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Introduction

Recurrent chest infections and wheezing in children may have a wide differential diagnosis; one of these etiologies include an intrathoracic mass that compresses the lungs and could have delayed presentation. In our reported case, we will present a case of a five year old boy with left diaphragmatic hernia which was discovered incidentally on the repeat chest X-ray for control of bilateral basal consolidation pneumonia. He was operated successfully without any complications. This case enlightens us to keep congenital diaphragmatic hernia as one of the differential diagnosis of recurrent chest infections. 

Case Report

A six year old boy was referred to our hospital for persistent signs of respiratory tract infection that included high grade fever, severe cough, dyspnea and decrease oral intake. Initially, he was treated at home by oral antibiotics:  cefpodoxime and azithromycin, along with ventorlin solution for nebulization. No improvement in primary clinical condition was seen after three days of treatment. Subsequently he was admitted to pediatric floor where an antero-posterior chest X-ray revealed bilateral basal consolidation. His inflammatory markers were not significantly increased, therefore he was given intravenous ceftriaxone at 50 mg/kg/dose twice daily for the presumed pneumonia. He continued to have persistent fever.  Oral oseltamivir was added twice daily. His fever responded on second day.

    In his past medical history, he was term neonate, by cesarean section, have had mild neonatal respiratory distress presumed to be due to transient tachypnea of the newborn, with no significant chest X-ray findings. He also have a seasonal reactive airways disease treated by inhaled b2 agonist and inhaled corticosteroid when needed. 

    A repeat chest X-ray performed on the fourth day of admission, the AP view did not showed any radiologic improvement [Fig.1], but the lateral view revealed a hypolucent lesion behind the sternum extending from the abdomen [Fig.2]. CT scan of the chest confirmed the presence of a left diaphragmatic hernia [Fig.3]. Next day the baby underwent a primary surgical repair through longitudinal laparotomy successfully without any post-operative complication and the follow-up chest X-ray showed complete resolution of the previous chest lesions [Fig.4]. He was discharged home two days post-surgery in a stable condition.


Discussion

CDH is a developmental disorder characterized by abnormal herniation of intestinal contents into the thoracic cavity due to absence or incomplete closure of the pleuroperitoneal canals on either side. The most common type is posterolateral defect called Bochdalek hernia. Herniated contents may include stomach, intestines, liver, or spleen [1]. The incidence of CDH ranges from 0.08 to 0.45 per 1,000 births. Eighty-five percent of defects are left-sided, 13% are right-sided, and 2% are bilateral. Most studies have found an equal representation of genders, although a 1.25 male-to-female ratio was reported in one large population-based study [2].

    Most infants with CDH are symptomatic immediately after birth. Much less frequently, CDH may present days, weeks, or even years after birth [3]. Early symptoms are severe respiratory distress, expiratory grunting, or respiratory arrest, most often accompanied by a scaphoid abdomen. Chest radiographs reveal mediastinal shift and intestinal contents in the thorax. Occasionally, the chest radiograph appears normal [4]. CDH in infants or older children may present with recurrent chest infections, acute respiratory distress, recurrent vomiting, diarrhea, constipation, or failure to thrive with late CDH presentation, radiographic findings may be misdiagnosed as pneumonia, pneumothorax, cystic lung bullae, or volvulus [5]. As in our reported case, the neonatal admission did not revealed any abnormal finding on the first chest X-ray performed and despite the history of recurrent chest wheezing and bronchitis that the patient suffered he was responding very well to the appropriate asthma treatment available but in his current condition, he was seen after a period of two years of absenteeism during which parents did not report any complicated disease except for the unusual fatigue he recently felt. The persistent pneumonia findings on the chest X-ray promoted us to request a lateral view on the repeat imaging that confirmed the bowel herniation into the left chest cavity that could most probably occurred after the neonatal period, which explain the delayed presentation of this not to miss diaphragmatic hernia.

    Standard chest X-ray is considered the first-line diagnostic imaging tool. The literature indicates that plain chest X-ray is diagnostic in 73% of patients with diaphragmatic hernia. The signs of diaphragmatic injury on plain radiographs are intrathoracic herniation of abdominal viscera, marked elevation of the hemi-diaphragm, distortion of the diaphragmatic margin and contralateral mediastinal shift [6].

    Postnatal management strategies include delivery in a tertiary center, immediate intubation after birth, and avoidance of high inflation pressure ventilations [7]. The goals of ventilation include correcting hypoxemia and acidosis, and minimizing barotrauma. In severe, persisting pulmonary hypertension and intractable respiratory failure, nitric oxide and/or ECMO are recommended; definite treatment is surgical repair, usually performed 48 hours after delivery after stabilization [7].

    Survival with modern-day management has increased to 90% [7]. Mortality is higher with bilateral CDH, prematurity, severe pulmonary hypertension, and use of ECMO. Long-term morbidity from CDH includes pulmonary hypoplasia, pulmonary hypertension, chronic lung disease, oxygen dependence, gastrointestinal dysmotility, swallowing dysfunction, neurocognitive deficits and bronchial hyper-reactivity [8].

Conclusion

Delayed presentation of a congenital diaphragmatic hernia must be kept in mind whenever dealing with a child with difficult to resolve pneumonia or history of recurrent chest infection. It is always recommended to order a two-view chest X-ray in order to increase its sensitivity, however antenatal diagnosis  must be made initially to arrange the in utero transfer of the baby to be delivered in a tertiary care hospital that offer the most advanced medical support required and for the final surgical repair.

References

1. Aliva De, Stein JE, Thompson M, Perez I. Persistent Pneumonia in an Infant. Keeping an Open Mind. Annals of the American Thoracic Society 2013; pp. 514-517.
2. Meurs KV Short BL, Congenital Diaphragmatic Hernia: The Neonatologist’s Perspective, Pediatrics in Review. 1999; 20:e79-e87.
3. Mei-Zahav M, Solomon M, Trachsel D, Langer JC. Bochdalek diaphragmatic hernia: not only a neonatal disease. Arch Dis Child. 2003; 88:532-535.
4. Gleeson F, Spitz L. Pitfalls in the diagnosis of congenital diaphragmatic hernia. Arch Dis Child. 1991;66:670-671.
5. Berman L, Stringer D, Ein SH, Shandling B. The late-presenting pediatric Bochdalek hernia: a 20-year review. J Pediatr Surg. 1988;23:735-739
6. Silker CW. Imaging of diaphragma injuries. Radiol Clin N Am. 2006;44:199-121.
7. Muratore CS, Wilson JM. Congenital diaphragmatic hernia: where are we and where do we go from here? Semin Perinatol. 2000;24:418-428.
8. Abel RM, Bush A, Chitty LS, Harcourt J, Nicholson AG. Congenital lung disease. In: Wilmott RW, Boat TF, Bush A, et al. (eds). Kendig and Chernick’s disorders of the respiratory tract in children, 8th ed. Philadelphia: Saunders Elsevier; 2012. pp. 317-357.