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 Table of Contents  
IP CHRONICLES
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 272-273

Lipoid pneumonia: Fifty years of unmasking an unusual cause of pneumonia


Department of Pediatrics, Lady Hardinge Medical College and Associated Kalawati Saran Children's Hospital, New Delhi, India

Date of Submission29-Oct-2021
Date of Acceptance30-Oct-2021
Date of Web Publication29-Nov-2021

Correspondence Address:
Dr. Ravitanaya Sodani
Department of Pediatrics, Lady Hardinge Medical College and Associated Kalawati Saran Children's Hospital, New Delhi
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ipcares.ipcares_331_21

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How to cite this article:
Sodani R. Lipoid pneumonia: Fifty years of unmasking an unusual cause of pneumonia. Indian Pediatr Case Rep 2021;1:272-3

How to cite this URL:
Sodani R. Lipoid pneumonia: Fifty years of unmasking an unusual cause of pneumonia. Indian Pediatr Case Rep [serial online] 2021 [cited 2022 Jan 20];1:272-3. Available from: http://www.ipcares.org/text.asp?2021/1/4/272/331386

Lipoid pneumonia (LP) is a rare form of pneumonia caused by inhalation or aspiration of lipids/oils of animal or plant origin. In the last 50 years, LP resulting from various cultural, medical, and behavioral practices has been reported in literature worldwide, in all age groups. A peak was noted between 1987 and 2006; however, with improved understanding and awareness of this entity, there has been a decline in its incidence. Due to the nonspecific presentation, diagnosis is often delayed or missed in children. We selected this case report from 50 years ago, as it is an important and entirely preventable pediatric cause of persistent pneumonia and interstitial lung disease (ILD).


  Clinical Case Description Top


The case report by Bakshi et al.[1] describes a 3-month-old female child who was apparently well till 1 month of age. The infant presented with a history of recurrent episodes of fever, vomiting, and diarrhea associated with marked irritability for 1.5 months. Past, antenatal, perinatal, and family history was noncontributory. General physical examination was normal. Salient systemic examination findings included firm and nontender enlargement of the liver and spleen; 5 cm and 7 cm below costal margin, respectively. Respiratory examination revealed reduced breath sounds on the right side.

Blood investigations were normal, except for mild anemia and high ESR (55 mm). Chest radiography showed dense opacity on the right upper and middle zone. The tomography report described it as a mass in the posterior mediastinum. The skeletal survey and bone marrow examination were inconclusive. The differential diagnoses of posterior mediastinum mass considered were ganglioneuroma, cyst, teratoma, and gut duplication. Surgery revealed a solid mass involving the right upper and lower lobes, for which right pneumonectomy was done. Histopathological examination found inflammatory granulomas with large masses of sudanophilic material. The baby died due to postoperative complications. Autopsy revealed extensive pneumonia, a fatty liver, and reactive hyperplasia in the spleen. The final diagnosis was foreign body granuloma in the lung due to lipid aspiration or LP. On reviewing the history, it was elicited that the infant had been fed “ghee” daily from the age of a month due to some social custom. The authors hypothesized that the hepatosplenomegaly was the reticuloendothelial response to the foreign body granuloma formation.


  Brief Historical Review Top


LP has been reported in the literature since 1925, when it was first associated with chronic use of laxative- and oil-based nasal drops. It has been described under different names such as paraffinoma, cholesterol pneumonia, and lipid granulomatosis, each denoting the association with inhalation or ingestion of oily substances. The exact incidence of LP is still not known. Initially, it was usually reported in children with defects in deglutition or neuromuscular diseases due to increased chances of aspiration, but there are several case reports in healthy individuals as well. In most of the earlier cases, diagnosis was established either on surgically resected specimens or autopsy. Treatment was based upon individual experience.


  Advances in the Last 50 Years Top


We briefly discuss various aspects of LP that have become clearer in the past decades.

Risk factors

Besides the aforementioned causes, the most causes were due to the use of home remedies such as oily nasal drops, feeding animal fat (like “ghee” in this case) to establish regular bowel habits, or transnasal administration of medicines for coughs and colds. Medical causes include Lorenzo's oil in the management of adrenoleukodystrophy,[2] and more recently with a ketogenic diet for management intractable seizures.[3] A systemic review by Marangu et al.[4] observed that out of 44 studies, 25 reported co-infection cultured from blood or respiratory tract secretions. The most common infection (6 studies) was nontuberculous mycobacteria (NTM). Other infections noted included Branhamella catarrhalis, Pseudomonas, Acinetobacter, Klebsiella, and common respiratory viruses.

Clinical presentation and diagnosis

Children with LP may present acutely with fever, cough, and respiratory distress or with nonspecific symptoms that are insidious in onset and depend upon the degree of exposure to lipid/oil. Physical examination is usually normal, although dullness on percussion, crackles, wheezes, and/or rhonchi may be present. In long-standing progressive disease, physical findings related to chronic hypoxia such as clubbing may develop.[5] Blood investigations are usually normal, unless a coinfection exists, in which case there will be leukocytosis. Once a history compatible with exposure to lipids/oils is certain, the diagnosis can be confirmed by demonstration of intra-alveolar lipid and lipid laden macrophages in the sputum or on bronchoalveolar lavage (BAL).

Radiologic features

The findings of suspected LP on a chest radiograph are neither specific nor diagnostic. Patients who present acutely may show homogenous dense opacities, often with air bronchograms and sometimes a fine, “spun glass” appearance. These changes may be seen as early as 30 min and in most cases by 24 h of acute exposure. Those with chronic symptoms may have ground-glass or dense opacities (as seen in consolidation) that involve one or more segments, typically with peribronchovascular distribution and involving the lower lobes.[5] A computerized tomographic (CT) scan of the chest may reveal diffuse airspace/ground-glass opacities with basal/lower lobe predominance. Less frequent findings are fatty attenuation, interstitial septal, thickening/crazy-paving appearance, expansile pneumonia, and nodules. CT can detect areas of fat attenuation as low as −30 HU. Recently, by using advanced fat saturation techniques, magnetic resonance imaging (MRI) has been utilized to identify fat depressed signals diagnostic of LP that are found in the middle of the lobe, or within the consolidation and/or nodules and get missed on CT.[5]

Treatment

The optimal treatment of LP is still unknown. Avoiding risk factors that cause exposure to lipids and supportive care is the mainstay of current management. The use of anti-inflammatory agents such as steroids has been reported by some authors, but there is lack of clinical trials to validate this. There are a few reports of successful treatment with intravenous immunoglobulin.[4] Mycobacterial infection must be excluded before immunosuppression. Other modalities include repeated lung lavage and surgical resection of the involved segment.

Prognosis and outcome

LP is usually indolent, if recognized early, and the risk factors modified. In patients with chronic symptoms and delayed diagnosis, the disease can be persistent and difficult to treat. Though patients show clinical improvement, radiological signs may persist. Mortality is reported inconsistently.[4],[5]

To conclude, LP is a frequently missed diagnosis in children because of nonspecific clinical-radiological presentation and lack of knowledge about the various risk factors for aspiration or inhalation of oils/fats. A high index of suspicion should be kept in any child presenting with unexplained pneumonia, and history should be taken for exposure to lipid-containing substances. LP is easily preventable if we increase awareness among the general population and professional colleagues about this entity and modify our cultural and medical practices.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Bakshi S, Bhakoo ON, Singh S, et al. Lipoid pneumonia. (A case report). Indian Pediatr 1971;8:793-5.  Back to cited text no. 1
    
2.
Cheon KR, Cho HJ, Kim SS, et al. Lipoid pneumonia following aspiration of Lorenzo's oil in a child with X-linked adrenoleukodystrophy. Hong Kong J Paediatr 2017;22:225-8.  Back to cited text no. 2
    
3.
Kang HC, Chung DE, Kim DW, et al. Early and late-onset complications of the ketogenic diet for intractable epilepsy. Epilepsia 2004;45:1116-23.  Back to cited text no. 3
    
4.
Marangu D, Gray D, Vanker A, et al. Exogenous lipoid pneumonia in children: A systematic review. Paediatr Respir Rev 2020;33:45-51.  Back to cited text no. 4
    
5.
Hadda V, Khilnani GC. Lipoid pneumonia: An overview. Expert Rev Respir Med 2010;4:799-807.  Back to cited text no. 5
    




 

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