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although their histologic and immunohistochemical properties are similar. Localized MM must also be differentiated from solitary fibrous tumor, a pure spindle cell tumor without a histologic well-defined pattern (patternless pattern), and without atypical features or necrosis. The mitotic index is generally low, and they have a well-described immunohistochemical marker such as CD34. Most of them are benign, although there are described some local recurrences in not well-resected cases. Our case had many architectural patterns, CD34 was consistently negative, and mesothelial markers such as thrombomodulin and calretinin were both positive. Other important differential diagnoses are synovial sarcoma and other sarcomas that could arise from the chest wall. Synovial sarcomas rarely affect the pleura, and most of them are biphasic, epithelial, and sarcomatous. As for the rest of the sarcomas, they are easily distinguished with immunohistochemical staining. Prognosis in this kind of localized mesotheliomas is unknown because of the few cases reported. Our patient is alive and well 8 months after operation, and there are many cases described in which complete surgical resection might be curative. The recurrent pattern for localized type of mesotheliomas is diffuse type [8] without distant metastasis, and so a correct diagnosis and surgical treatment in the early stages are essential for long-term survival. In summary, we describe the case of a localized MM proliferation. Localized MM must be included in differential diagnosis of chest wall-based masses. A complete surgical resection may be curative in some of these cases.
Ann Thorac Surg 2002;73:1294 – 6
Spontaneous Esophageal Perforation Presenting as Meningitis Carlo C. Jurani, MD, Gerald L. Early, MD, and Shauna R. Roberts, MD Department of General Surgery, University of Kansas, Kansas City, Kansas, and Department of Cardiothoracic Surgery, Genesis Heart Institute, Davenport, Iowa
This report describes a unique case of spontaneous esophageal perforation (Boerhaave’s syndrome) presenting as meningitis. After a delay in diagnosis (16 days), the patient was successfully treated with debridement, primary closure, and drainage. Although rare, central nervous system infections have been reported in association with esophageal perforation caused by instrumentation, trauma, and malignancy. We report this case of spontaneous esophageal perforation giving rise to meningitis. (Ann Thorac Surg 2002;73:1294 – 6) © 2002 by The Society of Thoracic Surgeons
T
he deposition of oral aerobic and anaerobic bacteria into the mediastinum after esophageal disruption leads to a rapidly spreading and often lethal infection. Although uncommon, the infection can extend to the central nervous system (CNS) and manifest as meningitis, brain abscess, or vertebral osteomyelitis. The following case report is a description of spontaneous esophageal perforation leading to meningitis.
References 1. Crotty TB, Myers JL, Katzenstein AL, Tazelaar HD, Swensen SJ, Churg A. Localized malignant mesothelioma. A clinicopathologic and flow cytometric study. Am J Surg Pathol 1994; 18:357– 63. 2. Ojeda HF, Mech K, Hicken WJ. Localized malignant mesothelioma: a case report. Am Surg 1998;64:881–5. 3. Walz A, Koch HK. Malignant pleural mesothelioma: some aspects of epidemiology, differential diagnosis and prognosis: histological and immunohistochemical evaluation and follow-up of mesotheliomas diagnosed from 1964 to January 1985. Pathol Res Pract 1990;186:124–34. 4. Okike N, Bernatz PE, Woolner LB. Localized mesothelioma of the pleura: benign and malignant variants. J Thorac Cardiovasc Surg 1978;75:363–72. 5. Sato K, Maeda K, Ichinose Y, Yano T, Hara N, Ohta M. An operated case of malignant localized mesothelioma of the pleura. Nippon Kyobu Geka Gakkai Zasshi 1992;40: 1291– 4. 6. Matsukuma S, Aida S, Hata Y, Sugiura Y, Tamai S. Localized malignant peritoneal mesothelioma containing rhabdoid cells. Pathol Int 1996;46:389–91. 7. Waridel D, Lanitis G. Localized malignant mesothelioma of the peritoneum revealed by a parietal mass. Schweiz Med Wochensch 1975;105:1026–30. 8. Watanabe S, Shimokawa S, Sakasegawa K, Nakamura Y, Sakata R. Surgical treatment for malignant pleural mesothelioma in eight cases. Kyobu Geka 2000;53:1101– 4. © 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
A 42-year-old woman presented to an outside hospital with a 2-week history of progressive upper back and neck pain that began after straining to lift an object at work. She complained of confusion, blurred vision, weakness, and fever. She admitted to severe coughing spells, but no emesis. Initial studies showed a white blood cell count of 36,000/mm3 and a urinary tract infection for which she was started on ciprofloxacin. Blood cultures grew Staphylococcus aureus. Chest roentgenogram uncovered a right apical lesion, and computed tomography scan of the chest revealed a right apical lung mass extending into the mediastinum and right neck. Computed tomography of the head demonstrated marked pneumocephalus. Because of uncertainty of the primary diagnosis and her deteriorating clinical status, the patient was transferred to our institution. She was resuscitated in the intensive care unit, and broad-spectrum antibiotics were started. Chest computed tomography demonstrated a pneumomediastiAccepted for publication Aug 17, 2001. Address reprint requests to Dr Jurani, Department of General Surgery, University of Kansas, 4002 Murphy Administration Building, 3901 Rainbow Blvd, Kansas City, KS 66160-7119.
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CASE REPORT JURANI ET AL ESOPHAGEAL PERFORATION AND MENINGITIS
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tomy was created, and a gastrostomy was performed to establish drainage from above and below. The patient remained septic and ventilator dependent in the early postoperative period. An esophagram 10 days postoperatively demonstrated a small leak at the area of the repair, and a percutaneous drain was placed into the fluid collection to provide a controlled esophagocutaneous fistula. During the next several weeks the patient’s clinical status waxed and waned but gradually improved. An esophagram 11 weeks after the repair demonstrated no leak, and after 13 weeks of hospitalization she was discharged home on a full liquid diet with no drains, and no neurologic deficits.
Comment Central nervous system infections are rare complications of esophageal perforation. They have been reported after stenting for benign stricture [1], after blunt traumatic esophageal rupture [2], in a patient with esophageal cancer [3], and as a delayed complication of dilatation of lye strictures in children [4]. The spontaneous origin of the esophageal rupture and the apparent esophagosubarachnoid fistula make this case unique.
Fig 1. Magnetic resonance imaging, sagittal view, T1-weighted image demonstrating air in the thecal sac (arrowheads) and paraesophageal abscess (arrow).
num, and magnetic resonance imaging characterized the right paraspinous mass as containing gas and necrotic debris and showed air in the spinal canal (Fig 1). A lumbar puncture was discussed; however, an esophagram revealed extravasation of contrast from the posterolateral aspect of the proximal esophagus with accumulation in the right apex (Fig 2), and the patient was taken immediately to the operating room. Through a right thoracotomy incision the esophagus was exposed and found to have a perforation at the level of the second thoracic vertebral body. Purulent material from the area subsequently grew out Staphylococcus aureus, Streptococcus organisms, and Bacteroides organisms. The area was debrided, exposing the vertebrae, but no gross communication with the spinal canal was identified. The esophagus was repaired primarily in two layers with a pleural flap, and the mediastinum was widely incised and irrigated. The chest was closed with three thoracostomy tubes draining the area, and esophagoscopy was performed, demonstrating no masses or strictures. The right neck was then drained, a pharyngos-
Fig 2. Esophagram demonstrating free rupture into the right pleural space.
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In our case, mental status changes, fever, chest mass, and bacteremia were all present without a unifying primary diagnosis, and until pneumomediastinum was manifest on computed tomography of the chest, the integrity of the esophagus was not questioned. The location (proximal) and cause (spontaneous without emesis) in the present patient are atypical for Boerhaave’s syndrome, which is usually associated with a distal esophageal perforation after forceful emesis. Contrast study of the esophagus, which is both highly sensitive and specific, was diagnostic. Although unusual under any circumstances, CNS infections seem to have been more frequently reported with proximal esophageal perforations [1–3]. The posterior cricopharyngeal region is the most common site of iatrogenic perforation, and at this area the outer longitudinal esophageal muscle layer diverges as two fasciculi exposing a V-shaped area [5]. The proximity of the cervical esophagus to the spinal column may play a role in the development of CNS infections, and this may help explain the rare occurrence of CNS infections after distal perforations. The management of patients with CNS infections associated with esophageal perforation varies according to the clinical circumstance. Kotler and associates [4] have reported CNS infections 4 to 9 months after the occurrence of esophageal perforation in which brain abscesses were drained operatively and meningitis was treated with antibiotics. When CNS infection is identified while there is ongoing contamination from the esophageal perforation, the management is not straightforward. One patient with an esophageal subarachnoid fistula initially improved with antibiotic treatment, then died when meningitis recurred [3]. Subarachnoid fistulas if identified intraoperatively can be repaired by simple ligation. Like others [6], we favor primary esophageal repair for Boerhaave’s syndrome even if it is more than 24 hours since perforation. This approach, combined with mediastinal debridement and drainage plus a pharyngostomy and gastrostomy, resulted in a good outcome in this desperately ill woman.
References 1. Boulis NM, Armstrong WS, Chandler WF, Orringer MB. Epidural abscess: a delayed complication of esophageal stenting for benign stricture. Ann Thorac Surg 1999;68:568–70. 2. Ring D, Vaccaro AR, Scuderi G, Green D. Vertebral osteomyelitis after blunt traumatic esophageal rupture. Spine 1995;20: 98 –101. 3. Cornwell J, Walden C, Ghahremani GG. CT demonstration of fistula between esophageal carcinoma and spinal canal. J Comput Assist Tomogr 1986;10:871–3. 4. Kotler R, Schild JA, Holinger PH. Delayed CNS complications. Laryngoscope 1975;85:1379– 86. 5. Jones WG, Ginsberg RJ. Esophageal perforation: a continuing challenge. Ann Thorac Surg 1992;53:534– 43. 6. Lawrence DR, Ohri SK, Moxon RE, Townsend ER, Fountain SW. Primary esophageal repair for Boerhaave’s syndrome. Ann Thorac Surg 1999;67:818–20. © 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
Ann Thorac Surg 2002;73:1296 – 8
Proliferative Myositis: A Rare Pseudosarcoma of the Chest Wall Michael S. Kent, MD, Douglas B. Flieder, MD, Jeffrey L. Port, MD, and Nasser K. Altorki, MD Departments of Cardiothoracic Surgery and Pathology, The New York-Presbyterian Hospital, Weill-Cornell Medical Center, New York, New York
Proliferative myositis is a rare, inflammatory tumor that is often misdiagnosed as sarcoma. The clinical course of proliferative myositis is benign, and local recurrence after simple excision is uncommon. Typically, the lesion presents in the extremities or the head and neck. We present an unusual case of proliferative myositis with involvement of the anterior chest wall. (Ann Thorac Surg 2002;73:1296 – 8) © 2002 by The Society of Thoracic Surgeons
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roliferative myositis (PM) is one of several benign tumors of the soft tissue. Its rapid growth and bizarre microscopic appearance often lead to the misdiagnosis of a high-grade sarcoma. Fortunately, recurrence even after subtotal excision is infrequent and therefore radical surgery is rarely required. We present an unusual case of PM affecting the chest wall and discuss the clinical and microscopic features of this uncommon tumor. A 48-year-old man presented to the thoracic service for evaluation of a painful, rapidly enlarging chest wall mass. The tumor was in the left parasternal area and had increased in size during the past 4 weeks. A computed tomographic scan of the chest (Fig 1) demonstrated a 5-cm ⫻ 7-cm bulky mass involving the left pectoralis major muscle with extension into the sternum. An incisional biopsy revealed a benign, spindle-cell process involving the skeletal muscle, consistent with proliferative myositis. At operation, the mass was noted to erode into the lateral aspect of the sternum and the first three ribs. To obtain a complete resection, a partial sternal and rib resection was performed. The resulting defect was reconstructed with Marlex mesh (Meadox Medical Systems, Oakland, NJ) and methyl methacrylate. Soft tissue coverage was obtained with a pectoralis major rotational flap. Final pathologic examination confirmed the diagnosis of proliferative myositis. At 1-year follow-up the patient is well and free of recurrence.
Comment Proliferative myositis is one of several benign, rapidly enlarging tumors of the soft tissue that are often confused with sarcoma. Although their rapid growth and oftenAccepted for publication Aug 14, 2001. Address reprint requests to Dr Altorki, Department of Cardiothoracic Surgery, The New York-Presbyterian Hospital, Suite F22, 525 East 68th St, New York, NY 10021; e-mail:
[email protected].
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