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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 8  |  Issue : 4  |  Page : 200-202

Spontaneous pneumomediastinum and pneumopericardium in a child with severe diabetic ketoacidosis: A case report


DepartmentofPediatrics,BharatiVidyapeethMedicalCollegeandHospital,Pune,Maharashtra, India

Date of Submission26-Feb-2021
Date of Decision28-Apr-2021
Date of Acceptance07-May-2021
Date of Web Publication10-Jul-2021

Correspondence Address:
Dr. Bhakti U Sarangi
DepartmentofPediatrics,BharatiVidyapeethMedicalCollegeandHospital,Pune-411043,Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpcc.jpcc_21_21

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  Abstract 


Apart from being the most common endocrine emergency in the pediatric intensive care unit, some clinical manifestations of diabetic ketoacidosis such as vomiting and continued acidotic breathing may, on the rare occasion, lead to unexpected complications such as pneumomediastinum, pneumopericardium, pneumothorax, and subcutaneous emphysema. The differentials for the same include Hamman's syndrome secondary to forceful coughing or Valsalva maneuver, Boerhaave syndrome resulting from forceful vomiting, and even spontaneous air leaks due to severe swings in intrathoracic pressure during Kussmaul breathing, leading to alveolar rupture causing air leak. These complications may have subtle signs as well as a significant overlap in presentation and require a high index of suspicion to diagnose and careful evaluation to differentiate. We hereby report a 3-year-old child with new-onset type 1 diabetes mellitus in severe DKA, leading to pneumomediastinum and pneumopericardium where computed tomography of the thorax helped rule out differentials and the air leak resolved spontaneously with the resolution of acidosis.

Keywords: Air leak, Boerhaave syndrome, diabetic ketoacidosis, spontaneous pneumopericardium


How to cite this article:
Walimbe A, Shankar GH, Sarangi BU, Sharma V. Spontaneous pneumomediastinum and pneumopericardium in a child with severe diabetic ketoacidosis: A case report. J Pediatr Crit Care 2021;8:200-2

How to cite this URL:
Walimbe A, Shankar GH, Sarangi BU, Sharma V. Spontaneous pneumomediastinum and pneumopericardium in a child with severe diabetic ketoacidosis: A case report. J Pediatr Crit Care [serial online] 2021 [cited 2021 Sep 19];8:200-2. Available from: http://www.jpcc.org.in/text.asp?2021/8/4/200/321100




  Introduction Top


Diabetic ketoacidosis (DKA) is a routine endocrine emergency encountered in the pediatric intensive care unit associated with several metabolic complications, leading to morbidity and mortality, of which cerebral injury/edema remains a major contributor.[1] Although aspiration remains a potential respiratory complication, mechanical problems such as pneumomediastinum and pneumopericardium have only seldom been reported. Spontaneous air leaks such as pneumothorax in children remain a rarity, with majority of secondary cases attributed to underlying congenital lung malformations, infections, airway diseases, and connective tissue diseases.[2] Management in these situations ranges from close observation and oxygen supplementation to needle aspiration and surgical intervention.[2] We hereby report a 3-year-old girl with new-onset type 1 diabetes mellitus with severe DKA on presentation with a coexisting pneumomediastinum and pneumopericardium.


  Case Report Top


A 3-year-old girl was referred to us with a history of polyuria, polydipsia, and polyphagia ongoing for 15 days followed by deep and rapid breathing for the past 1 day. A chest radiograph was done by the referring clinician owing to the fast breathing and alleged abnormal breath sounds. On arrival, she was lethargic, tachycardic, clinically dehydrated, hyperglycemic (RBS – 537 mg/dL), and acidotic (pH – 6.9, HCO3 – 1.7 mmol/L) with Kussmaul breathing. With features of compensated shock, her examination otherwise was unremarkable. However, her chest radiograph from outside showed accumulation of air in the pericardial space [Figure 1]. She had no other significant medical history, particularly no respiratory disease, and no history of any cough, forceful emesis, or trauma. Bedside echocardiography did not reveal any abnormality with ventricular filling and contractility. A computed tomography (CT) of the thorax confirmed the presence of both pneumomediastinum and pneumopericardium with no other significant abnormality [Figure 1]. She was managed with the hospital protocol for DKA with fixed fluid and insulin infusion rates and responded well with an uneventful recovery. A repeat chest radiograph done on day 7 of stay showed a significant decrease in the air in the pericardial sac [Figure 2].
Figure 1: Chest radiograph and computed tomography thorax showing evidence of pneumomediastinum and pneumopericardium on day 1 of admission

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Figure 2: Posteroanterior and lateral chest radiographs before discharge showing resolving pneumomediastinum and pneumopericardium

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  Discussion Top


Pneumomediastinum, pneumopericardium, pneumothorax, and subcutaneous emphysema are the air leak syndromes reported rarely with DKA.[3] Presence of Hamman's syndrome, a rare condition characterized by the spontaneous simultaneous presence of a pneumomediastinum with subcutaneous emphysema, has been reported in adults with severe DKA confounding the physical findings and course of the illness.[4] Attributed mostly to forceful coughing in adults with airway diseases or postpartum women secondary to prolonged Valsalva maneuver, Hamman's syndrome typically presents with retrosternal chest pain, radiating to the back or neck, and may be associated with dysphagia, dysphonia, and dyspnea. A close differential diagnosis in patients with DKA may be Boerhaave syndrome, a classical triad of vomiting, abdominal or chest pain, and subcutaneous emphysema with the emphysema, resulting from a transmural tear of the distal esophagus induced by a sudden increase in intraluminal pressure secondary to forceful vomiting.[5] In our case, the child had neither cough nor vomiting and did not complain of chest pain or change in voice. The proposed pathophysiologic mechanisms for the presence of air leaks in patients with DKA remain obscure.[6] Following forceful vomiting, as maybe seen in DKA, there is an increase in intrathoracic pressures, secondary to the Valsalva maneuver. Intragastric air may be thrust into the mediastinum creating a pressure effect depending on the volume of air and the force of purging. Vomiting in DKA is caused by acidosis and further exacerbated by gastroparesis secondary to hyperglycemia. Other mechanisms have been ascribed to the expiratory effort and grunting associated with the hyperventilation secondary to ketoacidosis seen in DKA. Acidotic breathing with high tidal volumes as seen in severe DKA is said to increase the alveolar pressure 20–30 mmHg above normal inspiratory pressures, predisposing to alveolar rupture.[4] This mechanism predisposes to patient-self-inflicted lung injury where uncontrolled swings in transpulmonary pressure abnormally increase lung stress, worsen atelectrauma, predispose to negative pressure pulmonary edema, and even cause diaphragmatic injury.[7] The air from ruptured alveoli traverses the bronchovascular connective tissue planes into the mediastinum and hilum, leading to pneumomediastinum or pneumopericardium. Air may also track to the subcutaneous tissues of the neck, retroperitoneum or through posterior mediastinum and intervertebral foramen to the epidural space while rupture of the mediastinal pleura can also lead to pneumothorax. The CT thorax in our child ruled out the presence of pneumothorax and also the possibility of esophageal injury/tear and any pre-existing lung malformations/infection. The examination in Hamman's syndrome reveals a crunching or crackling precordial sound heard during systole on auscultation referred to as Hamman's sign and also presence of subcutaneous emphysema. Both of these were absent in our child on examination. The fact that there may be presence of any one or more or even none of the clinical symptoms and signs in this situation compounds the diagnosis of pneumomediastinum/pneumopericardium in a scenario where the clinician's mind is preoccupied with the ongoing metabolic disturbances. The overlap of hyperventilation and hemodynamic instability due to severe volume loss in a critically ill child also makes it harder to focus on rarer associations or complications such as pneumomediastinum. Moreover, chest radiographs are hardly ever done in children with DKA unless an obvious precipitating lung parenchymal problem is considered. To add to that, the subtle signs on a posteroanterior chest radiograph indicating free air in the mediastinum warrant substantial alertness from the clinician as well as the radiologist. All of the above put together may account for the failure to pick up these instances, with the true incidence of air leaks in DKA being under-diagnosed as compared to what has been reported.[6] Apart from Boerhaave syndrome, where the possibility of ensuing infection looms large, the air leak syndromes in patients with DKA resolve spontaneously with resolution of the acidosis. The same was seen in our child.

Apart from the usual metabolic complications, DKA may present with air leak syndromes including pneumomediastinum, pneumopericardium, pneumothorax, and subcutaneous emphysema. It is up to the clinician to carefully examine these children to elicit signs of such complications, with chest radiography being crucial to pick them up. In cases where esophageal rupture is suspected, a CT thorax is advisable.

Although the air leak syndromes in DKA are self-resolving and prognosis, in general, remains excellent, they may lead to added morbidity/mortality. A collective database/registry of children with DKA and associated complications may give more insight into the true incidence of this apparently rare complication.

Acknowledgments

We would like to acknowledge the Pediatric Intensive Care Team and Pediatric Endocrinology team at Bharati Vidyapeeth Medical College, Hospital and Research Centre, Pune.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient's parent has given consent for images and other clinical information to be reported in the journal. The patient's parent understands that the names and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Wolfsdorf JI, Glaser N, Agus M, Fritsch M, Hanas R, Rewers A, et al. ISPAD Clinical Practice Consensus Guidelines 2018: Diabetic ketoacidosis and the hyperglycemic hyperosmolar state. Pediatr Diabetes 2018;19 Suppl 27:155-77.  Back to cited text no. 1
    
2.
Kuo PY, Nong BR, Huang YF, Chiou YH. Primary spontaneous pneumothorax in children: A literature review. Pediatr Respirol Crit Care Med 2018;2:25-31.  Back to cited text no. 2
  [Full text]  
3.
Pereira O, Baburaj R, Abraham P. Spontaneous pneumomediastinum and pneumopericardium: A rare complication of diabetic ketoacidosis. Pract Diab Int 2004;21:300b.  Back to cited text no. 3
    
4.
Pain AR, Pomroy J, Benjamin A. Hamman's syndrome in diabetic ketoacidosis. Endocrinol Diabetes Metab Case Rep 2017;2017:17-0135.  Back to cited text no. 4
    
5.
Alkhuja S, Gazizov N, Charles G. Pneumomediastinum complicating diabetic ketoacidosis and Boerhaave's syndrome. Case Rep Med 2013;2013:598720.  Back to cited text no. 5
    
6.
Girard DE, Carlson V, Natelson EA, Fred HL. Pneumomediastinum in diabetic ketoacidosis: Comments on mechanism, incidence, and management. Chest 1971;60:455-9.  Back to cited text no. 6
    
7.
Grieco DL, Menga LS, Eleuteri D, Antonelli M. Patient self-inflicted lung injury: Implications for acute hypoxemic respiratory failure and ARDS patients on non-invasive support. Minerva Anestesiol 2019;85:1014-23.  Back to cited text no. 7
    


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  [Figure 1], [Figure 2]



 

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