Rom J Morphol Embryol 2015, 56(3):1191–1194

ISSN (print) 1220–0522 ISSN (on-line) 2066–8279

CCAASSEE RREEPPOORRTT

Aspiration pneumonia in an infant with neurological sequelae – case report

DALIA DOP1), CRISTIAN GHEONEA1), GEORGETA LIGIA STĂNESCU1), ARITINA-ELVIRA MOROŞANU1), RADU DIACONU1), ELENA CARMEN NICULESCU1), MARIA LIVIA OGNEAN2), DRAGOŞ NICULESCU3)

1)Department of Pediatrics, Faculty of Medicine, University of Medicine and Pharmacy of Craiova, Romania 2)Department of Pediatrics, “Victor Papilian” Faculty of Medicine, “Lucian Blaga” University of Sibiu, Romania 3)Department of Orthopedics and Traumatology, University of Medicine and Pharmacy of Craiova, Romania

Abstract Aspiration pneumonia is a frequent cause of morbidity and mortality in children with neurological deficits. We present the case of a 4-month-old infant from the Foster Care Center, with severe psychomotor retardation, blindness, and associated cardiac malformation, who was admitted to the Pediatrics Clinic of the Emergency County Hospital of Craiova, Romania, presenting aspiration pneumonia and moderate respiratory insufficiency. Under sustained, early instituted treatment, the evolution was towards death. The chest radiography and histo-pathological examination of the pulmonary tissue confirmed the diagnosis. The neurological impairment was not only a favoring factor for aspiration, through the deglutition disorders, but it was also an aggravating one, through the bacterial colonization of the lungs.

Keywords: aspiration pneumonia, neurological impairment, mortality increase.

Introduction

The incidence of aspiration syndromes associated to anatomical or neurological disorders is unknown. Aspira-tion pneumonia represents a frequent cause of morbidity and mortality in children with neurological deficits [1–3]. Infants and children, who presented pulmonary impairment secondary to prematurity or respiratory distress syndrome, are more vulnerable than those with healthy, unaffected lungs [4]. Neurological impairment secondary to neonatal hypoxia or the presence of congenital cardiac malforma-tions constitute predisposing factors for pulmonary aspi-ration [5].

Although, in the last years, progress has been made in understanding the pathogenic mechanisms that are at the basis of dysphagia and the protection reflexes of the airways, and new diagnostic techniques have been intro-duced, many children are diagnosed and treated for aspi-ration only after the occurrence of pulmonary impairment [6, 7].

The aim of this paper is to present a case of aspiration pneumonia that affected an institutionalized child with malnutrition, neurological deficit and congenital heart malformation, who was diagnosed and treated in our institution. We discuss the case presentation, clinical, radiological and especially the histological diagnosis, and also the management for this condition.

Case report

The authors present the case of a 4-month-old male infant, who was admitted to the Pediatrics Clinic of the Emergency County Hospital of Craiova, Romania, for inefficient frequent spasmodic cough, cyanosis and respi-ratory difficulties with an onset of a few hours after feeding. The child came from a teenage mother (15-year-

old), he was born prematurely, naturally delivered, and was abandoned in the maternity ward.

We have carried out the paraclinical investigations (complete blood count, glycemia, urea, creatinine, trans-aminases, acid–base balance, serum electrolytes, serum iron), cultures, heart and chest radiography, heart ultra-sound, pneumology and phthisiology examination, ophthal-mology and neurological examination, and we have insti-tuted the appropriate therapy.

After death occurred, the material sampled from the lungs was subjected to a histopathological examination through the traditional paraffin inclusion method, followed by Hematoxylin and Eosin (HE) staining, after fixation with 10% formalin.

The patient named T.I.M. presented, upon admission, inefficient frequent spasmodic cough, cyanosis of the peri-oronasal region and extremities, signs of respiratory insufficiency (grunts, expiratory dyspnea, tachypnea, intercostal and subcostal retraction). The onset of the symptoms had been 5–6 hours before, with frequent spasmodic cough and difficult feeding.

The child came from a teenage mother. The birth began spontaneously at 35 weeks of gestation, with pelvic presentation, and prolonged labor that caused perinatal hypoxia. At birth, the child’s weight was 2150 g, the height was 46 cm, the head circumference was 30 cm, and the Apgar score at one minute and at five minutes was 5. The prolonged perinatal hypoxia caused right lung atelec-tasis and intraventricular hemorrhage; in the end, it led to significant neurological and motor sequelae (microcephaly, blindness, generalized muscle hypotonia, mental retarda-tion).

Physical examination on admission: moderate general condition, deficient nutritional status, pale skin with diminished elasticity, fever 38.40C, cyanosis of the peri-

R J M ERomanian Journal of

Morphology & Embryologyhttp://www.rjme.ro/

Dalia Dop et al.

1192

oronasal region and extremities, inefficient frequent spasmodic cough, tachypnea with intercostal retraction, the pulmonary auscultation reveals diminished vesicular murmur in the right hemithorax, rounded abdomen, vomiting, bilateral cryptorchidism, microcephaly, blind-ness, generalized muscle hypotonia, sleepiness.

The patient was subjected to oxygen therapy, intra-venous perfusion for water, electrolyte and acid–base balance, antibiotic (Sulcef), bronchodilator and sympto-matic treatment.

The first investigations carried out revealed alterations of the complete blood count (anemia, leukocytosis, and thrombocytopenia), of the serum electrolytes, and hypo-calcemia (Table 1).

Table 1 – Paraclinical data

Parameter Initial value Evolution value

Hemoglobin [g/dL] 10.4 5.2

Platelets [No./mm3] 125 000 62 000

Leukocytes [No./mm3] 17 600 27 000

Serum iron [μg/dL] 25 –

Na+ [mmol/L] 121 130

K+ [mmol/L] 5.4 3.2

Blood calcium [mg/dL] 7.6 –

Urea [mg/dL] 12 55

Creatinine [mg/dL] 0.29 1.8

AST [IU] 28 88

ALT [IU] 14 76

TB [mg/dL] 0.17 1

AST: Aspartate transaminase; ALT: Alanine transaminase; TB: Total bilirubin.

The chest X-ray revealed the presence of a homogenous opacity in the middle region of the right lung field and the increase in size of the cardiac silhouette (Figure 1).

The heart ultrasound revealed the presence of an ostium secundum (inter)atrial septal defect and the persistence of the arterial duct.

The pneumology, phthisiology and pediatric exami-nation raised the suspicion of aspiration pneumonia – paramedian and right superior lobe pneumonia with an atelectatic component.

The ophthalmology examination revealed bilateral blindness.

During the hospitalization period, the general condi-tions deteriorated, fever and the respiratory difficulties

persisted, the water, electrolyte and acid–base imbalances were maintained, and there was also registered severe anemia, thrombocytopenia and signs of multi-organ dys-function. The culture collected from the tracheal and bronchial secretion aspirated revealed the presence of Klebsiella pneumoniae.

We continued with the water, electrolyte and acid–base rebalancing, and we instituted repeated transfusions of isogroup isoRh erythrocyte and platelet mass, Aminoven, antibiotic treatment (Cefort, Zyvoxid, Medaxone). The general conditions of the patient continued to gradually deteriorate, with limited response to the administered therapy; on the 7th day since admission, the patient died.

Figure 1 – Radiography revealed the presence of a homogenous opacity in the middle region of the right lung field and the increase in size of the cardiac silhouette.

The histopathological analysis of the lungs indicated massive interstitial stasis, with blood extravasations and intra-alveolar macrophages, as well as areas of edematous alveolitis (Figures 2–4).

We could also observe emphysematous areas, with breaks in the alveolar septa and the appearance of air spaces of variable sizes, as well as the presence of areas of alveolar collapse. In the bronchioles, we discovered focal ulcerations, epithelial scaling and peribronchial focal lymphocyte accumulations (Figure 5).

In the liver, we discovered sinusoid, centrilobular and portal stasis, as well as steatosis loading of the periportal hepatocytes (Figure 6). In the brain, we could observe meningeal and intraparenchymal stasis, cerebral edema and blood extravasations (Figure 7).

Figure 2 – Interstitial stasis and edematous alveolitis (HE staining, ×100).

Figure 3 – Intra-alveolar hematic extravasations and hemosideremic deposits (HE staining, ×100).

Aspiration pneumonia in an infant with neurological sequelae – case report

1193

Figure 4 – Macrophages at septum and intra-alveolar level (HE staining, ×200).

Figure 5 – Multinucleate exudates and macrophages at bronchiolar level (HE staining, ×100).

Figure 6 – Sinusoidal and portal stasis and liver steatosis (HE staining, ×100).

Figure 7 – Intra-parenchymatous stasis at brain level (HE staining, ×100).

Discussion

In the presented case, aspiration pneumonia affected an institutionalized child who presented a number of risk factors: prematurity, malnutrition, neurological deficit, congenital heart malformation; each of these factors increased the risk of morbidity and mortality.

Aspiration pneumonia is the consequence of food and secretions from the oral cavity, the upper respiratory tract or the stomach making their way into the lungs, and it manifests when the reflex defense mechanisms of the air-ways disappear, especially when dysphagia is also present [1]. The result of aspiration is the colonization of the lungs by the bacteria present in the upper respiratory tract and in the oral cavity, which leads to atelectasis infiltrates, abscess, empyema, sepsis, shock and, in severe forms, death [8, 9].

Studies have shown that, from a bacteriological point of view, just as in the case of adults, anaerobic bacteria are more frequently involved in the production of aspira-tion pneumonias, especially Gram-positive cocci (Pepto-streptococcus), fusobacteria and bacteroides fragilis. When aspiration occurs in institutionalized children, the noso-comial germs are usually aerobic or, at times, aerobic ones (alpha-hemolytic streptococcus, Escherichia coli, K. pneumoniae, etc.) [10, 11].

There is no standard diagnosis test for most types of aspiration; therefore, the differential diagnosis in children remains a challenge. The diagnosis for aspiration is clinical

and anamnestic, and certain diagnosis-supporting evalua-tions are added to it [complete blood count, heart and chest radiography, bacterial cultures, CT (computerized tomo-graphy) scans, endotracheal aspirate]. The chest radio-graphy is an indicator for the presence of pulmonary disease, with a characteristic distribution of lung aspiration, as well as for the progression or resolution of the process in time. Radiological alterations are present in all children with foreign body aspiration [12]. In the studied case, the diagnosis was suggested by the patient’s medical history (sudden onset in an institutionalized child with neurological deficits and secondary malnutrition) and the physical examination, and it was supported by the paraclinical data, the characteristic pleural and pulmonary radiography and the culture collected from the tracheal and bronchial secretions aspirate.

In a study conducted in Pakistan on 107 children with pulmonary aspiration, neurological disorders were in present in 29% of them, 5.6% presented asphyxia at birth and 10% were premature. The most frequent causes of pulmonary aspiration were caused by milk (31.8%) and oral secretions (19.6%); these children were also the ones who needed a longer hospital stay and recorded a higher percentage of mortality [13].

Pulmonary aspiration was the most frequent cause of recurrent/persistent pneumonia in children (17.7%), in a study conducted in Egypt [14].

Eating difficulties and malnutrition are constantly

Dalia Dop et al.

1194

present in children with disabilities because of the pre-sence of chewing and swallowing dysfunctions, spasticity, anorexia, vomiting and recurrent infections. Malnutrition causes, in its turn, the alteration of growth and damages to all organs and systems, especially the neurological and immune ones. Therefore, a multidisciplinary team (neurologist, gastroenterologist, and dietician) is needed to determine the presence of eating disorders and motor and neurological deficits, and to constantly reevaluate the feeding [15, 16]. Eating disorders in children with disabi-lities are associated to an increase in morbidity and mor-tality secondary to gastroesophageal reflux, dumping syn-drome or aspiration [16]. Studies show that approximately 37–40% of children who present eating disorders are pre-mature [17, 18], while the prevalence of eating disorders in children with neurological damage is estimated between 33 and 80% [17, 19].

Patients with massive aspiration have a 25% mortality rate [20]. In a study conducted by Kohda et al. (1994), 2/3 of the patients with neurological disorders present repeated pulmonary aspirations in their history [21], while in Sheikh et al. study approximately 12% of the 112 neuro-logically normal children with chronic respiratory symptoms presented swallowing dysfunctions and chronic silent aspi-ration, with no evidence of gastroesophageal reflux [22].

From a histopathological point of view, the most suggestive aspect for aspiration pneumonia is the presence in the lung of multinucleated giant cells, or even the pre-sence of foreign body granulomas or lymphocyte accumu-lations [23]. At the same time, acute inflammatory aspects can be present both in the alveoli and the bronchioles [24]. This was also revealed in our case, as the histopathological analysis confirmed the suspicion of pulmonary aspiration raised by the patient’s medical history, by the clinical examination and by the chest radiography.

Conclusions

In the case of our patient, prematurity, pelvic pre-sentation and prolonged labor caused perinatal hypoxia, which led to neurological sequelae. Pulmonary aspirations occurred on the background of severe malnutrition secon-dary to eating disorders characteristic for children with neurological disorders. It is necessary to carefully follow, detect and appropriately treat, as the case may be, the nutritional problems, considering their impact on growth, quality of life and increase in the risk of mortality, especially in very small children (less than one year old). Aspiration pneumonia represents a frequent cause of morbidity and mortality in children who present neuro-logical deficits. The result of aspiration is the colonization of the lungs by bacteria, which leads to atelectasis infil-trates, abscess, sepsis, shock and, in severe forms, death.

Conflict of interests The authors declare that they have no conflict of

interests.

References [1] Tutor JD, Gosa MM. Dysphagia and aspiration in children.

Pediatr Pulmonol, 2012, 47(4):321–337. [2] Weir K, McMahon S, Barry L, Ware R, Masters IB, Chang AB.

Oropharyngeal aspiration and pneumonia in children. Pediatr Pulmonol, 2007, 42(11):1024–1031.

[3] Morton RE, Wheatley R, Minford J. Respiratory tract infections due to direct and reflux aspiration in children with severe neurodisability. Dev Med Child Neurol, 1999, 41(5):329–334.

[4] Cornfield DN. Acute respiratory distress syndrome in children: physiology and management. Curr Opin Pediatr, 2013, 25(3): 338–343.

[5] Weir KA, McMahon S, Taylor S, Chang AB. Oropharyngeal aspiration and silent aspiration in children. Chest, 2011, 140(3):589–597.

[6] de Benedictis FM, Carnielli VP, de Benedictis D. Aspiration lung disease. Pediatr Clin North Am, 2009, 56(1):173–190, xi.

[7] Boesch RP, Daines C, Willging JP, Kaul A, Cohen AP, Wood RE, Amin RS. Advances in the diagnosis and management of chronic pulmonary aspiration in children. Eur Respir J, 2006, 28(4):847–861.

[8] Frakking TT, Chang AB, O’Grady KA, Walker-Smith K, Weir KA. Cervical auscultation in the diagnosis of oropharyngeal aspi-ration in children: a study protocol for a randomised controlled trial. Trials, 2013, 14:377.

[9] Weir K, McMahon S, Chang AB. Restriction of oral intake of water for aspiration lung disease in children. Cochrane Database Syst Rev, 2005, 4:CD005303.

[10] Brook I. Anaerobic pulmonary infections in children. Pediatr Emerg Care, 2004, 20(9):636–640.

[11] Brook I, Finegold SM. Bacteriology of aspiration pneumonia in children. Pediatrics, 1980, 65(6):1115–1120.

[12] Tenjović D, Petrović S, Dautović GV, Lovrenski J, Rodić BB. Clinical and radiological characteristics of unrecognised foreign body aspiration into respiratory tract in children. Med Pregl, 2013, 66(5–6):214–219.

[13] Karim RM, Momin IA, Lalani II, Merchant SS, Sewani AA, Hassan BS, Mahmood N. Aspiration pneumonia in pediatric age group: etiology, predisposing factors and clinical outcome. J Pak Med Assoc, 1999, 49(4):105–108.

[14] Saad K, Mohamed SA, Metwalley KA. Recurrent/persistent pneumonia among children in Upper Egypt. Mediterr J Hematol Infect Dis, 2013, 5(1):e2013028.

[15] Trier E, Thomas AG. Feeding the disabled child. Nutrition, 1998, 14(10):801–805.

[16] Sullivan PB. Gastrointestinal problems in the neurologically impaired child. Baillieres Clin Gastroenterol, 1997, 11(3):529–546.

[17] Bae SO, Lee GP, Seo HG, Oh BM, Han TR. Clinical charac-teristics associated with aspiration or penetration in children with swallowing problem. Ann Rehabil Med, 2014, 38(6):734–741.

[18] Rommel N, De Meyer AM, Feenstra L, Veereman-Wauters G. The complexity of feeding problems in 700 infants and young children presenting to a tertiary care institution. J Pediatr Gastroenterol Nutr, 2003, 37(1):75–84.

[19] Linscheid TR. Behavioral treatments for pediatric feeding disorders. Behav Modif, 2006, 30(1):6–23.

[20] Yoshikawa H, Yamazaki S, Abe T. Acute respiratory distress syndrome in children with severe motor and intellectual dis-abilities. Brain Dev, 2005, 27(6):395–399.

[21] Kohda E, Hisazumi H, Hiramatsu K. Swallowing dysfunction and aspiration in neonates and infants. Acta Otolaryngol Suppl, 1994, 517:11–16.

[22] Sheikh S, Allen E, Shell R, Hruschak J, Iram D, Castile R, McCoy K. Chronic aspiration without gastroesophageal reflux as a cause of chronic respiratory symptoms in neurologically normal infants. Chest, 2001, 120(4):1190–1195.

[23] Yousem SA, Faber C. Histopathology of aspiration pneumonia not associated with food or other particulate matter: a clinico-pathologic study of 10 cases diagnosed on biopsy. Am J Surg Pathol, 2011, 35(3):426–431.

[24] Mukhopadhyay S, Katzenstein AL. Pulmonary disease due to aspiration of food and other particulate matter: a clinico-pathologic study of 59 cases diagnosed on biopsy or resection specimens. Am J Surg Pathol, 2007, 31(5):752–759.

Corresponding author Maria Livia Ognean, Associate Professor, MD, PhD, Department of Pediatrics, “Victor Papilian” Faculty of Medicine, “Lucian Blaga” University, 2A Lucian Blaga Street, 550169 Sibiu, Romania; Phone +40745–276 584, e-mail: drliviaognean@yahoo.com Received: March 26, 2015 Accepted: November 24, 2015