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Interact CardioVasc Thorac Surg 2009;9:374-376. doi:10.1510/icvts.2009.203075 © 2009 European Association of Cardio-Thoracic Surgery
Diaphragmatic rupture during labour, two years after an intra-oesophageal rupture of a bronchogenic cyst treated by an omental wrappingCHU de Reims, Hôpital Robert Debré, Service de Chirurgie Thoracique et Cardiovasculaire, avenue du Général Koenig, F-51092, Reims Cedex, France Received 17 January 2009; received in revised form 14 April 2009; accepted 15 April 2009
*Corresponding author. Tel.: +(33) 3 26 78 71 05; fax: +(33) 3 26 78 32 73.
We report the case of a 27-year-old pregnant woman, presenting a rare but potentially fatal complication of an omental wrapping, transposed in the thorax by a phrenotomy due to an intra-oesophageal rupture of a bronchogenic cyst. Two years later, on time of delivery of her first pregnancy, she has presented a diaphragmatic rupture with a complete transposition of the volvuled stomach. The diagnosis was done using the Gastrographin transit and the CT-scan, showing the specific ‘Collar sign’. An urgent surgical correction was performed including the re-instatement of all abdominal organs in the peritoneum, the omental wrapping section and the diaphragmatic closure. The two-year follow-up was uneventful. We discuss the case, the investigations needed and the possibility to cut the omental wrapping without any complication two years after this oesophageal re-inforcement. In conclusion, we believe that omental transpositions must always be done using the retrosternal route or by transhiatal approach to avoid this serious complication after delivery.
Key Words: Diaphragmatic rupture; Labour; Abdominal organs; Outcomes; Imaging
Diaphragmatic ruptures are frequently related to severe thoracic and abdominal traumas [1]. Only a few cases of spontaneous diaphragmatic ruptures have been reported during labour [2, 3]. More published cases, in similar circumstances, have been related to previous hernias or traumas. We report the case of a 27-year-old female having presented a left diaphragmatic rupture in time of delivery. Two years before, she had presented a mediastinal bronchogenic cyst perforated in the oesophagus [4]. The resection of the cyst was performed by a left thoracotomy and the oesophagus was closed using the omentum, transposed through the left hemi-diaphragm. The follow-up was uneventful for more than two years. Two CT-scans (at 6 and 15 months) had confirmed the correct position of all organs and the absence of postoperative diaphragmatic hernia. During this first pregnancy, all clinical examinations were normal and no digestive symptoms were related until the labour. It was considered as normal, without need of additional procedure, despite some strange and acute chest pains. Two days later, the patient presented nausea, vomiting, and a progressive dyspnoea. At this time, a gastric suction improved, partially and temporarily, all symptoms. However, due to the persistent dyspnoea, a thoracic X-ray was performed and showed a large opacity of the inferior part of the left chest. We performed a Gastrographin injection to understand these unusual symptoms (Fig. 1). The investigation showed a complex image associating multiple hydroaeric levels compatible with a left diaphragmatic rupture. The CT-scan confirmed this diagnosis associated to the intrathoracic transposition of the small bowel and the colon. The ‘Collar sign’, specific of the diaphragmatic rupture, is well illustrated (Fig. 2). It is directly related to the herniation and strangulation of the stomach. This last one, partially fixed by the omentum to the thoracic oesophagus, was volvuled and transposed in the thorax; the first segment of the duodenum beginning at its top, and the oesophagus at its bottom.
As a consequence, we performed a medial laparotomy, removed all adherences related to the first surgery, and then, the stomach, the duodenum and the left colon were removed easily from the chest after the section of the omentum. The inspection of the diaphragm showed a rupture larger than the previous ‘omental hiatus’ surgically performed. An evident recent rupture was easily visualized. Finally, the diaphragm was closed using non-absorbable interrupted sutures, after a thoracic and abdominal drainage. No prosthesis was used due to the intrinsic diaphragmatic solidity around the rupture area and because of the sepsis risk related to the digestive suffering. The follow-up was uneventful with a rapid and total recovery. She was discharged on postoperative day 8. During the follow-up, clinical examinations and the 1, 12 and 24 months CT-scans were normal.
If diaphragmatic hernias are frequently reported in the literature, essentially for newborns and young children, diaphragmatic ruptures during labour are infrequent. The review of the adult literature based on these two last decades found no more than 15 articles concerning this issue. No extensive studies have been published and the exact incidence is still unknown. Some authors have reported exceptional cases of spontaneous diaphragmatic rupture during delivery [3, 5]. In all cases, acute and durable thoracic and/or abdominal pains were associated to nausea and vomiting, just before or a few days after the birth. These initial symptoms must alarm the physician due to the risk of rapid death. Radiological investigations must be done without delay. The chest X-ray can guide the diagnostic in a significant percentage of these cases [5]. The presence of a large bulla between the abdomen and the chest, with a constriction at the diaphragmatic level, called ‘the Collar sign’ is specific of a diaphragmatic defect. However, the CT-scan or magnetic resonance imaging is useful to diagnose and understand some cases, as in our patient [6, 7]. The Gastrographin transit can also be useful to appreciate the exact location of the oesophagus, the stomach or the duodenum. When the diagnosis is done, the surgical repair must be carried out without delay. Two ways are available: the thoracic or the abdominal ways [8]. In our case, we chose the abdominal way due to the previous thoracic surgery and the risk of abdominal suffering, needing a potential digestive resection. The omental wrapping is an easy and effective technique to treat an oesophageal fistula, essentially in this inferior segment [8]. The healing of the omentum on the oesophagus induces a local neovascularization, allowing the section of the omentum at the diaphragmatic level, two years after the first surgery, without any consequence. In this case, we have previously transposed the omentum through the left part of the diaphragm, and not as actually recommended, by the retrosternal route. The relative diaphragmatic weakness induced by the previous phrenotomy is probably the main cause of this rare and serious complication. In conclusion, we believe that in case of pregnancy after a previous phrenotomy, a preventive caesarean section could be discussed to avoid potentially fatal complications. However, to date, the transdiaphragmatic approach must be avoided for all patients. The best way to prevent this kind of complication is to systematically perform an omental transposition using a limited disinsertion of the diaphragm, as described in the ‘retrosternal route’, or a transhiatal approach.
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Abstract
1. Introduction
