Ann Thorac Surg 2006;81:e9-e10
© 2006 The Society of Thoracic Surgeons
Case report
Congenital Diaphragmatic Hernia Presenting as Splenic Rupture in an Adult
Bruce W. Robb, MD
a
,
Michael F. Reed, MD
b
,
*
a Department of Surgery, University of Cincinnati College of Medicine, Cincinnati, Ohio
b Division of Thoracic Surgery, University of Cincinnati College of Medicine, Cincinnati, Ohio
Accepted for publication November 8, 2005.
* Address correspondence to Dr Reed, University of Cincinnati College of Medicine, Department of Surgery, 231 Albert B. Sabin Way, PO Box 670558, Cincinnati, OH 45267-0558 (Email: michael.reed{at}uc.edu).
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Abstract
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We present the case of a 66-year-old man with a congenital diaphragmatic hernia and splenic rupture. The adult presentation of congenital posterolateral diaphragmatic (Bochdalek) hernia is rare. We describe the operative approach and provide a brief review of the literature.
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Introduction
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Congenital posterolateral diaphragmatic hernia is rare in adults. First described by Bochdalek in 1848, these hernias usually become evident during the neonatal period with signs and symptoms of respiratory failure. The prevalence of posterolateral diaphragmatic hernia has been estimated to be 1 in 2,200 to 12,500 live births. Although a series of congenital diaphragmatic hernias in infancy documents thousands of cases, fewer than 200 adult patients with congenital posterolateral diaphragmatic hernia have been reported [1]. Unlike infants who present with respiratory distress, the most frequent presentation in adults is abdominal pain. Here we present the case of a ruptured spleen within a posterolateral diaphragmatic hernia.
The patient is a 66-year-old man who first noted mild left upper quadrant and left flank pain 6 months prior to the time of operation. Chest roentgenograms and computed tomographic scans obtained by his primary care physician revealed a left diaphragmatic hernia. He was referred for thoracic surgical evaluation and operative repair. During the course of his preoperative clearance, he presented to The University Hospital with acute, worsening pain. He was hemodynamically stable. A repeat chest computed tomography was performed, which again demonstrated a left diaphragmatic hernia with a large intestine and spleen in the left chest. In addition, fluid collection was noted around the spleen without evidence of active extravasation of intravenous contrast (Fig 1). The patient was admitted to the thoracic surgery service, placed on bedrest, directed to have nothing by mouth, and followed-up with serial hematocrit measurement and physical examination. Cardiology evaluation was performed due to his cardiac risk factors. He was then taken to the operating room for definitive repair.
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Fig 1. Chest computed tomographic scan (with intravenous and oral contrast) demonstrating herniation of the spleen, colon, and mesentery into the left hemithorax, with peri-splenic hematoma and associated pleural effusion.
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The defect was approached through a standard left posterolateral thoracotomy entering the chest through the seventh intercostal space. The hernia contained clotted blood, the spleen had a large subcapsular hematoma, and the colon and omentum were normal (Fig 2). Although there was no evidence of active bleeding, it was believed that the spleen could not be safely reduced into the abdomen, and a splenectomy was performed. The hernia defect was then dissected circumferentially after the reduction of the remainder of the abdominal contents. The defect was a lateral Bochdalek-type that measured approximately 8 cm x 10 cm. The defect was closed primarily without significant tension, except for the area at the lateral chest wall. A 1-mm Gore-Tex (W.L. Gore & Assoc, Flagstaff, AZ) dual mesh patch was then sewn as an onlay with interrupted 0 polypropylene mattress sutures, thereby buttressing the primary repair and closing the lateral defect. The postoperative course of the patient was uneventful.
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Fig 2. Intraoperative photograph of the left hemithorax containing the left lung, the spleen with an associated hematoma, and the splenic flexure of the colon.
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Comment
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Described by Bochdalek in 1848 [2], congenital diaphragmatic hernias usually become evident during the neonatal period with signs and symptoms of respiratory failure. Although a series of congenital diaphragmatic hernias in infancy documents thousands of cases, fewer than 200 adult patients with congenital posterolateral diaphragmatic hernia have been reported [1]. Kirkland [3] was the first to extensively review the literature and identify characteristics of adults presenting with congenital diaphragmatic hernias from 1853 to 1958. Unlike infants who present with respiratory distress, the most frequent presentation in adults is abdominal pain (50%), followed by vomiting, dyspnea, and chest pain. Many patients are asymptomatic (25% in many series) [3, 4].
Several explanations for the delayed diagnosis have been forwarded. Osebold and Soper [5] suggested that the rupture of a previously confining pleuroperitoneal sac coincided with the onset of symptoms in adults. Others believe that trauma or increasing abdominal pressure from pregnancy or obesity is typically necessary to produce symptoms, and that the true incidence is much higher than that reported in the surgical literature. In a review of 940 consecutive chest and abdominal computed tomographic scans obtained at a university medical center in 1984, a 6% prevalence of Bochdalek hernias was reported [6]. Even if this number is greatly in excess of the true incidence, the important point is that these hernias are difficult to diagnose if not suspected.
We believe that only one other intrathoracic rupture of the spleen has been described in patients with congenital diaphragmatic hernia [7]. Our case differs in the age of our patient (66 years vs 15 years) and the absence of any known trauma to the thorax. Even in those patients suspected of traumatic diaphragmatic hernia, splenic rupture is relatively uncommon.
In this reported case, the indication to operate was clear, given the splenic rupture. In general, the indications to repair congenital diaphragmatic hernia are the same as those to treat any hernia, and should take into account the patient's overall medical condition, the frequency of symptoms, and the risk of life-threatening ischemia to hernia contents if the repair should not be undertaken. Surgical approach to the repair of the diaphragm has been much debated in the literature. Generally we believe as many others that congenital or longstanding diaphragmatic hernias are best approached through a thoracotomy, whereas acute, traumatic herniation may be best dealt with a celiotomy.
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References
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- Bujanda L, Larrucea I, Ramos F, et al. Bochdalek's hernia in adults J Clin Gastroenterol 2001;32:155-157.[Medline]
- Bochdalek VA. Einige Betrachtungen uber die Entstehung des angelborenen Zwerchfellbruches. Als Bietrag zur pathologischen. Anatomie der Hernien Vjsch Prakt Heik 1848;19:89-97.
- Kirkland JA. Congenital posterolateral diaphragmatic hernia in the adult Br J Surg 1959;47:16-22.[Medline]
- Ahrend TR, Thompson BW. Hernia of the foramen of Bochdalek in the adult Am J Surg 1971;122(5):612-615.[Medline]
- Osebold WR, Soper RT. Congenital posterolateral diaphragmatic hernia past infancy Am J Surg 1976;131(6):748-754.[Medline]
- Gale ME. Bochdalek herniaprevalence and CT characteristics. Radiology 1985;156(2):449-452.[Abstract/Free Full Text]
- Hocking MA, Bolton PM. Intrathoracic rupture of the spleen Br J Surg 1978;65(8):546-548.[Medline]
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Abstract
Introduction
