| Home
> Table of Contents
> Case Reports |
| |
| 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. |
| |
|
RASAGILINE FOR MOTOR COMPLICATIONS IN PARKINSON’S DISEASE
Rascol and colleagues point out that rasagiline is taken as a single oral daily dose with no need for titration, and is thus easier to use for both patients and clinicians than most other adjuvant therapies. As such, this drug is likely to find favour with both neurologists and geriatricians as a useful addition to Parkinson’s disease therapeutics.
Carl E Clarke, The Lancet, 2005; 915.
SSRIs AND SUICIDE : EVIDENCE SUPPORTS THE ASSOCIATION
People taking selective serotonin reuptake inhibitors (SSRIs) are more likely to attempt suicide than people taking placebo or a non-SSRI drug. Fergusson and colleagues systematically reviewed 702 randomised controlled trial including 87,650 participants. They found significantly higher odds of suicide attempts for SSRIs than for placebo (number needed to treat to harm 684) and for therapeutic interventions other than tricyclic antidepressants (239). The meta-analysis did not detect a difference between SSRIs and tricyclic antidepressants. A number of major methodological limitations of the published trials may have led to underestimates of the risk of suicide attempts, say the authors.findings.
BMJ, 2005; 330 : 396.
|
*Resident, **Professor, ***Associate Professor;
+Assistant Professor; Department of Surgery, Medical College and SSG Hospital, Baroda - 390 001, Gujarat.
|
|
|
|
| |
|