|Year : 2021 | Volume
| Issue : 2 | Page : 151-152
Uncommon causes of a common presentation: Respiratory distress in children
Department of Pediatrics, Hamdard Institute of Medical Sciences and Research, Delhi, India
|Date of Submission||27-Mar-2021|
|Date of Decision||15-Apr-2021|
|Date of Acceptance||02-May-2021|
|Date of Web Publication||31-May-2021|
Dr. Nidhi Bedi
Department of Pediatrics, Hamdard Institute of Medical Sciences and Research, Delhi
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Bedi N. Uncommon causes of a common presentation: Respiratory distress in children. Indian Pediatr Case Rep 2021;1:151-2
Respiratory distress is a common presentation in pediatric patients. All of us are familiar with the usual underlying causes of distress and their management, with children responding to standard care in most cases. Occasionally, one may find a patient who does not respond and poses a diagnostic dilemma. We hereby present some case reports of uncommon causes of respiratory distress from the neonatal period to adolescence.
Healey D, Ron N, Hromada A, et al. Perinatal/neonatal case presentation: Pulmonary artery sling associated with respiratory distress. Springer Plus 2016;5. DOI 10.1186/s40064-015-1656-5.
A 34-week appropriate for gestational age male baby was born to fourth gravida and para one mother who had been covered with steroids before birth. The Apgar score was normal. The baby was admitted in neonatal intensive care unit for respiratory distress that developed immediately after birth and was associated with decreased air entry on examination. A chest X-ray showed left-sided pneumothorax and bilateral opacities suggesting respiratory distress syndrome. The baby was initiated on noninvasive positive pressure ventilation. His clinical status gradually improved over the next 72 h, and he was subsequently discharged in the next few days. At 2 months of age, the baby developed cough and wheezing which were diagnosed as a viral lower respiratory tract infection and treated as an outpatient. At 4 months, the child required admission for another episode of respiratory distress and wheezing. This time the diagnosis was made as bronchiolitis, due to respiratory syncytial virus infection. In-depth probing of history revealed that the baby had persistent tachypnea and feeding difficulties since birth. The X-ray showed hyperinflation, increased peribronchial markings and focal infiltrates in the right infrahilar region, suggestive of atelectasis. A barium contrast esophagogram was planned that displayed subtle contour change in the esophagus, above the level of carina. Careful re-examination identified a systolic murmur at the left upper sternal border. An echocardiography revealed a pulmonary artery sling with the left pulmonary artery arising from right pulmonary artery. The ventricular function was good with mild hypoplasia of the left pulmonary artery.
Disu EA, Kehinde OA, Anga AL, et al. Congenital pulmonary airway malformation: A case report of a rare cause of neonatal respiratory distress and review of literature. Niger J Clin Pract 2019;22:1621-5.
A newborn was referred to a hospital on the 13th day of life as a case of persistent pneumonia for the 3rd day after birth. The baby was born to a 34-year-old primigravida with no significant antenatal or perinatal history. The birth weight was 3.1 kg. On examination, the baby had a respiratory rate of 100/min, severe respiratory distress with intercostal and subcostal recessions, bulging anterior chest wall, hyperresonant right hemithorax, and bronchovesicular breath sounds which were reduced in the lower half of the right posterior hemithorax. All other systems were normal. X-ray chest showed leftward shift of the mediastinum, hyperlucency of the right lower lobe with reduced lung markings, and haziness of the right upper and middle lobes. Differentials of congenital lobar emphysema and congenital pneumonia were kept. A computed tomography scan of chest done showed a multicystic mass in the right lower lobe suggestive of congenital cystic adenoid malformation. Other tests including abdominal ultrasound scan and barium meal with follow-through were normal.
Thakur N, Agarwal D, Narayan S, et al. Recurrent pneumonia in an infant with an esophageal lung. Indian Pediatr 2020;57:266-7.
A 7-month-old girl baby presented with respiratory distress for a week that had not responded to a short course of antibiotics. There was a significant history of episodes of choking during feeding recurrent lower respiratory infections. At admission, the infant had tachypnea, tachycardia, chest retractions, and decreased breath sounds on the right side of the chest. The apex beat was localized to the right, suggestive of dextrocardia. Investigations revealed neutrophilic leukocytosis and a positive C-reactive protein. Chest X-ray showed hazy right hemithorax with right side mediastinal shift. This prompted a computed tomography chest which demonstrated right lung hypoplasia with cystic bronchiectatic changes and nonvisualization of the right main bronchus, hypoplastic right main pulmonary artery, and an abnormal bronchoesophageal communication. A barium swallow study showed filling of right main bronchus directly from the esophagus. Rigid bronchoscopy showed a blind-ended right bronchial stump. The final diagnosis was esophageal lung. Ultrasound abdomen and echocardiography were normal. The patient improved on antibiotics and supportive care. Parents were advised surgical intervention for the underlying condition.
Ahmed H, Ndiaye C, Barry MW, et al. A rare cause of upper airway obstruction in a child. Case Rep Otolaryngol 2017;2017265.
A 4-year-old girl was hospitalized with a history of difficulties in breathing for a year. The nature of dyspnea had progressively changed from intermittent episodes to persistent respiratory distressed that had worsened for a week before presentation. Salient examination findings included a hyperextended neck and Stage 4 laryngeal obstruction. Direct laryngoscopy showed a round cyst, with vascular markings on the wall, originating in the left ventricle, encroaching on the root of the epiglottis, and completely blocking the vocal cords. Thick mucoid fluid was seen on incision of the cyst, which was then marsupialized. The final diagnosis was a ductal ventricular band cyst. Epiglottic and ventricular band cysts are retention-type cysts that occur due to chronic inflammation and lead to blockage of mucus glands. Such type of chronic inflammation happens during episodes of superinfection or laryngeal trauma in sick patients requiring multiple intubations.
Powell AW, Hanke S, Tweddell JS, et al. A 14-year-old boy with unusual presentation of respiratory distress. Case Rep Pediatr 2016;7313942.
A 14-year-old boy was hospitalized with sudden episode of respiratory distress for 6 h before admission. There was a history of associated cough, congestion, high-grade fever, fatigue, myalgia, and vomiting. He was a known case of mild persistent asthma who usually responded to albuterol/salbutamol by metered-dose inhalers. However, this time there had not been any response. Oxygen saturation was low, varying from 70' to 80'. Air entry was decreased bilaterally. There was no audible wheeze. Suspecting an acute exacerbation of asthma, initial management included albuterol-ipratropium nebulization and injection methylprednisolone, to no avail. Chest X-ray revealed extensive airspace disease bilaterally which appeared such as multifocal pneumonia or pulmonary edema. Salient investigations revealed respiratory acidosis, leukocytosis with left shift, negative blood culture, and respiratory viral testing. Despite initiation of noninvasive respiratory support by BiPAP with 100' FiO2, the child developed hypotension and shock. He was intubated and started on inotropes, antibiotics (vancomycin and ceftriaxone), and antiviral agents (Tamiflu). Frothy drainage in the endotracheal tube and requirement of high ventilator pressures were noted, consistent with diffuse pulmonary edema. An echocardiogram revealed cor triatriatum with a membrane separating the left atrium into two chambers. Doppler flow demonstrated the pulmonary veins entering the left atrium on the proximal side of the atrial membrane. Right ventricular hypertension (pressure estimated to be greater than half the systemic blood pressure) was noted. A transesophageal echocardiogram confirmed the presence of severely restrictive cor triatriatum for which was emergent resection of the cor triatriatum membrane was done.
Duke C, Alexander K, Hageman JR. An unusual cause of respiratory distress in a 17-year-old boy. Pediatr Ann 2014;43:20-3.
A 17-year-old boy was being treated as a viral syndrome in view of frontal headache, body aches, neck pain, and stiffness for 5 days and fever for 4 days. Worsening, in the form of nonbilious emesis and nonbloody diarrhea, with notable increase in neck pain, prompted the parents to get him hospitalized. He was dehydrated and appeared ill. There was no history of sore throat, chest pain, or abdominal pain. An urgent chest radiograph was done which was suggestive of bilateral lower lobe pneumonia. The patient was treated empirically with ceftriaxone, vancomycin, and azithromycin. Subsequent blood culture was negative. Lumbar puncture result showed only few red blood cells in cerebrospinal fluid. In spite of the above child developed hypoxia, following day for which oxygen support was escalated and the patient was referred to higher center. Subsequently, as the child did not maintain 10 l of oxygen, he was shifted on BiPAP. Respiratory examination revealed decreased breath sounds at bases bilaterally and coarse breath sounds in remaining lung fields with intermittent crackles. Transverse leukonychia was noted. Repeat chest radiograph showed bilateral lower lobe opacities more than the previous one. Laboratory reports showed neutrophilia, thrombocytopenia, highly raised CRP (229 mg/L), and hypoalbuminemia. Complement levels C3 (30 mg/dL) and C4 (5 mg/dL) were low. Respiratory viral panel was negative. Additional laboratory studies included tuberculosis interferon release test, cytomegalovirus polymerase chain reaction, Epstein–Barr virus capsid antibody, HIV antibody, histoplasma serum/urinary antigen, Bartonella antibody, and pneumococcal urinary antigen which came negative on follow-up. A computed tomography of the head, neck, and chest done which showed bilateral partial thrombosis of the internal jugular veins, more extensive on the right than the left; opacification of the right ethmoid air cells and sphenoid sinus (air-fluid level in the maxillary sinus was consistent with acute sinusitis); adjacent broken molar with a periodontal abscess; and images of the lung apices showed bilateral multifocal air space opacities and mediastinal lymph nodes. The child was intubated due to worsening status. Some other rheumatology tests done including anticardiolipin, antiextractable nuclear antigens, anti-SSA or B antibodies, vascular antineutrophil cytoplasmic antibodies, antinuclear antibodies, and anti-DNA native double-stranded antibodies were all negative. The patient was given anticoagulant therapy with enoxaparin to treat the internal jugular venous thromboses. Laboratory studies of antithrombin III, lupus anticoagulant (double-Russell's viper venom time), activated protein C resistance, factor V Leiden, protein C, protein S, and homocysteine were all within normal limits. The patient was diagnosed with atypical Lemierre's syndrome; however, an organism was not isolated from the blood culture, and the periodontal abscess was the most likely source of infection.
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