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CASE REPORT |
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Year : 2013 | Volume
: 1
| Issue : 2 | Page : 62-64 |
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Congenital Bochdalek's hernia in a 18-year-old boy
Supriya Sarkar1, Arnab Maji1, Kaushik Saha2, Debraj Jash1
1 Department of Pulmonary Medicine, Nil Ratan Sarkar Medical College and Hospital, Kolkata, West Bengal, India 2 Department of Pulmonary Medicine, Burdwan Medical College and Hospital, Burdwan, West Bengal, India
Date of Web Publication | 18-Dec-2013 |
Correspondence Address: Arnab Maji Department of Pulmonary Medicine, Nil Ratan Sarkar Medical College and Hospital, 2 No Ichlabad, Barabenepara, P. O. Sripally, Burdwan - 713 103, West Bengal India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/2320-8775.123218
Adult presentation of congenital Bochdalek's hernia is rare. Here we report the case of a 18-year-old boy who presented to us with dyspnea, low grade fever, and cough for whom the diagnosis of congenital Bochdalek's hernia was confirmed by computed tomography (CT) scan of thorax after barium ingestion. Problems of this adult presentation because of difficulty in diagnosis, operative management, and rudimentary lung were properly and successfully dealt with in our case. We concluded that before undergoing operation in adult Bochdalek's hernia, thorough check-up is necessary as rudimentary lung may not expand resulting in ventilatory insufficiency during postoperative phase. Keywords: Adult presentation, Bochdalek′s hernia, congenital, congenital hernia, diaphragmatic hernia
How to cite this article: Sarkar S, Maji A, Saha K, Jash D. Congenital Bochdalek's hernia in a 18-year-old boy. J Assoc Chest Physicians 2013;1:62-4 |
Introduction | |  |
Congenital Bochdalek's hernia occurs due to defect in the posterior part of the diaphragm, which allows a free communication between the thoracic and abdominal cavities. It usually presents in neonates and preschool children who present with clinical symptoms caused by respiratory insufficiency. [1] Late presentation of Bochdalek's hernia is rare and sometimes misleads the clinicians. [2] We are reporting a case of Bochdalek's herniain an 18-year-old boy.
Case Report | |  |
An 18-year-old boy presented with low grade fever, exertional shortness of breath (MMRC grade 2), and dry cough for last 1 month. There was no history of hemoptysis or chest pain and he denied any history of chest trauma in the past. On general examination, the vitals were stable. On percussion, dullness was elicitedover left inframammary region, left infra-axillary region, and left infrascapular region. Breath sound was absent over the corresponding areas. Routine blood investigations were normal and sputum smears for acid fast bacilli were negative. His chest X-ray posteroanterior view showed homogenous opacity involving the left mid and lower zone with free left cardiophrenic angle [Figure 1]. Combined clinic-radiological picture was suggestive of encysted effusion on the left side. Since emergency ultrasonography (USG) facility was not available in our chest outpatient department and clinically we got features of pleural effusion on the left, we tried to aspirate pleural fluid from the left side but ended up in a dry tap. Planned thoracic USG on the next day revealed herniated gut loops with peristalsis in the left hemithorax. Barium meal study showed the presence of gut loops in the left hemithorax, but stomach and duodenum remained in the abdomen. Computed tomography (CT) scan of thorax after barium contrast ingestion with three-dimensional reconstruction revealed the gut loops (both small and large gut) extending into the left hemithorax andan underdeveloped left lung [Figure 2]. Thus, the diagnosis of Bochdalek's hernia was confirmed. Operative intervention to reposition the gut loops in the abdomen and repair the diaphragmatic vent was planned. The diaphragmatic vent was repaired, but due to lack of space in the abdominal cavity, gut loops could not be accommodated there, and the excess gut loops were kept on the anterior abdominal wall in a covered mesh [Figure 3]. Afterwards, a space was created in the abdomen to accommodate the gut loops and the whole procedure was completed in three stages. The boy had a stormy postoperative period in the form of postoperative ventilatory failure for which he was on mechanical ventilation for 7 days. Finally, after 2 months hewas discharged in a stable condition. | Figure 1: Chest X-ray posteroanterior view showing homogenous opacity involving the left mid and lower zone with free left cardiophrenic angle
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 | Figure 2: Computed tomography scan of the thorax after barium contrast ingestion with three-dimensional reconstruction showing hypoplastic lung (white arrow) and intestinal loops in left side of the hemithorax (black arrow)
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 | Figure 3: Thoracotomy showing abdominal content in the thoracic cavity and lack of space in the abdomen to accommodate the content
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Discussion | |  |
A Bochdalek's hernia, resulting from inadequate closure of the posterolateral pleuroperitoneal membrane, is the most commonly seen congenital diaphragmatic hernia. Defects are more commonly seen on the left side (70-90%) of the diaphragm with displacement of abdominal content like stomach, bowel loops, liver, spleen, or fat tissues into thoracic cavity. [3] In our case both large and small bowel loops were found as the content of hernia.
Bochdalek's hernia most commonly present during the first few weeks of life. Diagnosis beyond first 8 weeks of life occurs nearly in 5-25% of all Bochdalek's hernias. [4] After fetal life the most common age for its presentation ranges from neonatal life to preschooling age. [2] Around 10% of all reported cases of Bochdalek's hernia occur in adults. [3] The incidence of Bochdalek's hernia in routine CT scans has been estimated to vary from as low as 0.17% to as high as 6%. [3] In adult, most of them remain asymptomatic and thus finding of the hernias is incidental. [4] In adulthood, congenital diaphragmatic hernia can present with vague gastrointestinal and respiratory symptoms. [4] Our case presented exclusively with respiratory symptoms consisting of exertional breathlessness and dry cough. Acute presentation, though rare, may be present due to incarceration, obstruction, orstrangulation of the hernia which were absent in our case. [5]
Diagnosis is made by a combination of chest X-rays, CT scan, magnetic resonance imaging (MRI), as well as upper gastrointestinal and bowel double-contrast study. Chest X-ray may be normal or may simulate pleural effusion, hydropneumothorax, tension pneumothorax, atelectasis, pulmonary sequestration, and foreign body aspiration. [6] Investigation of choice remains CT scan of thorax with contrast ingestion which in our case confirmed the presence of diaphragmatic hernia. [7]
Management of a Bochdalek hernia includes reducing the abdominal contents and repairing the defect through a laparotomy or thoracotomy. Sometimes it needs step-wise reduction of abdominal contents because of less space in the abdomen which in our case took three steps to complete the whole procedure within a span of 10 days. The mortality rate for elective surgery has been reported at less than 4%, but may be as high as 32% in acute complicated states. [8]
In our case, the clues to the congenital nature of the hernia were postero-lateral location of the diaphragmatic vent on CT scan, underdeveloped left lung, andlack of space in the abdomen during surgery. Though Bochdalek's hernia rarely presents in adult, the awareness of the possibility should be kept in mind. The second problem is the lack of space in abdominal cavity that may lead to difficulties during operation and therefore, operation may have to be performed in stages. Last but not the least is the problem of rudimentary lung that may not develop if operation is delayed.
References | |  |
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[Figure 1], [Figure 2], [Figure 3]
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