Reporte de caso: hemoptisis masiva como manifestación clínica de un síndrome de Swyer-James-Mac Leod / Case report: massive hemoptysis as a clinical manifestation of Swyer-James-Mac Leod syndrome

El síndrome de Swyer-James-Mac Leod es una entidad poco frecuente adquirida en la infancia, generalmente tras una infección pulmonar moderada a grave de tipo bronquiolitis o neumonía, generalmente virales. Ocasionalmente se tiene el antecedente de infecciones repetidas de este tipo. Consiste en el desarrollo de enfisema hipoplásico pulmonar unilateral, que puede a veces relacionarse con bronquiectasias ipsilaterales o bilaterales, obstrucción fija al flujo aéreo y puede también asociarse a reducción del flujo sanguíneo del pulmón hipoplásico, de manera focal o difusa, con o sin tortuosidad de la vascularización proximal y a veces con una compensación del pulmón contralateral, en forma de sobredistensión e hiperflujo vascular relativo. Presentamos el caso de un varón de 79 años de edad con antecedentes de infecciones tipo bronquiolitis virales repetidas en la infancia, obstrucción fija grave al flujo aéreo y hemoptisis masiva secundaria a una infección por Pseudomonas aeruginosa sensible a la terapia antibiótica habitual.

Swyer-James-Mac Leod syndrome is a rare condition acquired in childhood, usually after a moderate to severe lung infection such as bronchiolitis or pneumonia, usually viral. Occasionally there is a history of repeated infections of this type. It consists of the development of unilateral pulmonary hypoplastic emphysema, which can sometimes be related to ipsilateral or bilateral bronchiectasis, fixed airflow obstruction, and may also be associated with reduced blood flow in the hypoplastic lung, with or without tortuosity of the proximal vascular supply and sometimes with compensation from the contralateral lung, in the form of overdistension and relative vascular hyperflow. We present the case of a 79-year-old man with a history of recurrent viral bronchiolitis-type infections in childhood, severe fixed airflow obstruction, and massive hemoptysis secondary to a Pseudomonas aeruginosa infection sensitive to usual antibiotic therapy.

Incidental Diagnosis of Swyer-James-MacLeod Syndrome in a Military Pilot.

BACKGROUND: Swyer-James-MacLeod syndrome (SJMS) is a specific form of bronchiolitis obliterans that occurs rarely, but represents recognized sequelae of common pediatric respiratory illness, and presents as unilateral hyperlucency on chest imaging. This case study describes such an incidental radiographic finding identified during the assessment of chest wall discomfort in a military pilot.CASE REPORT: A 35-yr-old military pilot presented to his flight surgeon with vague intermittent chest discomfort. Initial evaluation revealed an abnormal chest radiograph with unilateral hyperlucency and mild expiratory airflow limitation on pulmonary function testing. The evaluation also included computed tomography imaging with contrast infusion and echocardiography, though the presenting complaint had resolved. The airman was referred to our clinic for further evaluation and aeromedical recommendations regarding returning to flight duties. He was diagnosed with SJMS and recommended to be returned to flight duties.DISCUSSION: SJMS can be challenging to recognize to the untrained eye. An inflammatory response from viral or bacterial infection in childhood results in dysfunctional growth of the affected region of the lung, causing radiographic asymmetry. Although destruction of the alveoli and emphysema may occur, for most cases, there are minimal clinical sequelae. SJMS is not known to be progressive and is not associated with systemic conditions. The pilot likely had the abnormal chest radiograph at the time of commission and had not experienced any in-flight complications. His chest pain had resolved without intervention and SJMS was determined to be unlikely to impact his flight performance (such as response to supplemental oxygen) or life expectancy.Harrison MF, Cowl CT. Incidental diagnosis of Swyer-James-MacLeod syndrome in a military pilot. Aerosp Med Hum Perform. 2021; 92(4):281285.

Swyer-James-MacLeod syndrome and pulmonary arteriovenous malformations: a rare combination.

This case describes a female patient who presented with an acute on chronic deterioration in respiratory symptoms, on a background of chronic obstructive pulmonary disease and heavy cigarette smoking. Chest radiograph demonstrated long-standing hyperlucency of the right lower lobe, with further imaging later confirming the rare combination of Swyer-James-MacLeod syndrome and multiple pulmonary arteriovenous malformations within the affected lung.

Hiperclaridad pulmonar unilateral en un niño: Síndrome de Swyer-James-Macleod / A pediatric clinical case of unilateral hyperlucency of the lung: Swyer-James-Macleod syndrome

El síndrome de Swyer-James-MacLeod es un cuadro pulmonar muy infrecuente que se caracteriza radiológicamente por hiperclaridad pulmonar lobar o multilobar, producido por la disminución de la vascularización pulmonar, por lo general, por un proceso infeccioso previo. En ocasiones, es un hallazgo casual en el estudio por infecciones respiratorias o crisis asmáticas repetidas. Se presenta el caso de un paciente de 6 años de edad que, en el contexto de probable neumonía persistente, se realizó un amplio estudio que permitió el diagnóstico del síndrome de Swyer-James-MacLeod. Se desarrolló un abordaje terapéutico multidisciplinar con el que, hasta la fecha, la evolución ha sido satisfactoria, con tratamiento médico y fisioterapia respiratoria.

Swyer-James-MacLeod syndrome is a rare pulmonary disorder that is characterized radiologically by pulmonary lobar or multilobar hyperlucency produced by decreased pulmonary vascularization usually due to a previous infectious process. It is frequently a casual finding in recurrent pulmonary infections or in the study of asthma symptoms. We present the case of a 6-year-old patient who was diagnosed with Swyer-James-MacLeod syndrome after a persistent pneumoniae. A multidisciplinary approach was conducted, with a satisfactory progress up to date, combining medical treatment with respiratory physiotherapy.

An incidental image of a patient with chest pain after fall from a tree: Swyer-James-MacLeod syndrome?

Unilateral hyperlucent lung was firstly described by Swyer and James 1950s. After that, some patients with same disease were detected by Macleod . Then this syndrome was named as Swyer- James-Macleod syndrome (SJMS), and this syndrome includes a smaller or normal sized unilateral hyperlucent lung. The diagnosis of SJMS includes a detailed evaluation and the exclusion of other reasons of unilateral hypertranslucency. In literature, small groups of patients with this syndrome have been described. This paper reports a 45-year-old male presented to our emergency department with chest pain after fall from height 24 h ago. In his computed tomography no rib fracture, pneumothorax and hemothorax. But an abnormal image (5.5 cm hyperlucent area) was seen in his left lung. This report aims to present one of incidentally diagnosed rare case of SJMS.

Swyer-James-Macleod syndrome mimicking an acute pulmonary embolism: a report of six adult cases and a retrospective analysis.

BACKGROUND AND AIM: In patients with pulmonary embolism (PE), a pulmonary radiograph may reveal oligemic fields (the Westermark sign) associated with sites of occlusion of the pulmonary arteries, interruption or loss of the artery line (the knuckle sign), and even unilateral hyperlucency attributable to reduced overall lung vascularity. In Swyer-James-Macleod syndrome (SJMS), which develops as a result of bronchiolitis obliterans, unilateral hyperlucency is evident because of emphysema and hypoplasia of the pulmonary artery and its branches. Therefore, SJMS cases with clinical and laboratory data compatible with PE may in fact be confused with PE. The cases of six adult patients who were initially presumed to have PE but on further investigation were diagnosed with SJMS are presented in this report, which thus can serve as a guide for diagnosis of similar cases in future. METHODS: We studied six adult patients who presented with dyspnea. Their pulmonary radiographs revealed lobar/unilateral hyperlucency and PE was initially suspected. The pulmonary artery and branches thereof exhibited parenchymal emphysema and hypoplasia, and we thus diagnosed SJMS. RESULTS: We studied 4 males and 2 females with a mean age of 51 years (range, 20-73 years). Left lung involvement was evident in five cases. CONCLUSION: Unilateral hyperlucency may be a feature of both PE and SJMS. Although these conditions are very different, both present similarly in radiographic terms and may be easily confused when the clinical data and the anamnesis raise a suspicion of PE, causing unnecessary testing and treatment.

Complication of post-infectious bronchiolitis obliterans (Swyer-James syndrome).

Swyer-James syndrome is a complication of post-infectious bronchiolitis obliterans that causes inflammation and fibrosis of the bronchial walls. There are two types: asymptomatic, with most cases diagnosed in adults during routine radiological examinations; and symptomatic, most commonly found in children. Here, we report the case of a 6-year-old child with recurrent dyspnea since the age of 3, who showed signs and symptoms of bronchiolitis obliterans and radiological signs of bronchial wall thickening and air trapping. The clinical and radiological findings led to the diagnosis of Swyer-James syndrome. Treatment of this syndrome is intended to reduce the pulmonary lesions and improve the patient's quality of life.

Swyer-James-Macleod Syndrome: A Rare Entity.

Swyer-James-MacLeod syndrome is a radiological entity characterised by hyperlucency of one or more lobes or of the entire lung associated with decreased number and diameter of ipsilateral peripheral pulmonary vessels resulting in difficult visibility of the arterial network, and unobstructed bronchial system. We report the case of a 12-year-old girl who presented with chief complaint of pain in the chest. Chest radiograph (postero-anterior view) showed hyperlucent left lung field with increased lung volume and shift of the mediastinum to the right side. Contrast-enhanced computed tomography (CECT) of the chest showed hyperlucency and diminished vascularity in the upper lobe and lingula of left lung with hyperinflation of the pulmonary parenchyma. On the basis of clinical and radiological findings, the patient was diagnosed to have Swyer-James-MacLeod syndrome.