Ann Thorac Surg 2000;69:1928-1929
© 2000 The Society of Thoracic Surgeons
Case reports
Angioleiomyoma of the diaphragm
Aart B.B. van Rijn, MD, PhDa,
Klaas W. van Kralingen, MD, PhDb,
Ingeborg A. Koelma, MDc
a Department of General Surgery, Bronovo Hospital, The Hague, The Netherlands
b Department of Pulmonology, Bronovo Hospital, The Hague, The Netherlands
c Department of Pathology, Bronovo Hospital, The Hague, The Netherlands
Address reprint requests to Dr van Rijn, Department of General Surgery, Bronovo Hospital, Bronovolaan 5, 2597 AX The Hague, The Netherlands
e-mail: avanrijn{at}worldonline.nl
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Abstract
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We report a case of a large symptomatic angioleiomyoma of the diaphragm. The tumor was diagnosed by computed tomography and videothoracoscopic biopsy. We removed the tumor through a left thoracolaparotomy. Three months later, a local recurrence was removed through an upper midline laparotomy. The patient remains without evidence of a recurrence 48 months after the second operation.
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Introduction
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Primary tumors of the diaphragm are rare. We describe a patient with a large symptomatic angioleiomyoma of the diaphragm. The tumor was removed through a left thoracolaparotomy. The diaphragm was reconstructed with a prosthetic patch.
A 53-year-old previously healthy man presented with severe pain in the left lower thoracic region and the left shoulder. The pain had started 9 months earlier and since then it had gradually worsened. On physical examination, we suspected a small fluid collection in the left pleural cavity, but no other abnormalities were found. Because of the severity of the pain, the patient was admitted to our hospital for clinical evaluation and treatment. Shortly after admission, it appeared that only opiates provided adequate pain relief. Biochemical and hematological blood analysis showed no abnormalities. A chest film confirmed a small fluid collection in the left pleural cavity. A computed tomographic (CT) scan showed a tumor with a diameter of approximately 6 cm in the left crus of the diaphragm and the medial part of the left diaphragmatic dome (Fig 1). The CT scan showed no connection of the tumor with the left lung or any of the abdominal organs. No other intraabdominal abnormalities were found. A first diagnosis of mesothelioma was made. To confirm the diagnosis by biopsy and to assess operability, we carried out a left videothoracoscopy under general anesthesia. In the dome of the left diaphragm, we saw a tumor of which we took a biopsy. Taking the biopsy caused brisk intrathoracic bleeding, which suggested a highly vascular lesion. Microscopic examination showed a highly vascular mesenchymal tumor without cellular dysplasia or mitotic figures. Because of the persistent and severe pain, we decided to remove the tumor operatively. The area was exposed through a left thoracolaparotomy, carried through the left costal margin. We removed the tumor together with a margin of normal diaphragmatic tissue, leaving a defect in the diaphragm measuring 12 cm in diameter. We placed clips to delineate the excision area, and closed the diaphragmatic defect with a 2-mm polytetrafluoroethylene (PTFE) patch (Gore-Tex; W. L. Gore & Associates, Flagstaff, AZ). The patient had an uneventful postoperative recovery. The preoperative complaints of pain did not recur during the postoperative hospital stay. A postoperative CT scan did not show any residual tumor.
Microscopic examination of the tumor showed the same histology as in the biopsy specimen. Although there was no definite capsule, there were no other characteristics of malignancy. The tumor was classified as a benign angioleiomyoma (Fig 2).
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Fig 2. Angioleiomyoma with many thin-walled vessels surrounded by spindle-shaped monomorphic cells. Mitotic figures are rare.
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Three months postoperatively, the patient presented again with severe pain, now localized in the right thoracic region. A CT scan showed a tumor mass with a diameter of 5 cm, adjacent to the previous excision area (Fig 3). We removed the tumor through a midline incision in the upper abdomen. Microscopic examination showed a noncapsular benign tumor with the same histological characteristics as the first tumor. Again, recovery was uneventful. A follow-up CT scan 6 months after the second operation was negative for recurrence. The patient remains in good health and free of pain 48 months after the second operation.
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Fig 3. CT scan with recurrent tumor (R) in the lower part of the right crus of the diaphragm. The metal clip at the left border of the recurrent tumor was placed during removal of the first tumor to mark the tumor bed.
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Comment
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Primary tumors of the diaphragm are rare. Isolated cases of hemangiopericytoma, solitary fibrous tumor, lipoma, and neurilemoma have been reported [1–4]. We present a patient with a large angioleiomyoma of the diaphragm that caused severe pain in the left thoracic region and the left shoulder. In our patient, accurate anatomical information could be obtained preoperatively with regular chest films and a CT scan. A special problem was presented by the large diaphragmatic defect resulting from the removal of the tumor. A large diaphragmatic defect should preferably be closed with prosthetic material [5]. Attempts at primary repair under tension may lead to rupture of the diaphragm with herniation. Marlex mesh (Bard, Murray Hill, NJ) and PTFE cardiovascular patch are used most frequently. In an animal model, Newman and associates demonstrated the PTFE patch to be superior to silastic sheeting [6]. In our patient, a Gore-Tex 2-mm PTFE patch provided satisfactory closure of the diaphragmatic defect. The lack of a well-defined tumor capsule may have led to incomplete excision followed by a recurrence. The fact that the patient remains free of pain and without signs of recurrence 48 months later suggests that the second operation resulted in complete excision of the tumor.
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References
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El-Jabbour J.N., McLaren K.M., Sedgwick D. Haemangiopericytoma of the diaphragm. Scott Med J 1988;33:375-376.[Medline]
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Papachristos I.C., Laoutides G., Papaefthimiou O., Andrianopoulos E.G. Gigantic primary lipoma of the diaphragm presenting with respiratory failure. Eur J Cardiothorac Surg 1998;13:609-611.[Abstract/Free Full Text]
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McClenathan J.H., Okada F. Primary neurilemoma of the diaphragm. Ann Thorac Surg 1989;48:126-128.[Abstract]
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Weksler B., Ginsberg R.J. Tumors of the diaphragm. Chest Surg Clin N Am 1998;8:441-447.[Medline]
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Newman B.M., Jewett T.C., Lewis A., et al. Prosthetic materials and muscle flaps in the repair of extensive diaphragmatic defects. J Pediatr Surg 1985;20:362-367.[Medline]
Accepted for publication October 18, 1999.
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1653 - 1654.
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Abstract
Introduction
