E-ISSN 1858-8360 | ISSN 0256-4408
 

Case Report 


SUDANESE JOURNAL OF PAEDIATRICS

2021; Vol 21, Issue No. 1

CASE REPORT

A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage

Abdullah Saeed AlShamrani (1), Mohammed Abdullah Alzaid (2), Sarrah M. Fadl (1), Mohammed Ahmed AlFaki (1)

(1) Pediatric Pulmonary Section, Pediatric Department, Prince Sultan Military Medical City, Riyadh, Saudi Arabia

(2) Pediatric Pulmonary Section, Children Specialized Hospital, King Fahad Medical City, Riyadh, Saudi Arabia

Correspondence to:

Abdullah S. Al-Shamrani

Pediatric Pulmonary Section, Pediatric Department, Prince Sultan Military Medical City, Riyadh, Saudi Arabia

Email: dr.alshamrani99 [at] gmail.com

Received: 26 April 2020 | Accepted: 13 July 2020

How to cite this article:

Al-Shamrani A, AlZaid MA, Fadl SM, Faki M. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudan J Paediatr. 2021;21(1):82–88.

https://doi.org//10.24911/SJP.106-1587896995


ABSTRACT

Exogenous lipoid pneumonia (ELP) is a known complication of lipid administration through either the nasal or oral route. ELP in paediatric patients is usually managed by discontinuing lipid administration and supportive care, including respiratory support and the use of antibiotics for secondary bacterial infection. The other modalities that remain controversial include the use of corticosteroids and clearing lipids by whole lung lavage. We report a 2-month-old infant who presented with pneumonia and whose further history revealed exposure to ghee over a month period while visiting her grandparents in the south region of Saudi Arabia. The patient was later diagnosed as having ELP and was successfully managed with modified whole lung lavage till weaned off from oxygen. Unfortunately, the case was complicated by nontuberculous Mycobacterium chelonae, and the patient died despite maximum intervention.


KEYWORDS

Pneumonia; Lipoid pneumonia; Infant; Bronchoscopy; Lavage; Ghee.


INTRODUCTION

Exogenous lipoid pneumonia (ELP) in infants and children is a consequence of the aspiration of either vegetable oil, mineral oil or animal fat that is given orally or instilled intranasally [1,2]. It has been reported in areas, where the use of oils is a part of traditional remedies [36]. In Saudi Arabia, the prevalence of the practice of herbal medicine was 59.3% in 155 children admitted with acute lower respiratory illnesses, especially in bronchiolitis, where sesame, fenugreek and olive oils were the most common [7]. Ghee is an animal source oil that is often used by Saudi older generation as a traditional habit to increase nutritional support and clear the nasal blockage [4, 5, 8, 9]. In 1995, Annobil et al. [4] were the first to report the clinical and pathological change of four children following the practice of force feeding with animal fat (ghee) in infancy in the southern part of Saudi Arabia. The aspiration of fat animals induces diffuse lung injury and causes interstitial mononuclear pneumonitis, intra-alveolar desquamation of pneumocytes, lipid granuloma formation and confluent bronchopneumonia with microabscess formation due to superadded infection. Bronchiectasis was observed as a complication of lipoid pneumonia, and lung fibrosis was reported as well. Unfortunately, the outcome was very alarming, similar to what we experienced in the patient. For this, we believe that the present report could be beneficial in the region and may improve the outcome [10].


CASE REPORT

A 2-month-old girl was admitted through the Emergency Department with a history of bluish discoloration of the face, shortness of breath, cough, poor feeding and chocking for 2 weeks before admission. She was born in a specialised medical centre in Riyadh following a full-term, uneventful pregnancy and normal delivery and had a birth weight of 2.6 kg. She was discharged on the 2nd day in good condition. She was the first baby to young non-consanguineous parents. The family moved to the southern part of Saudi Arabia for social support for the mother after delivery, the baby was predominantly on bottle milk and was nursed as well by grandmother (maternal side) and there was no concern for her in the 1st month regarding both feeding and breathing. At the age of 6 weeks, the baby was admitted to Al-Sarat Hospital (in the southern region of Saudi Arabia) due to pneumonia following choking by milk but without vomiting. The girl had no history of fever, nasal congestion, apnoea, eye discharge, skin rash or abnormal movement. The diagnosis of pneumonia was made based on clinical findings and bilateral lung infiltration, which was more predominant on the right side. She was started on cefotaxime and vancomycin for 2 weeks with no improvement in her clinical condition. Therefore, the family left the hospital against medical advice, and the patient was taken to King Fahad Medical City (KFMC) in Riyadh for the further evaluation.

The examination on arrival to the Emergency Department at KFMC revealed an alert baby with all growth parameters below the third percentile. The weight was 3.03 kg, the length was 46 cm and head circumference was 34 cm. She was afebrile, respiratory rate was 55/minute, heart rate was 170/ minute, blood pressure was 75/43 mmHg and her oxygen saturation was 60% in room air. Therefore, she was judged to be suffering from moderate to severe respiratory distress with significant tachycardia and severe hypoxemia. She required 2 l/minute of nasal cannula oxygen to maintain a saturation of greater than 90%. She had decreased breath sounds bilaterally (more so, in the right lung), vesicular type of breathing, with no additional sounds [10]. Apart from tachycardia, the cardiovascular system examination was within normal limits. She was alert with good tone and reflexes, and the examination of the rest of the other systems was normal.

The laboratory investigation results were as follows.

The blood gas analysis showed partially compensated respiratory acidosis (pH 7.28, PCO2 65 mmHg and HCO3 30.2 meq/l). A complete blood count showed a markedly elevated haemoglobin level (15.4 g/dl) despite the expected physiological anaemia at these 2 months of age (8 g/dl), indicating that the patient suffers from significant hypoxemia and is compensating by increasing the haemoglobin level. The white blood cell count (10.4 × 109/L-normal 4-10 × 109/L) and platelet count (237 × 109/l-normal 150-450 × 109/l) were both normal. Serum electrolyte levels were normal. Blood culture was negative, and the nasopharyngeal aspirate was negative for respiratory syncytial virus, parainfluenza, human metapneumovirus and influenza viruses. The patient’s chest X-ray on arrival is shown in Figure 1, and the computed tomography (CT) scan is shown in Figure 2.

Based on the high index of suspicion for a baby just coming from an endemic area, who was fed for some time by the grandmother, with the presence of ground-glass opacity on the chest X-ray in the absence of any genetic or metabolic condition in her family, the pulmonology team raised the suspicion of ELP. Therefore, bronchoscopy and lavage were performed. The bronchoalveolar lavage fluid (BALF) was milky (Figure 3), and the cell density was within the normal range (72 cells/mm3) but with a neutrophilic predominance of 39%. The lymphocyte, eosinophil and macrophage counts were within the normal range.

Figure 1. Anteroposterior chest radiograph showing homogenous opacity involving the whole right side of the lungs with a little opacity in the left middle zone and no shift of the trachea or mediastinum. No air bronchogram and no significant deviation of the trachea to the right side are visible, which is suggestive of right-side alveolar infiltrate.

Figure 2. A 1-mm axial section of the lung window image obtained using high-resolution CT of the chest. The image shows low attenuation, ground-glass opacity, septal thickening and air bronchogram involving the right middle and lower lobes. These results are very suggestive of intrapulmonary fat infiltrate with a Hounsfield unit score of −40.

The culture of the alveolar fluid was negative. We processed the lavage to determine the lipid profile because it was milky, and the levels were surprisingly high: cholesterol (0.1 mmol/l), triglycerides (0.84 mmol/l), serum cholesterol (5.1 mmol/l) and serum triglyceride (0.83 mmol/l). The BALF was strongly positive for lipid-laden macrophages (LLMs) and showed Oil Red O staining (Figure 4); unfortunately, the LLM index (LLMI) was not determined.

At this stage, we were convinced that the patient had ELP. This was based on history that the grandmother was giving a daily cup of ghee (buttered milk) as forceful feeding to improve the weight of the child. Furthermore, the low attenuated ground-glass appearance with negative Hounsfield unit (HFU) in the range from −20 to −150 was consistent with lipid material. Furthermore, the floating of the milk on the upper part of the lavage above the saline is very consistent with lipid material as the density of lipid is less than that of saline (0 HFU), and finally, the presence of LLM was confirming the intraparenchymal lipid material, with no hepatospleenomegaly and negative family history for any metabolic disease such as Niemann-Pick disease or Gaucher disease. Her swallowing reflux was normal excluding the palate-pharyngeal incoordination, her tone and reflexes were normal excluding an underlying myopathy or neuropathy and there was no high arched or cleft palate to suspect aspiration. H-type tracheoesophageal fistula could be included in the differential of such a case, but we were convinced with the lipoid pneumonia as it is common in the southern part of Saudi Arabia. We did not expose the child to a modified barium study, and fistula was not noted during the repeated bronchoscopies for this patient.

Figure 3. Milky appearance of the BALF taken from both sides of the lung.

Figure 4. Oil Red O-stained cytospin preparation showing numerous fat vacuoles occupying at least 50% of the macrophage.

The patient was treated with modified whole-lung lavage with significant improvement exhibited by weaning from oxygen and achieving a saturation of 95% on room air within 48 hours. She was subjected to three bronchoscopies; the first was diagnostic. For the second bronchoscopy, the patient was subjected to right lung lavage with the instillation of warm (body temperature) normal saline (total amount, 160 ml; multiple washouts of 15 ml each). This procedure returned to 137 ml (86%) of milky fluid that separated into two layers with a white oily layer floating on top within 1 hour, which is suggestive of a low-density lipid compared to normal saline. This was followed by a third bronchoscopy with lavage of the left lung, instilling a total of 200 ml of normal saline with a return of 130 ml (65%) of milky fluid. The patient tolerated the procedure well without any significant desaturation. Within 2 days, she was weaned off oxygen with a mean saturation of 94% in room air. A week later after the intervention, the patient remained well oxygenated with oxygen saturation in mid-90%, feeding was well and she was discharged home off oxygen in good condition (Figure 5).

Figure 5. Anteroposterior chest radiograph showing marked resolution of the right lung infiltration with residual mild lung hyperinflation and a normal cardiac shadow appearance.

Unfortunately and surprisingly, 2 days after discharge, the patient was readmitted with pneumonia and respiratory failure and required mechanical ventilation and critical care management. Initially, we confirmed that her symptoms were not caused by ongoing oil exposure. The complication of ELP was suspected, and we started the patient on anti-tuberculous medications (isoniazid, rifampicin and azithromycin) as it is known from the literature that ghee could be contaminated by Mycobacterium fortuitum or Mycobacterium chelonae [10,11].

Later, the previous blood culture revealed a positive test result for nontuberculous M. chelonae from both blood and gastric aspirate. The patient’s condition was further complicated by sepsis and disseminated intravascular coagulation with the further isolation of Enterobacter cloacae and Pseudomonas from tracheal aspirate. Despite good antibiotic coverage (vancomycin, meropenem and ciprofloxacin), the patient died within 4 days of the second admission.


DISCUSSION

Exogenous lipoid pneumonia occurs due to the build-up of lipids within alveoli caused by the aspiration of oily material from animal, vegetable, or industrial sources [12,13]. The patient developed lipoid pneumonia after aspirating ghee, an animal-based oil that is used in the region because it is believed to relieve cough and cold symptoms, improve bowel movements, have nutritional value and enhance overall health [3,4,6,9]. This rare disease has been reported in countries, where traditional practices involve the use of oil that is instilled as nasal drops or given orally.

In southern Saudi Arabia, this practice is, especially, common among the elderly, and although it has declined in recent years, cases are still reported. Cases have also been reported in the other countries with similar beliefs, such as Oman, India, Mexico, Brazil and Ghana [5,9]. Instilling oil in the nose of a baby is thought to relieve nasal obstruction, especially during cold weather or flu illness. It is thought to improve the immunity and increase the weight, and the case was very unlucky to have been exposed to ghee reported to cause the worst type of lipoid pneumonia [1416].

Exogenous lipoid pneumonia presents with symptoms similar to community-acquired pneumonia and is often managed initially as such [17]. Although this type of pneumonia can be difficult to manage, recurrent respiratory illnesses and/or associated failure to thrive are common presentations [18]. A history of oil administration is usually not reported by young parents unless specifically requested; therefore, the diagnosis requires a high index of suspicion [9]. For this reason, we believe that asking questions pertaining to ELP should be routine in areas, in which the traditional practice of oil administration is common.

The diagnosis of ELP is based on clinical findings coupled with the index of suspicion. The diagnosis is confirmed by radiological findings and a positive result for LLMs in bronchoalveolar lavage [19]. Radiological features include low attenuation consolidation (the typical range is between −20 and −150) and a ‘crazy-paving’ pattern [20]. The patient’s chest CT scan showed bilateral ground-glass opacities (which are not classically observed for airspace consolidation), interlobular septal thickening of the lower lobes and a negative Hounsfield unit opacity, which is consistent with ELP [20].

We believe that if there is a history of exogenous lipid being given to the child, chest CT scan could be enough to confirm the diagnosis, especially when the HFU is consistent with negative attenuation with certain density of HFU. Nevertheless, the strongest evidence is LLM being strongly positive with a high LLMI. The patient was a definitive case due to the presence of all parameters (history, radiological finding and positive LLM). It is noteworthy that Mycobacterium infection has been reported to be associated with ELP, and one of the reported cases was specifically associated with M. chelonae [10,11].

The first step in managing ELP is to terminate the exposure to the oily substances, and supportive management includes respiratory support with oxygen and treatment with antibiotics for secondary bacterial infection plus adequate nutrition. Patients should be investigated for a possible underlying cause of their aspiration, and the other modalities of treatment remain controversial, including a prolonged course of corticosteroids [9,17,18] and whole lung lavage in cases of symptomatic children with respiratory compromise [21,22].


CONCLUSION

Exogenous lipoid pneumonia should be assigned a high index of suspicion and should be considered in any case of unresolved pneumonia because misguided traditional habits (the forced feeding of infants with ghee) continue to occur, especially in Asir Region [23]. Bronchoalveolar lavage is a simple method for confirming the diagnosis. Modified therapeutic lung lavage is the optimal therapy for such cases. The cases of ELP can be complicated by Mycobacterium infection with expected high mortality. Popular awareness can be raised through the national media.


CONFLICTS OF INTEREST

The authors declare that there is no conflict of interest regarding the publication of this article.


FUNDING

None.


CONSENT FOR PUBLICATION

Written informed consent was obtained from all the participants.


ETHICAL APPROVAL

Signed informed consent for participation and publication of medical details was obtained from the parents of this child. Confidentiality of patient’s data was ensured at all stages. The authors declare that ethics approval was not required for this case report.


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  23. Ridaura-Sanz C, López-Corella E, Salazar-Flores M. Exogenous lipoid pneumonia superinfected with acid-fast bacilli in infants: a report of nine cases. Fetal Pediatr Pathol. 2006;25(2):107–17. https://doi.org/10.1080/15513810600788798


How to Cite this Article
Pubmed Style

AlShamrani AS, Alzaid MA, Fadl SM, AlFaki MA. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudan J Paed. 2021; 21(1): 82-88. doi:10.24911/SJP.106-1587896995


Web Style

AlShamrani AS, Alzaid MA, Fadl SM, AlFaki MA. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. http://www.sudanjp.com/?mno=102259 [Access: May 13, 2021]. doi:10.24911/SJP.106-1587896995


AMA (American Medical Association) Style

AlShamrani AS, Alzaid MA, Fadl SM, AlFaki MA. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudan J Paed. 2021; 21(1): 82-88. doi:10.24911/SJP.106-1587896995



Vancouver/ICMJE Style

AlShamrani AS, Alzaid MA, Fadl SM, AlFaki MA. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudan J Paed. (2021), [cited May 13, 2021]; 21(1): 82-88. doi:10.24911/SJP.106-1587896995



Harvard Style

AlShamrani, A. S., Alzaid, . M. A., Fadl, . S. M. & AlFaki, . M. A. (2021) A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudan J Paed, 21 (1), 82-88. doi:10.24911/SJP.106-1587896995



Turabian Style

AlShamrani, Abdullah Saeed, Mohammed Abdullah Alzaid, Sarrah M. Fadl, and Mohammed Ahmed AlFaki. 2021. A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudanese Journal of Paediatrics, 21 (1), 82-88. doi:10.24911/SJP.106-1587896995



Chicago Style

AlShamrani, Abdullah Saeed, Mohammed Abdullah Alzaid, Sarrah M. Fadl, and Mohammed Ahmed AlFaki. "A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage." Sudanese Journal of Paediatrics 21 (2021), 82-88. doi:10.24911/SJP.106-1587896995



MLA (The Modern Language Association) Style

AlShamrani, Abdullah Saeed, Mohammed Abdullah Alzaid, Sarrah M. Fadl, and Mohammed Ahmed AlFaki. "A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage." Sudanese Journal of Paediatrics 21.1 (2021), 82-88. Print. doi:10.24911/SJP.106-1587896995



APA (American Psychological Association) Style

AlShamrani, A. S., Alzaid, . M. A., Fadl, . S. M. & AlFaki, . M. A. (2021) A case of infantile exogenous lipoid pneumonia with an unusual complication managed by modified whole lung lavage. Sudanese Journal of Paediatrics, 21 (1), 82-88. doi:10.24911/SJP.106-1587896995





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