- Email: [email protected]
Proliferative myositis: a rare pseudosarcoma of the chest wall
1296
CASE REPORT KENT ET AL PROLIFERATIVE MYOSITIS
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
P
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].
0003-4975/02/$22.00 PII S0003-4975(01)03266-0
Ann Thorac Surg 2002;73:1296 – 8
Fig 1. Computed tomographic scan demonstrating erosion of the mass into the sternum and pectoralis major.
bizarre microscopic appearance may suggest an aggressive course, these pseudosarcomas are clinically benign and rarely recur after subtotal excision. The term pseudosarcoma encompasses three benign tumors of the soft tissue: nodular fasciitis, PM, and proliferative fasciitis. Of the three, PM is the least common, with fewer than 100 cases reported in the Englishlanguage literature. The first three cases were presented in abstract form by Ackerman in 1958 [1]. The disease was subsequently named by Kern, who reported seven cases in 1960 [2]. Enzinger and Dulcey reported the largest series of 33 cases in 1967 [3]. Although extremely rare, PM is an important clinical entity given its propensity for confusion with high-grade sarcoma. The clinical presentation of PM is nonspecific. Adults are usually affected with a peak incidence between 40 and 70 years. Reports of cases in children are extremely uncommon. Most patients report a rapidly enlarging, soft tissue mass, usually affecting the shoulder girdle and upper extremity. Proliferative myositis may also involve the soft tissue of the head and neck. In the series of 33 cases reported by Enzinger and Dulcey [3], 18 involved the shoulder and arm whereas only 4 cases arose from the anterior chest wall. The mass often evolves over the course of a few weeks, and may double in size during several days. The lesion is usually 3 to 6 cm at presentation, and is typically firm and deep-seated. Several microscopic features are characteristic of PM. First, a dense fibroblastic proliferation is noted to affect the epimysium, perimysium, and endomysium of the muscle bundles. This reaction is most pronounced in the septa surrounding the larger muscle fibers. Unlike sarcoma, PM rarely invades the surrounding muscle. On occasion, however, secondary muscle atrophy or focal ischemic necrosis may be observed. This alternation of proliferative connective tissue surrounding viable muscle fibers often gives rise to the so-called checkerboard effect, which is characteristic of the disease (Fig 2). Secondly, a proliferation of bizarre, giant cells is often seen within this connective tissue matrix. These cells resemble ganglion cells or rhabdomyoblasts, and their presence often leads to the misdiagnosis of malignancy,
CASE REPORT KENT ET AL PROLIFERATIVE MYOSITIS
1297
such as ganglioneuroblastoma or rhabdomyosarcoma. However, the immunohistochemical profile of these cells (vimentin and actin positive and myoglobin negative) suggests a myofibroblastic origin. In addition, electron microscopy has demonstrated these cells to contain fine microfilaments and a prominent rough endoplasmic reticulum, also consistent with a myofibroblast lineage [4]. These cells are universally diploid on flow cytometry, lending further evidence to the belief that these cells are not malignant [5]. The distinction between PM and other pseudosarcomas may be difficult even for the experienced pathologist. Microscopically, PM is indistinguishable from proliferative fasciitis. The latter, however, typically involves the subcutaneous tissue rather than the deeper muscle fascia [6]. Differentiation from nodular fasciitis is usually more straightforward. Nodular fasciitis often appears as a well-demarcated nodule that arises from the superficial fascia of the muscle [7]. Unlike PM, nodular fasciitis is common in children. Additionally, the microscopic appearance of nodular fasciitis is distinct, without the large, basophilic cells seen in PM. Although the precise cause is unknown, PM is believed to be a reactive, inflammatory process. Approximately one third of patients will report a history of recent trauma to the affected area. Subclinical vascular injury may also be a precipitating event. Microscopic evidence of endarteritis has been observed in the lesions of some patients who have a history of thrombophlebitis and pulmonary embolism [7]. The prognosis for patients with PM is excellent. Although operation is often performed for diagnosis and removal of a cosmetically disfiguring mass, recurrence even after simple excision (ie, with positive microscopic margins) is extremely rare. Indeed, with a follow-up period of 1 to 16 years no recurrences were noted in the series reported by Enzinger and Dulcey [3]. However, among the 33 patients reported, 14 had been misdiagnosed as sarcoma and had undergone radical resection. In contrast to sarcoma, PM rarely invades normal tissue and can usually be
Fig 2. Muscle fibers and surrounding connective tissue give rise to the “checkerboard effect” characteristic of proliferative myositis. (Hematoxylin & eosin ⫻40.)
1298
CASE REPORT OKUYAMA ET AL FISTULA HEALING AFTER ESOPHAGECTOMY
readily excised. The erosion of the thoracic cage seen in this case is distinctly uncommon. Even with subtotal resection, PM is unlikely to recur. Consultation with an experienced pathologist after an incisional biopsy will confirm the benign nature of the disease. The majority of patients, therefore, can be spared radical resection if the diagnosis has been made with certainty.
References 1. Ackerman LV. Extra-osseous localized non-neoplastic bone and cartilage formation (so-called myositis ossificans). J Bone Joint Surg Am 1958;40:279–98. 2. Kern WH. Proliferative myositis: a pseudosarcomatous reaction to injury. Arch Pathol 1960;69:209–16. 3. Enzinger FM, Dulcey F. Proliferative myositis: report of thirtythree cases. Cancer 1967;29:2213–23. 4. Craver JL, McDivitt RW. Proliferative fasciitis: ultrastructural study of two cases. Arch Pathol Lab Med 1981;105:542–5. 5. el-Jabbour JN, Wilson GD, Bennett MH, Burke MM, Davey AT, Eames K. Flow cytometric study of nodular fasciitis, proliferative fasciitis and proliferative myositis. Hum Pathol 1991;22:1146–9. 6. Chung EB, Enzinger FM. Proliferative myositis. Cancer 1975; 36:1450– 8. 7. Hutter R, Stewart W, Foote F. Fasciitis. A report of 70 cases with follow-up proving the benignity of the lesion. Cancer 1962;15:992–1003.
Histological Confirmation of Healing of Gastrobronchial Fistula Using A Muscle Flap Manabu Okuyama, MD, Reijiro Saito, MD, Satoru Motoyama, MD, Michihiko Kitamura, MD, and Jun-ichi Ogawa, MD Second Department of Surgery, Akita University School of Medicine, Akita and Department of Surgery, Isawa Prefectural Hospital, Iwate, Japan
We report a case of gastrobronchial fistula that developed after esophagectomy for esophageal cancer. The fistula was repaired successfully by transposing a pectoralis major muscle flap. Complete healing was confirmed histologically by epithelialization of the fistula site and at autopsy 12 months after surgery. Muscle flap transposition effectively repairs gastrobronchial fistula. (Ann Thorac Surg 2002;73:1298 –9) © 2002 by The Society of Thoracic Surgeons
B
ronchial or tracheal fistulas of the reconstructed stomach are rare complications of esophagectomy, but they are nonetheless life threatening and difficult to manage clinically. Although several groups have previously reported the successful repair of gastrobronchial fistula using muscle flaps [1– 4], no evidence of the
Accepted for publication July 31, 2001. Address reprint requests to Dr Okuyama, Second Department of Surgery, Akita University School of Medicine, 1-1-1 Hondo, Akita 010-8543, Japan; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
Ann Thorac Surg 2002;73:1298 –9
healing process has been documented to date. In this article, we confirm the successful repair of a gastrobronchial fistula using a muscle flap from histological findings. The perioperative management of this patient is documented in the Japanese literature [2], and the current report focuses on epithelialization observed at 12 postoperative months as well as autopsy findings of complete healing of the fistula. This report provides histological evidence of complete healing of gastrobronchial fistula following muscle flap transposition. A 72-year-old man was admitted to our institution in February 1998 due to dyspnea. Two years previously, he had undergone total thoracic esophagectomy with right thoracotomy accompanied by reconstruction of the stomach via the posterior mediastinal route for esophageal cancer. He received preoperative chemoradiation therapy and separate courses of postoperative chemotherapy and radiation therapy. Upon admission for sudden-onset dyspnea, bronchoscopy revealed the presence of a fistula localized to the membranous portion of the right intermediate bronchus, while gastroscopy revealed a fistula in an ulcerated area of the stomach. A biopsy specimen obtained from this ulcerated region showed no evidence of malignancy. The fistula gradually enlarged and the patient developed severe respiratory failure on the 10th day of hospitalization. An emergency operation to close the fistula was performed. In the dorsal position, a right pectoralis major muscle flap with a vascular pedicle consisting of the pectoral branch of the acromiothoracic trunk was mobilized. A segment of the right second costal cartilage was removed for transposition of the flap through the thorax. In the lateral position, the stomach was dissected from the trachea via a right thoracotomy. The opening in the stomach (3 cm in diameter) was closed directly by an absorbable interrupted suture. As the bronchial defect (1 cm in diameter) was too large to be closed directly, a muscle flap was used for indirect closure. The flap was sutured around the bronchial defect with an absorbable interrupted suture, and also interposed between the stomach and bronchus. Bronchoscopy to monitor the operative site and tracheobronchial toilet were conducted daily. The fistula was seen to be completely closed by the muscle flap. Granulation tissue gradually accumulated within the bronchus between 2 and 5 months after surgery, but decreased spontaneously. The fistula was undetectable endoscopically 10 months after surgery. Despite a good recovery from the gastrobronchial fistula repair, the patient died of renal failure 12 months after surgery. An autopsy revealed that the gastrobronchial fistula had indeed completely healed and was visible only by a fine white scar. No narrowing or stricture of the bronchus was observed. Histologic examination found that ciliated epithelial cells partially covered the accumulation of granulation tissue at the bronchial defect (Figs 1 and 2). 0003-4975/02/$22.00 PII S0003-4975(01)03238-6
CASE REPORT KENT ET AL PROLIFERATIVE MYOSITIS
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
P
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].
0003-4975/02/$22.00 PII S0003-4975(01)03266-0
Ann Thorac Surg 2002;73:1296 – 8
Fig 1. Computed tomographic scan demonstrating erosion of the mass into the sternum and pectoralis major.
bizarre microscopic appearance may suggest an aggressive course, these pseudosarcomas are clinically benign and rarely recur after subtotal excision. The term pseudosarcoma encompasses three benign tumors of the soft tissue: nodular fasciitis, PM, and proliferative fasciitis. Of the three, PM is the least common, with fewer than 100 cases reported in the Englishlanguage literature. The first three cases were presented in abstract form by Ackerman in 1958 [1]. The disease was subsequently named by Kern, who reported seven cases in 1960 [2]. Enzinger and Dulcey reported the largest series of 33 cases in 1967 [3]. Although extremely rare, PM is an important clinical entity given its propensity for confusion with high-grade sarcoma. The clinical presentation of PM is nonspecific. Adults are usually affected with a peak incidence between 40 and 70 years. Reports of cases in children are extremely uncommon. Most patients report a rapidly enlarging, soft tissue mass, usually affecting the shoulder girdle and upper extremity. Proliferative myositis may also involve the soft tissue of the head and neck. In the series of 33 cases reported by Enzinger and Dulcey [3], 18 involved the shoulder and arm whereas only 4 cases arose from the anterior chest wall. The mass often evolves over the course of a few weeks, and may double in size during several days. The lesion is usually 3 to 6 cm at presentation, and is typically firm and deep-seated. Several microscopic features are characteristic of PM. First, a dense fibroblastic proliferation is noted to affect the epimysium, perimysium, and endomysium of the muscle bundles. This reaction is most pronounced in the septa surrounding the larger muscle fibers. Unlike sarcoma, PM rarely invades the surrounding muscle. On occasion, however, secondary muscle atrophy or focal ischemic necrosis may be observed. This alternation of proliferative connective tissue surrounding viable muscle fibers often gives rise to the so-called checkerboard effect, which is characteristic of the disease (Fig 2). Secondly, a proliferation of bizarre, giant cells is often seen within this connective tissue matrix. These cells resemble ganglion cells or rhabdomyoblasts, and their presence often leads to the misdiagnosis of malignancy,
CASE REPORT KENT ET AL PROLIFERATIVE MYOSITIS
1297
such as ganglioneuroblastoma or rhabdomyosarcoma. However, the immunohistochemical profile of these cells (vimentin and actin positive and myoglobin negative) suggests a myofibroblastic origin. In addition, electron microscopy has demonstrated these cells to contain fine microfilaments and a prominent rough endoplasmic reticulum, also consistent with a myofibroblast lineage [4]. These cells are universally diploid on flow cytometry, lending further evidence to the belief that these cells are not malignant [5]. The distinction between PM and other pseudosarcomas may be difficult even for the experienced pathologist. Microscopically, PM is indistinguishable from proliferative fasciitis. The latter, however, typically involves the subcutaneous tissue rather than the deeper muscle fascia [6]. Differentiation from nodular fasciitis is usually more straightforward. Nodular fasciitis often appears as a well-demarcated nodule that arises from the superficial fascia of the muscle [7]. Unlike PM, nodular fasciitis is common in children. Additionally, the microscopic appearance of nodular fasciitis is distinct, without the large, basophilic cells seen in PM. Although the precise cause is unknown, PM is believed to be a reactive, inflammatory process. Approximately one third of patients will report a history of recent trauma to the affected area. Subclinical vascular injury may also be a precipitating event. Microscopic evidence of endarteritis has been observed in the lesions of some patients who have a history of thrombophlebitis and pulmonary embolism [7]. The prognosis for patients with PM is excellent. Although operation is often performed for diagnosis and removal of a cosmetically disfiguring mass, recurrence even after simple excision (ie, with positive microscopic margins) is extremely rare. Indeed, with a follow-up period of 1 to 16 years no recurrences were noted in the series reported by Enzinger and Dulcey [3]. However, among the 33 patients reported, 14 had been misdiagnosed as sarcoma and had undergone radical resection. In contrast to sarcoma, PM rarely invades normal tissue and can usually be
Fig 2. Muscle fibers and surrounding connective tissue give rise to the “checkerboard effect” characteristic of proliferative myositis. (Hematoxylin & eosin ⫻40.)
1298
CASE REPORT OKUYAMA ET AL FISTULA HEALING AFTER ESOPHAGECTOMY
readily excised. The erosion of the thoracic cage seen in this case is distinctly uncommon. Even with subtotal resection, PM is unlikely to recur. Consultation with an experienced pathologist after an incisional biopsy will confirm the benign nature of the disease. The majority of patients, therefore, can be spared radical resection if the diagnosis has been made with certainty.
References 1. Ackerman LV. Extra-osseous localized non-neoplastic bone and cartilage formation (so-called myositis ossificans). J Bone Joint Surg Am 1958;40:279–98. 2. Kern WH. Proliferative myositis: a pseudosarcomatous reaction to injury. Arch Pathol 1960;69:209–16. 3. Enzinger FM, Dulcey F. Proliferative myositis: report of thirtythree cases. Cancer 1967;29:2213–23. 4. Craver JL, McDivitt RW. Proliferative fasciitis: ultrastructural study of two cases. Arch Pathol Lab Med 1981;105:542–5. 5. el-Jabbour JN, Wilson GD, Bennett MH, Burke MM, Davey AT, Eames K. Flow cytometric study of nodular fasciitis, proliferative fasciitis and proliferative myositis. Hum Pathol 1991;22:1146–9. 6. Chung EB, Enzinger FM. Proliferative myositis. Cancer 1975; 36:1450– 8. 7. Hutter R, Stewart W, Foote F. Fasciitis. A report of 70 cases with follow-up proving the benignity of the lesion. Cancer 1962;15:992–1003.
Histological Confirmation of Healing of Gastrobronchial Fistula Using A Muscle Flap Manabu Okuyama, MD, Reijiro Saito, MD, Satoru Motoyama, MD, Michihiko Kitamura, MD, and Jun-ichi Ogawa, MD Second Department of Surgery, Akita University School of Medicine, Akita and Department of Surgery, Isawa Prefectural Hospital, Iwate, Japan
We report a case of gastrobronchial fistula that developed after esophagectomy for esophageal cancer. The fistula was repaired successfully by transposing a pectoralis major muscle flap. Complete healing was confirmed histologically by epithelialization of the fistula site and at autopsy 12 months after surgery. Muscle flap transposition effectively repairs gastrobronchial fistula. (Ann Thorac Surg 2002;73:1298 –9) © 2002 by The Society of Thoracic Surgeons
B
ronchial or tracheal fistulas of the reconstructed stomach are rare complications of esophagectomy, but they are nonetheless life threatening and difficult to manage clinically. Although several groups have previously reported the successful repair of gastrobronchial fistula using muscle flaps [1– 4], no evidence of the
Accepted for publication July 31, 2001. Address reprint requests to Dr Okuyama, Second Department of Surgery, Akita University School of Medicine, 1-1-1 Hondo, Akita 010-8543, Japan; e-mail: [email protected].
© 2002 by The Society of Thoracic Surgeons Published by Elsevier Science Inc
Ann Thorac Surg 2002;73:1298 –9
healing process has been documented to date. In this article, we confirm the successful repair of a gastrobronchial fistula using a muscle flap from histological findings. The perioperative management of this patient is documented in the Japanese literature [2], and the current report focuses on epithelialization observed at 12 postoperative months as well as autopsy findings of complete healing of the fistula. This report provides histological evidence of complete healing of gastrobronchial fistula following muscle flap transposition. A 72-year-old man was admitted to our institution in February 1998 due to dyspnea. Two years previously, he had undergone total thoracic esophagectomy with right thoracotomy accompanied by reconstruction of the stomach via the posterior mediastinal route for esophageal cancer. He received preoperative chemoradiation therapy and separate courses of postoperative chemotherapy and radiation therapy. Upon admission for sudden-onset dyspnea, bronchoscopy revealed the presence of a fistula localized to the membranous portion of the right intermediate bronchus, while gastroscopy revealed a fistula in an ulcerated area of the stomach. A biopsy specimen obtained from this ulcerated region showed no evidence of malignancy. The fistula gradually enlarged and the patient developed severe respiratory failure on the 10th day of hospitalization. An emergency operation to close the fistula was performed. In the dorsal position, a right pectoralis major muscle flap with a vascular pedicle consisting of the pectoral branch of the acromiothoracic trunk was mobilized. A segment of the right second costal cartilage was removed for transposition of the flap through the thorax. In the lateral position, the stomach was dissected from the trachea via a right thoracotomy. The opening in the stomach (3 cm in diameter) was closed directly by an absorbable interrupted suture. As the bronchial defect (1 cm in diameter) was too large to be closed directly, a muscle flap was used for indirect closure. The flap was sutured around the bronchial defect with an absorbable interrupted suture, and also interposed between the stomach and bronchus. Bronchoscopy to monitor the operative site and tracheobronchial toilet were conducted daily. The fistula was seen to be completely closed by the muscle flap. Granulation tissue gradually accumulated within the bronchus between 2 and 5 months after surgery, but decreased spontaneously. The fistula was undetectable endoscopically 10 months after surgery. Despite a good recovery from the gastrobronchial fistula repair, the patient died of renal failure 12 months after surgery. An autopsy revealed that the gastrobronchial fistula had indeed completely healed and was visible only by a fine white scar. No narrowing or stricture of the bronchus was observed. Histologic examination found that ciliated epithelial cells partially covered the accumulation of granulation tissue at the bronchial defect (Figs 1 and 2). 0003-4975/02/$22.00 PII S0003-4975(01)03238-6