Ann Thorac Surg 2008;86:660-661. doi:10.1016/j.athoracsur.2008.02.004
© 2008 The Society of Thoracic Surgeons
Case Reports
Intrathoracic Multiple Schwannomas of a Single Intercostal Nerve
Fengshi Chen, MD,
Ei Nakayama, MD,
Kenichi Okubo, MD,
Hiroshi Date, MD*
Department of Thoracic Surgery, Kyoto University, Sakyo-ku, Kyoto, Japan
Accepted for publication February 4, 2008.
* Address correspondence to Dr Date, Department of Thoracic Surgery, Kyoto University, 54 Shogoin-Kawahara-cho, Sakyo-ku, Kyoto, 606-8507, Japan (Email: hdate{at}kuhp.kyoto-u.ac.jp).
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Abstract
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Intrathoracic schwannomas are solitary in general. We report a rare case of intrathoracic multiple schwannomas arising from a single intercostal nerve. A 27-year-old man was admitted with multiple tumors, along with the fourth intercostal nerve in the posterior chest wall. Thoracoscopic surgical exploration found a small tumor at 2 cm distal to the main tumors, which had not been detected preoperatively. All these tumors were completely resected en bloc and were diagnosed as schwannomas of the fourth intercostal nerve.
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Introduction
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Schwannomas are well-encapsulated benign tumors that originate from Schwann cells and are ordinarily solitary lesions [1]. Intrathoracic schwannomas are solitary in general and are usually found in the posterior mediastinum [2]. Herein we reported quite a rare case of intrathoracic multiple schwannomas arising from a single intercostal nerve.
A 27-year-old asymptomatic man with a chest wall mass visited our hospital. A chest computed tomographic scan showed multiple tumors along with the fourth intercostal nerve (Fig 1). The tumor was suspected as a neurogenic tumor originating from an intercostal nerve. Surgical excision was performed for a definitive diagnosis and treatment. Thoracoscopic surgical exploration found that the tumors originated from the fourth intercostal nerve. Interestingly enough, at 2 cm distal to the main tumors, we found a small nodule that had not been detected preoperatively (Fig 2). Retrospective review of the chest computed tomography on admission seemed to show a small tumor; however, it was impossible to confirm the existence of the tumor before surgery. These tumors (including a small nodule) were easily dissected from the intercostal muscle and ribs and were completely resected en bloc through thoracoscopic procedures (Fig 3). Microscopically, all the tumors (including the small nodule) consisted of areas that showed a dense spindle cell pattern with nuclear palisading and interlacing fasicles, and areas that were less cellular with myxomatous changes. The definite diagnosis was schawannoma without malignant components. The postoperative course of the patient was uneventful. He has been followed-up for 1 year with no evidence of recurrence.
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Fig 1. A chest computed tomographic scan on admission showed a multiple tumor along with the fourth intercostal nerve in the posterior chest wall.
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Fig 2. Thoracoscopic view at surgery revealed that there was a small tumor (arrows) near the main multiple tumors (arrowheads).
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Comment
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Peripheral tumors of the intercostal nerves are less common than neural tumors originating in the mediastinum. They are found in persons of any age and often cause no symptoms. Schwannoma is usually solitary and may arise from any cranial or peripheral nerve [3]. Schwannoma is the most common neurogenic tumor of the thorax, but it is very rare to find multiple schwannomas arising from a single intercostal nerve like our case. We believe that less than 10 cases have been reported to date. Ten percent of schwannomas are found to be malignant. Neurologic deficits can occur when the tumor arises in a confined apace, but only several published cases of intercostal pain caused by schwannoma of an intercostal nerve has been reported [4, 5]. The treatment of choice is complete resection of the tumor because malignant transformation has been reported [1]. As in the present case, thoracoscopic approach is appropriate for the resection of these intrathoracic neurogenic tumors [6].
In our case, it is of much interest that a very small nodule was detected at the time of thoracoscopic exploration. The pathology revealed that this small tumor was also schwannoma. As shown in Figure 1, it was difficult to detect its presence preoperatively because of the small size. This case suggests that undetectable tumors may remain after simple tumor excision for schwannoma; however, the appropriate extent of resection has not been discussed. Although the recurrence of schwannomas is reportedly very rare [7], we need to keep in mind that there may be a multiple occurrence of schwannoma in surgery of such patients, and we need to carefully search for preoperatively undetected tumors during surgery.
Schwannomatosis is defined as multiple schwannomas without the vestibular schwannomas that are diagnostic of neurofibrosis 2. Persons with schwannomatosis may develop intracranial, spinal nerve root, or peripheral nerve tumors [8]. Our patient did not have any other characteristics suggestive of neurofibrosis 2, but we need to conduct careful follow-up on the patient.
In conclusion, we experienced an unusual case of multiple schwannomas arising from a single intercostal nerve.
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References
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- Weiss SW, Goldblum JR. Schwannoma (neurilemoma)In: Weiss SW, Goldblum JR, editors. Enzinger and Weiss's soft tissue tumors. 4th ed.. St. Louis, MO: Mosby; 2001. pp. 1146-1167.
- Duwe BV, Sterman DH, Musani AI. Tumor of the mediastinum Chest 2005;128:2893-2909.[Medline]
- Das Gupta TK, Brasfield RD, Strong EW, Hajdu SI. Benign solitary schwannomas (neurilemmomas) Cancer 1969;24:355-366.[Medline]
- Stumpo M, Poppi M, Rizzo G, Martinelli P. Intercostal neuralgic schwannoma: a case report Muscle Nerve 2002;25:753-754.[Medline]
- McClenathan JH, Bloom RJ. Peripheral tumors of the intercostal nerves Ann Thorac Surg 2004;78:713-714.[Abstract/Free Full Text]
- Ishida T, Maruyama R, Saitoh G, Mitsudomi T, Sugimachi K. Thoracoscopy in the management of intrathoracic neurogenic tumors Int Surg 1996;81:347-349.[Medline]
- Davidson KG, Walbaum PR, McCormack RJ. Intrathoracic neural tumors Thorax 1978;33:359-367.[Abstract/Free Full Text]
- MacCollin M, Chiocca EA, Evans DG, et al. Diagnostic criteria for schwannomatosis Neurology 2005;64:1838-1845.[Abstract/Free Full Text]
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Abstract
Introduction
