Ann Thorac Surg 2006;81:716-718
© 2006 The Society of Thoracic Surgeons
Case report
Bilateral Cervical Lung Hernia With T1 Nerve Compression
Mesbah Rahman, FRCS
*
,
Keith G. Buchan, FRCS(CTh),
Kyapanda M. Mandana, MCh,
Eric G. Butchart, FRCS
Department of Cardiothoracic Surgery, University Hospital Wales, Cardiff, United Kingdom
Accepted for publication October 14, 2004.
* Address correspondence to Dr Rahman, Department of Cardiothoracic Surgery, University Hospital Wales, Cardiff CF14 4XW, UK (Email: mesbah{at}hotmail.com).
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Abstract
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Lung hernia is a rare condition. Approximately one third of cases occur in the cervical position. We report a case of bilateral cervical lung hernia associated with neuralgic pain that was repaired using bovine pericardium and biological glue.
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Introduction
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Lung hernia is a rare clinical condition, approximately one third of which are cervical. Ronald was the first to describe lung hernia in 1499 [1]. Approximately 300 cases have been reported in literature. Descriptions of cervical lung hernia fall into two categories: firstly, those in which a definite tear in the Sibson fascia is identified, and there is an associated hernial sac; secondly, those in which there is only a diffuse laxity of the suprapleural membrane with no associated hernial sac. In the first category, the precipitating factor is usually blunt trauma, often with an acute onset of lung herniation. In the second category, the onset is insidious and is associated with prolonged exposure to raised intrathoracic pressure. This report details a case of bilateral spontaneous cervical lung hernia presenting with neuralgic pain and intermittent cervical swelling.
A 36-year-old construction worker, whose fitness training involved weight lifting, presented with a 2-year history of intermittent swelling in the left supraclavicular fossa. The swelling appeared several times a day and was unpredictable in its occurrence, although it often followed a period of straining. It usually disappeared spontaneously after a minute or so. The patient described the swelling as the size of a tennis ball. Initially, the swelling was painless but during the 3 months before presentation he had intermittent severe sharp pain down the left arm medially in a T1 nerve root distribution whenever the swelling was present.
On examination the patient was a stocky, moderately overweight man; there was no visible or palpable swelling, and the swelling could not be reproduced by a Valsalva maneuver. However, after the patient ran up a few flights of stairs, the swelling appeared in the left supraclavicular fossa. It was soft, nontender, and resonant on percussion. Another smaller swelling over the right supraclavicular fossa was also noted (Fig 1). Ear, nose, and throat examination was unremarkable, and chest roentgenogram and computed tomography scan of the neck and thorax were normal. A diagnosis of bilateral cervical lung hernia was made.
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Fig 1. Swelling in both supraclavicular fossae (arrows), the left one being prominent and symptomatic.
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The patient decided to have surgical treatment because of the frequent appearance of the hernia associated with severe neuralgic pain. At left posterolateral thoracotomy, Sibson's fascia was noted to be abnormally lax although no actual defect at the apex could be detected. A bovine pericardial patch (10 x 16 cm) was folded on itself to double the thickness, cut to shape, and used to cover the apex from within the thorax. A thin, even coat of biological glue (BioGlue Surgical Adhesive; CryoLife, Kennesaw, Georgia) was applied to the surface of the pericardial patch, and it was then held against the apical pleura until the glue set. A few 5-0 polypropylene sutures and additional glue were applied at the edge of the patch. The patient made an uneventful recovery without any glue-related complication, and was followed up for 2 years without any episode of neuralgic pain in the left arm or left-sided neck swelling (Fig 2). In the meantime, the right-sided cervical hernia increased in size with similar symptoms and was also treated successfully using the same technique.
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Fig 2. One year after repair of left cervical hernia. There is no swelling on the left side. Note a small swelling (arrow) in the right supraclavicular fossa.
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Comment
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Morel-Lavalle classified lung hernia according to location (cervical, thoracic, and diaphragmatic) and etiology in 1845 [2]. The majority (65% to 83%) of the lung hernias occur through the chest wall [3, 4]. Cervical hernias occur through a defect in Sibson's fascia, usually anteromedially between the anterior scalene and sternocleidomastoid muscles [5]. In this location, a trapped portion of lung under pressure could be expected to compress the subclavian vein and the closely related T1 nerve root. In children, they are usually congenital, and often resolve spontaneously, whereas in adults, they are usually acquired. In general, the majority of lung hernias (82%) are acquired [4]. Acquired hernias can be traumatic, spontaneous, or, rarely, pathological. Two factors contribute to the development of spontaneous cervical lung hernia: weakness in Sibson's fascia and the parietal pleura or neck muscles, and increased intrathoracic pressure as produce by chronic coughing, straining, blowing of musical instruments, or weight lifting, as with our patient [4].
Most reported cervical lung hernias are asymptomatic but sometimes they can give rise to cough due to tracheal irritation, mild dysphagia, or pain due to incarceration [5]. Neuralgic pain in cervical lung hernia has not been reported previously. The size of the swelling varies, and in large hernias may reach up to thyroid cartilage. If present at the time of an roentgenogram, cervical lung hernias are seen as apical radiolucencies extending into the base of the neck. If very large, they can cause lateral tracheal deviation [5]. Because of their intermittent appearance, radiographs obtained at maximal inspiration or during Vasalva maneuver are required. Even with these techniques, it is sometimes difficult to visualize it, and the diagnosis is made on clinical grounds alone. The differential diagnosis of swellings in the supraclavicular fossa associated with radiologic lucency at the root of the neck includes pharyngocoele, laryngocoele, and esophageal diverticula [6]. In our case, full ear, nose, and throat examination excluded any other possibilities. In the absence of any ear, nose, and throat pathology and radiologic abnormality, the history of intermittent swelling in the root of the neck especially on coughing or straining favors the diagnosis of cervical lung hernia.
Cervical lung hernia does not need intervention unless symptoms become severe, as in this case. The principles of operative technique are the same as for hernias elsewhere: closing the defect and strengthening the anatomy. Thoracotomy provides the best exposure, but some surgeons have used the cervical approach in patients unsuitable for thoracotomy [2]. Several different operative techniques have been described, although no large series have been reported to allow comparison of one technique with another. Small defects in Sibson's fascia may be dealt with using interrupted nonabsorbable sutures [2], but the tissues are usually very thin. Larger defects require prosthetic materials for repair. The material used for repair should be relatively inert to avoid excessive fibrous reaction. In this case, we used biological prosthetic material and biological glue to fix it in place. It has been suggested in the literature that application of glutaraldehyde-based biological glues can cause tissue necrosis and nerve injury. To avoid inadvertent tissue damage, the biological glue was applied in accordance with recommendations from the manufacturer: dry the target site, prime the device to ensure proper mixing of the components, and apply a thin and even layer. Histology study shows biological glue penetration is usually very superficial (3 to 5 cell layers deep) [7]. In the case described here, the intact pleura served as a barrier to prevent contact with the phrenic nerve and the brachial plexus, and we believe that an intact pleura is essential for this technique.
Recently, a thoracoscopic approach has been described for reduction of cervical lung hernia [8]. It is possible that the method we have described could be adapted for thoracoscopic repair, although direct vision and access allows even pressure to be applied to the whole patch until the glue hardens and sets. The necessity of additional glue application around the edges is also easier to assess and implement.
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References
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- McAdams HP, Gordon DS, White CS. Apical lung herniaradiological findings in six cases. Am J Radiol 1996;167:927-930.[Abstract/Free Full Text]
- Li C, Miller WT. Roentgenogram of the monthair in the neck. Chest 1990;98:987-988.
- Hewitt CW, Maria SW, Kann BR, et al. BioGlue surgical adhesive for thoracic aortic repair during coagulopathyefficacy and histopathology. Ann Thorac Surg 2001;71:1609-1612.[Abstract/Free Full Text]
- Sanghoon J, Eung BL, Joon YC, Bong HC, Jongtae L, Kyu TK. Thoracoscopic repair of cervical lung hernia J Thorac Cardivasc Surg 2002;124:1030-1031.[Free Full Text]
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Abstract
Introduction
