J Korean Med Sci.  2009 Jun;24(3):517-519. 10.3346/jkms.2009.24.3.517.

Congenital Hemidiaphragmatic Agenesis Presenting as Reversible Mesenteroaxial Gastric Volvulus and Diaphragmatic Hernia: A Case Report

Affiliations
  • 1Division of Gastroenterology, Department of Internal Medicine, The Catholic University of Korea, Seoul, Korea. chowhang@catholic.ac.kr
  • 2Department of Thoracic Surgery, The Catholic University of Korea, Seoul, Korea.
  • 3Department of Radiology, The Catholic University of Korea, Seoul, Korea.

Abstract

A 70-yr-old woman complained of left sided chest pain and non-bilious vomiting for four days after taking a gastric bloating agent for an upper gastrointestinal study. The chest radiography revealed gastric air-fluid levels and bowel loops in the left thoracic cavity. An emergency thoracotomy was performed. The abdominal organs (stomach, spleen, splenic flexure of the colon) were in the left thorax and the entire left hemidiaphragm was absent. There were no diaphragmatic remnants visible for reconstruction of the left diaphragm. We provided warm saline irrigation and performed a left lower lobe adhesiotomy. Thirteen days after surgery, the chest radiography showed improvement in the herniation but mild haziness remained at the left lower lung field. Here we present the oldest case of congenital diaphragmatic agenesis presenting with transient gastric volvulus and diaphragmatic hernia.

Keyword

Congenital Hemidiaphragmatic Agenesis; Mesenteroaxial Gastric Volvulus; Hernia, Diaphragmatic; Spontaneous Resolution

MeSH Terms

Aged
Diagnosis, Differential
Diaphragm/*abnormalities/radiography/surgery
Female
Hernia, Diaphragmatic/*diagnosis/radiography/surgery
Humans
Stomach Volvulus/*diagnosis/surgery
Tomography, X-Ray Computed

Figure

  • Fig. 1 Chest radiographies. (A) There was no abnormal finding reported on from the local clinic 4 days before admission. (B) On admission, gastric air-fluid and bowel loops were observed in the left thoracic cavity and a coiled nasogastric tube was seen in the stomach. (C) The herniation improved but haziness was still seen at the left lower lobe after surgical intervention.

  • Fig. 2 Findings of upper gastrointestinal study (UGIS). (A) There was no disturbance of gastric passage at the local clinic 4 days before admission. (B) There were typical signs of a mesenteroaxial gastric volvulus; the stomach was in a vertical position with the pylorus higher than the cardia, and a double air-fluid level was seen; the antrum and pylorus were located inside the hernia with a hook like sign. (C) No passage disturbance of contrast media from the stomach into the duodenum; however, the stomach was still deviated into the thorax.

  • Fig. 3 CT finding at the level of the lower thorax demonstrating normal right (arrow) and absent left hemidiaphragm and loops of bowel in left thorax.

  • Fig. 4 Intraoperative findings. There was no diaphragmatic tissue. The abdominal organs (stomach, spleen, and splenic flexure of colon) were present in the left thoracic cavity.


Reference

1. Langman J. Medical embryology. 1981. 4th ed. Baltimore: Williams and Willkins;282–317.
2. Cunniff C, Jones KL, Jones MC. Patterns of malformation in children with congenital diaphragmatic defects. J Pediatr. 1990. 116:258–261.
Article
3. Tzelepis GE, Ettensohn DB, Shapiro B, McCool FD. Unilateral absence of the diaphragm in an asymptomatic adult. Chest. 1988. 94:1301–1303.
Article
4. Wakai A, Winter DC, O'Sullivan GC. Unilateral diaphragmatic agenesis precluding laparoscopic cholecystectomy. JSLS. 2000. 4:259–261.
5. Okoye BO, Bailey DM, Cusick EL, Spicer RD. Prophylactic gastropexy in the asplenia syndrome. Pediatr Surg Int. 1997. 12:28–29.
Article
6. Qazi A, Awadalla S. Wandering spleen: a rare cause of mesenteroaxial gastric volvulus. Pediatr Surg Int. 2004. 20:878–880.
Article
7. Singh G, Bose SM. Agenesis of hemidiaphragm in adults. Aust NZJ Surg. 1993. 63:327–328.
Article
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