Cureus. 2024 Feb 29;16(2):e55226. doi: 10.7759/cureus.55226. eCollection 2024 Feb.

DOI:10.7759/cureus.55226
PMID:38558685
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10981517/
Abstract

We present the case of a lung transplant candidate under veno-venous membrane oxygenation assistance (VV ECMO) whose diagnosis of emphysema of undetermined etiology was redefined as Langerhans cell histiocytosis (LCH) due to a scalp skin biopsy performed years after the beginning of his respiratory symptoms. A 20-year-old patient started three years before his admission with progressive dyspnea leading to a diagnosis of bullous emphysema of undetermined cause, which evolved into respiratory failure and evaluation for bilateral lung transplant. Three years later, he developed bilateral pneumonia requiring mechanical ventilation. When refractory hypoxemia ensued, he had to be placed on VV ECMO. Under these conditions, he was transferred to our center and listed for a bilateral pulmonary transplantation. Forty-eight hours after admission, and due to intense polyuria, central diabetes insipidus was diagnosed. In this clinical context, the presence of cutaneous lesions on the scalp was reconsidered and biopsied under the presumption of possible LCH, with pathology analysis confirming the diagnosis. He continued to be assisted with VV ECMO for 66 more days as a bridge to transplantation, developing multi-organ failure and passing away before a donor organ was available. The diagnosis of LCH should be considered in any adult patient with bullous emphysema of undetermined cause. Given the possibility of early therapeutic interventions, the search for its clinical associations (e.g., diabetes insipidus and/or skin lesions) should be a systematic part of the etiologic workup. The availability of skin specimens to reach a diagnosis makes its thorough search an important part of the diagnostic approach.

摘要

我们报告了一例在静脉-静脉膜肺氧合辅助(VV ECMO)下等待肺移植的患者,其最初病因不明的肺气肿诊断在出现呼吸道症状数年之后,经头皮皮肤活检重新定义为朗格汉斯细胞组织细胞增多症(LCH)。一名20岁患者在入院前三年开始出现进行性呼吸困难,诊断为病因不明的大疱性肺气肿,病情进展为呼吸衰竭并接受双侧肺移植评估。三年后,他发生双侧肺炎,需要机械通气。当出现难治性低氧血症时,他不得不接受VV ECMO治疗。在此情况下,他被转至我们中心并被列入双侧肺移植名单。入院48小时后,由于严重多尿,诊断为中枢性尿崩症。在这种临床背景下,重新考虑了头皮上皮肤病变的情况,并在可能为LCH的推测下进行了活检,病理分析证实了诊断。作为移植过渡,他继续接受VV ECMO治疗66天,期间出现多器官功能衰竭,在获得供体器官之前死亡。对于任何病因不明的大疱性肺气肿成年患者,均应考虑LCH的诊断。鉴于早期进行治疗干预的可能性,寻找其临床关联(如尿崩症和/或皮肤病变)应成为病因检查的常规部分。获取皮肤标本以明确诊断使得全面检查皮肤成为诊断方法的重要组成部分。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/fb4273be4f4b/cureus-0016-00000055226-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/87755c430a77/cureus-0016-00000055226-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/dbd42783731a/cureus-0016-00000055226-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/205f31bc085d/cureus-0016-00000055226-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/fb4273be4f4b/cureus-0016-00000055226-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/87755c430a77/cureus-0016-00000055226-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/dbd42783731a/cureus-0016-00000055226-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/205f31bc085d/cureus-0016-00000055226-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26f4/10981517/fb4273be4f4b/cureus-0016-00000055226-i04.jpg

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Incidence, prevalence and survival in patients with Langerhans cell histiocytosis: A national registry study from England, 2013-2019.朗格汉斯细胞组织细胞增生症患者的发病率、患病率和生存率:2013-2019 年英格兰国家登记研究。
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