Extracorporeal membrane oxygenation: Bridging therapy in paediatric pulmonary Langerhans cell histiocytosis

Bhatt, Reena; Semple, Thomas; Slater, Olga; Nicholson, Andrew G.; Casanueva, Lidia; Desai, Ajay; Hoschtitzky, Andreas; Milne, Paul; Langley, Ross

doi:10.1111/jpc.15712

Cited by 1 publication

(1 citation statement)

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“…The recommendations of the International Expert Consensus for the diagnosis and treatment of adult LCH recommend treatment with limited courses of chemotherapy [4]. There are two case reports of the use of ECMO in patients with multisystem LCH with pulmonary involvement (in one as a bridge to recovery [13] and in another as a bridge to lung transplantation [14], the former being diagnosed during ECMO as described in the case of our report). Both received chemotherapy treatment with favorable results.…”

Section: Discussionmentioning

confidence: 67%

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

Klein,

Otalora Lozano

et al. 2024

Cureus

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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.

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Section: Discussionmentioning

confidence: 67%