Exogenous lipid pneumonia with hyperpyrexia: a case report / 北京大学学报(医学版)

Lipoid pneumonia (LP) is an uncommon form of pneumonia that is characterized by the presence of intra-alveolar lipid and lipid-laden macrophages on microscopy. It categorized as exogenous lipoid pneumonia (ExLP) and endogenous lipoid pneumonia (EnLP). Exogenous lipoid pneumonia caused by inhalation of liposuction substances (animal fat, vegetable oil, or mineral oil), mostly, in adult cases, they were medicines for constipation or rhinopharyngitis. Most of these patients showed mild clinical manifestations, and chronic medical condition. There were reports of lipoid pneumonia being successfully treated with corticosteroids, immunoglobulins and whole lung lavage. We report a case of exogenous lipoid pneumonia characterized by high fever and acute medical condition. A 77-year-old woman with hypertension and diabetes mellitus, accepted paraffin oil treatment for "incomplete intestinal obstruction", then, an accident of aspiration happened, as she went through the history of coughing while eating, followed by persistent hyperthermia and increases of white blood cells (WBC). Chest CT showed progressive ground-glass opacities, accompanied with fusion of consolidation, her sputum etiological examination was negative, and the therapy of broad-spectrum antibiotic was invalid. The patient was subjected to bronchofibroscopy with bronchoalveolar lavage (BAL). The bronchoalveolar lavage fluid (BALF) appeared colorless and transparent, and did not show a milky appearence. Total cell count of the BALF was 2.0×109 cell/mL, including 7.2% macrophages and 92.8% neutrophils. Cultures of the BALF were negative for bacterial, fungal, and mycobacterial pathogens. The BALF cytologic findings showed vacuolated lipid-laden macrophages (Oil Red O staining). These findings revealed exogenous lipoid pneumonia. There were reports of lipoid pneumonia being successfully treated with corticosteroids, immunoglobulins, and whole-lung lavage. So this patient was treated with methylprednisolone 120 mg/d for 3 days and 80 mg/d for 6 days, at the same time, immunoglobulins was given to infusion, but the daily peak temperature of the patients fluctuated between 38 and 39 degrees. Then, whole lung lavage was performed 28 days after admission. Unfortunately, acute pulmonary edema occurred during the operation, as the tracheal intubation problems, and 6 days later, the patient died at last. The clinical manifestations of exogenous lipid pneumonia vary greatly, from asymptomatic to life-threatening symptoms, and as febrile low fever is the main manifestation, but hyperthermia may also be the remarkable presentation.

Neumonía lipoidea tras aspiración accidental de vaselina / Lipoid pneumonia after accidental aspiration of vaseline

La neumonía lipoidea es una patología pulmonar poco conocida que resulta de la acumulación de lípidos de origen endógenos o exógenos a nivel alveolar pulmonar. Suele ser subdiagnosticada dada que la presentación clínica es inespecífica, por lo que suele confundirse con otras patologías broncopulmonares, muchas veces no llegándose al diagnóstico preciso de esta. Dentro de los datos anamnesticos el antecedente, ya sea de uso reciente o de larga data, de la ingesta y/o inhalación accidental o voluntaria de alguna sustancia que contenga algún componente lipídico en su base es primordial. Pacientes de edades extremas suelen ser los más afectados y en quienes debemos tener la sospecha diagnóstica. Su diagnóstico es complejo requiriendo historia clínica, imágenes que apoyen un compromiso pulmonar (de preferencia tomografía computada) e idealmente la detección intraalveolar de lípidos y macrófagos cargados de éstos en el lavado broncopulmonar. Actualmente, no existe consenso sobre el manejo específico de esta patología. Lo importante es identificar la causa y descontinuar su uso. En neumonía lipoidea de causa exógena se recomienda manejo de soporte según sea la presentación clínica del paciente.

Lipoid pneumonia (LN) is an unknown lung disease due to lipid accumulation, of endogenous or exogenous origin, at the pulmonary alveolar level. Often it is underdiagnosed because of its nonespecific presentation whose differential diagnosis are other bronchopulmonary diseases. Anamnestic data, either recent or long-standing use of the intake and / or accidental or deliberate inhalationof any substance containing a lipid component as its base is relevant information. Extreme age groups are the most affected, and in whom wemust suspect LN diagnosis. The diagnosis of LN is complex and requires a complete medical history, images that support lung involvement (preferably computed tomography), and ideally intraalveolar detection of lipid-ladenmacrophages in the bronchoalveolar lavage. Currently, there is no consensus on the specific management of this disease. It is important to identify the cause and discontinue its exposure. Whatever the varity of the clinical presentation, support magement is recommended to treat LN.

Evolução da pneumonia lipoide exógena em crianças: aspectos clínicos e radiológicos e o papel da lavagem broncoalveolar / Evolution of exogenous lipoid pneumonia in children: clinical aspects, radiological aspects and the role of bronchoalveolar lavage

OBJETIVO: Descrever os aspectos da evolução da pneumonia lipoide em crianças, com base em aspectos clínicos, radiológicos e de achados no lavado broncoalveolar, enfatizando a importância diagnóstica e terapêutica da lavagem broncoalveolar. MÉTODOS: Foram incluídas 28 crianças, com idade média de 20 meses (1-108 meses) e diagnóstico de pneumonia crônica refratária a antimicrobianos e/ou TB. A maioria apresentava um fator de risco para aspiração, e todas apresentavam história de ingestão de óleo mineral para o tratamento de constipação intestinal (23/28) ou de ascaridíase complicada (5/28). A avaliação clínica e tomográfica e análises do lavado broncoalveolar foram realizadas no início do tratamento e em até 24 meses. RESULTADOS: Os sintomas mais frequentes foram taquipneia e tosse. As principais alterações radiológicas foram consolidações (23/28), infiltrado peri-hilar (13/28) e hiperinsuflação (11/28). A TC de tórax mostrou consolidações com broncograma aéreo (24/28), diminuição de atenuação nas áreas de consolidação (16/28), opacidade em vidro fosco (3/28) e padrão em mosaico (1/28). O estudo do lavado broncoalveolar apresentava macrófagos espumosos corados por Sudan, confirmando o diagnóstico da pneumonia lipoide. Após tratamento com múltiplas lavagens broncoalveolares (média = 9,6), 20 crianças tornaram-se assintomáticas, havendo normalização tomográfica em 18. CONCLUSÕES: O diagnóstico de pneumonia lipoide deve ser considerado na pneumonia crônica ou TB refratárias ao tratamento, especialmente se houver história de ingestão de óleo mineral. A broncoscopia com múltiplas lavagens broncoalveolares mostrou-se eficiente para a depuração do óleo aspirado do parênquima pulmonar e a prevenção da fibrose, contribuindo para a redução da morbidade dessa doença, que ainda é pouco diagnosticada.

OBJECTIVE: To present aspects of the evolution of lipoid pneumonia in children, based on clinical, radiological and bronchoalveolar lavage fluid findings, emphasizing the importance of bronchoalveolar lavage for the diagnosis and treatment. METHODS: We included 28 children, with a mean age of 20 months (range, 1-108 months), diagnosed with chronic pneumonia refractory to antimicrobial therapy, with TB or with a combination of the two. Most of the children had at least one risk factor for aspiration, and all of them had a history of mineral oil ingestion for intestinal constipation (23/28) or complicated ascaridiasis (5/28). Clinical evaluations, tomographic evaluations and analyses of bronchoalveolar lavage fluid were carried out at the beginning of treatment and throughout a follow-up period of 24 months. RESULTS: Tachypnea and cough were the most common symptoms. The most common radiological alterations were areas of consolidation (23/28), perihilar infiltrates (13/28) and hyperinflation (11/28). Chest CT scans showed areas of consolidation with air bronchogram (24/28), decreased attenuation in the areas of consolidation (16/28), ground-glass opacities (3/28) and crazy-paving pattern (1/28). In the analysis of the bronchoalveolar lavage fluid, Sudan staining revealed foamy macrophages, confirming the diagnosis of lipoid pneumonia. After treatment with multiple bronchoalveolar lavages (mean = 9.6), 20 children became asymptomatic, 18 of those presenting normal tomographic images. CONCLUSIONS: A diagnosis of lipoid pneumonia should be considered in patients with chronic refractory pneumonia or TB, especially if there is a history of mineral oil ingestion. Bronchoscopy with multiple bronchoalveolar lavages was an efficient treatment for the clearance of mineral oil from the lung parenchyma and the prevention of fibrosis. This strategy contributed to reducing the morbidity of lipoid pneumonia, which remains a rare diagnosis.

Neumonía lipoidea exógena aguda, a propósito de un come fuego / Acute exogenous lipoid pneumonia

Se presenta el caso de un adulto de 19 años de edad, malabarista y manipulador de fuego quien ingresa al servicio de emergencias médicas del Hospital Dr. Rafael A. Calderón Guardia, por un cuadro súbito de dolor tóracico derecho y disnea. Se documenta derrame pleural en base pulmonar derecha y masa redondeada en base pulmonar izquierda. Inicia clínica sugestiva de bronconeumonía horas despúes, abordándosele inicialmente como neumonía adquiridad en la comunidad. Considerando sus antecedentes laborales y características clínicas, se reorienta caso y se concluye que se trata de lo que en la bibliografía médica en habla inglesa se denomina "fire-eater pneumonia", infrecuente neumonía por aspiración de la que se hace una revisión. Descriptores: neumonía lipoidea, neumonía por aspiración, neumonía en come fuego.

Pneumonia lipóide: relato de caso / Lipoid pneumonia: case report

A pneumonia lipóide resulta, na maioria das vezes, da aspiração de óleo mineral. Tem seu diagnóstico subestimado, pois apresenta quadro clínico inespecífico ou ausente e alterações radiológicas que simulam outras doenças pulmonares. É mais freqüente na criança e no adulto debilitado, mas pode ocorrer em pessoas saudáveis. O objetivo é relatar o caso de uma lactente de dois meses, hígida, que fez uso prévio de óleo mineral, desenvolvendo pneumonia inicialmente tratada como bacteriana, mas a história compatível e a dissociação clínico-radiológica nos alertaram para o diagnóstico de pneumonia lipóide, que foi confirmado através do lavado broncoalveolar que mostrou glóbulos de gordura no interior dos macrófagos alveolares. Ressalta-se a importância deste diagnóstico nos casos de pneumonia de evolução arrastada sem resposta a antimicrobianos, especialmente em lactentes ou crianças com fatoresde risco para aspiração e com história de ingestão de óleo mineral.

Insuficiencia respiratoria aguda por neumonia lipoidea / Acute respiratory failure due to lipoid pneumonia

Se presenta un paciente de sexo masculino e3 63 años de edad que ingresó por un síndrome febril, tos, expectoración e insuficiencia respiratoria severa. El lavado broncoalveolar reveló una baciloscopia positiva por lo que indicó un tratamiento especifico con cuatro drogas antituberculosas. Ante la ausencia de mejoría el paciente requirió asistencia respiratoria mecánica. Una tomografía computada de tórax mostró signos compatibles con neomonía lipoidea tales como el compromiso de zonas declives del pulmón, hipodensidad de las mismas y el signo del angiograma. Un nuevo interrogatorio familiar constató ingesta crónica de vaselina por constipación. Se decidió tratamiento con altas dosis de esteroides y lavado pulmonar total, que permitió el retiro de la asistencia respiratoria mecánica. Se comenta la asociación de esta patología con infecciones por micobacterias atípicas.