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Posterolateral Congenital Diaphragmatic Hernia with Acute Gastric Volvulus and Splenic Herniation
 
Gaurang C Shah*, Piyush I Patel*, GN Shukla**, Dharmendra K Shah***, Anand A Trivedi+
 
The congenital diaphragmatic hernia (CDH) causing herniation of intra-abdominal viscera into the thorax presents usually in neonatal and early infantile period. They have respiratory distress at birth, which accounts for very high mortality. Delayed presentation of CDH occurs in 5-25% of cases from first few years of life upto adulthood with chronic respiratory or gastrointestinal symptoms. Plain and contrast radiological studies are essential for correct diagnosis. We report an extremely rare case of 8-year-old male child previously asymptomatic presented with acute abdominal pain. Plain and upper GI contrast study showed eventration of diaphragm with organoaxial type of gastric volvulus. Emergency laparotomy revealed hernia of Bochdalek with acute gastric volvulus (organoaxial) with herniation of omentum, small bowel, transverse colon, splenic flexure and spleen. Postoperative course was uneventful. High index of suspicion is required for correct diagnosis and to prevent morbidity from inappropriate intercostals to be insertions.
 
Introduction

There are five defects in the diaphragm to create herniation of intra-abdominal viscera into the thorax. The left CDH occurring through the posterolateral defect (Bochdalek) is the most common (80%)1 which occurs due to insult to the embryo at 8th week of gestation.1 It occurs probably once in 2200 births, when stillbirths are included.1 The common age of presentation is newborn and early infantile period with acute respiratory distress due to hypoplastic lungs with high mortality (65%).1 Delayed presentation with chronic respiratory or gastrointestinal complaints have also been reported (5-25%)1 from 1 month to late adulthood. The late presenting cases do not have extensive herniation and lungs are mature. The splenic herniation is rare outside neonatal period.2 These cases are difficult to differentiate from hydropneumothorax. Inappropriate thoracocentesis is frequently recorded, which increases morbidity. Chest X-rays and in doubtful cases contrast study is essential. They have low morbidity with correct diagnosis and surgical repair.

 
Case History

An 8-year-old male child otherwise healthy presented with sudden onset of severe pain in left hypochondrium without vomiting and dyspnoea.

On examination, he had tachycardia, severe tenderness and guarding in left hemithorax, normal position of heart sounds and no respiratory distress. Tuft of hair at lumbosacral region was present since birth.

Plain X-ray abdomen standing was suggestive of markedly raised left hemidiaphragm with clear lung field and normal cardiac shadow. Upper GI contrast study was required to confirm the diagnosis which showed the stomach in the left thoracic cavity with oesophagogastric junction at lower than normal and distorted antrum and duodenum. This suggested eventration of diaphragm with organoaxial type of gastric volvulus (Fig. 1). Spine radiographs showed lumbosacral spina bifida.

Nasogastric tube was inserted. Emergency laparotomy revealed left sided posterolateral diaphragmatic defect of 6 x 3 cm with acute gastric volvulus (organoaxial) with herniation of omentum, small bowel, transverse colon, spleen and splenic flexure of colon. There was no peritoneal sac. The left lung was hypoplastic. The contents were reduced back and defect was repaired with non-absorbable suture (Prolene 2-0) between diaphragm and chest wall. Left sided chest tube was inserted. Recovery was uneventful.

 
Discussion

Left CDH presenting through the foramen of Bochdalek in first few hours of life with respiratory distress is a common problem. Delayed presenting cases are (a) younger children with mainly respiratory symptoms, (b) older children with acute or chronic gastric volvulus.1,4 The patients with acute gastric volvulus present with triad of Borchardt (1) unproductive retching, (2) acute localized epigastric distension and (3) inability to pass a nasogastric tube. These features are difficult to assess in the child. Chronic volvulus may be symptomless and an incidental finding on a chest X-ray or barium meal examination. The complain of breathlessness is due to eventration of the diaphragm or a paraoesophageal hernia.

Abdominal radiology is essential for diagnosis. Mesentericoaxial volvulus of the stomach on plain films shows two fluid levels on erect films, one in the fundus (lower) and one in the antrum (upper) whereas organoaxial volvulus is less easy to diagnose on plain films (especially if unassociated with diaphragmatic defects) and may indeed be missed during a barium study. The stomach lies horizontally on the plain film with a single fluid level. They are frequently misdiagnosed as hydropneumothorax, hydrothorax, bronchogenic cyst and cystic adenomatoid malformation and subjected to inappropriate thoracocentesis.1 The normal previous X-ray does not exclude the diagnosis.3 Chest radiographs taken after nasogastric tube insertion help in early diagnosis.5 Upper GI contrast studies are required to confirm the diagnosis6 in doubtful cases which show oesophagogastric junction at lower than normal and distorted antrum and duodenum (Fig. 1).

Elhalaby et al did the study of 33 patients between 1993-2000. Ten patients had chronic respiratory or GI obstruction. Inappropriate ICD insertion was done in 3 patients diagnosed as having pleural effusion (in 2 cases) and pneumothorax (in one case). GI contrast study was necessary in 9 patients.6

Berman et al did the 20-year retrospective study of 26 patients of which 16 patients (62%) were originally misdiagnosed clinically and radiologically as infective lung changes, congenital lung cysts or pneumothorax. Four patients underwent inappropriate thoracocentesis.7

The splenic herniation is due to absence of ligamentous connections between stomach, posterior abdominal wall and spleen.8 These are the gastrophrenic, gastrosplenic, gastrocolic and gastrohepatic ligaments. Abnormal mobility and dificient fixation at hiatus of stomach accounts for the organoaxial type of volvulus of stomach. Gastric volvulus may be associated with asplenia9 and is usually idiopathic.10 Sehgal et al have reported only a single case of 3 year old child with 6 months history of respiratory symptoms and splenic herniation without gastric volvulus.2 Splenic herniation is rare outside the neonatal period.2

 
Conclusion
Thus this case emphasizes that late presenting hernia of Bochdalek is difficult to diagnose and requires very high index of suspicion. They are often misdiagnosed so awareness of the condition needs to be increased. Such patients should be diagnosed by plain radiographs and upper GI contrast studies. They have good outcome with surgical repair. Splenic herniation in older children is very uncommon.
 
References
1. de Lorimier Alfred A. Diaphragmatic hernia; pediatric surgery Ed. Ashcraft Keith W, Holder Thomas M, 2nd edition, chapter 19; WB Saunders Company, Philadelphia; 1993 : 204-17.
2. Sehgal A, Chandra J, Singh V, Dutta AK, Bagga D. Congenital diaphragmatic hernia : delayed presentation with asymptomatic splenic herniation. Indian J Chest Dis Allied Sci 2002; 44 (1) : 57-60.
3. Numanoglu A, Steiner Z, Millar A, Cywes S. Delayed presentation of congenital diaphragmatic hernia. S Afr J Surg 1997; 35 (2) : 74-6.
4. Newman BM, Afshani F, Karp MP, et al. Presentation of congenital diaphragmatic hernia past the neonatal period. Arch Surg 1986; 121 (7) : 813-6.
5. Quah BS, Hashim I, Simpson H. Bochdalek diaphragmatic hernia presenting with acute gastric dilatation. J Pediatr Surg 1999; 34 (3) : 512-4.
6. Elhalaby EA, Abo Sikeena MH. Delayed presentation of congenital diaphragmatic hernia. Pediatr Surg Int 2002; 18 (5-6) 480-5, Epub 2002, July 31.
7. Berman L, Stringer D, Ein SH, Shandling B. The late-presenting pediatric Bochdalek, hernia: A 20-year review. J Peditr Surg 1988; (23) 735-739.
8. Pelizz G, Lembo MA, Franchella A, Giombi A, D’Agostino F, Sala S. Gastric volvulus associated with congenital diaphragmatic hernia, wandering spleen and intra-thoracic left kidney : CT findings; Abdom Imaging, 2001; 26 (3) : 306-8.
9. Aoyamak, Tateishi K. Gastric volvulus in three children with asplenic syndrome. J Pediatr Surg 1986; 21 : 307-10.
10. Cameron AEP, Howard ER. Gastric volvulus in childhood. J Pediatr Surg 1987 : 944-7.
   

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*Resident, **Professor, ***Associate Professor;
+Assistant Professor; Department of Surgery, Medical College and SSG Hospital, Baroda - 390 001, Gujarat.