- Email: [email protected]
Elastofibroma Dorsi: Clinicopathologic Review of 6 Cases
1894
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
FEATURE ARTICLES
tumor (mucocele) appears malignant and appendicectomy if it appears benign. This could be a difficult decision to make. More recent protocols consist of aggressive cytoreductive surgery, including total omentectomy, and often splenectomy, rectosigmoid colectomy, and resection of other involved organs, followed by heated intraperitoneal chemotherapy. This treatment has been pioneered and developed by Sugarbaker and colleagues [2], who have just reported a 63% survival at 5 years from a series of 385 patients treated with this approach. The natural history of this disease is such that they state “in the absence of this [cytoreductive surgery and intraperitoneal chemotherapy] approach no long term survivors have ever been reported in the medical literature” [3]. Mortman and colleagues [4] have also reported 3 patients with metastatic pulmonary MCA appendix. All underwent initial cytoreductive surgery with heated intraperitoneal chemotherapy. On discovering their lung metastases on follow-up, 2 patients underwent further lobectomy with lymphadenectomy and 1 was treated with wedge resection. All were reported as being disease-free at 2 to 8 years of follow-up. The case presented here reports recurrent pulmonary metastatic MCA appendix and is one of a handful of cases of pulmonary metastases reported in the literature to date. Also, despite receiving suboptimal treatment at presentation compared with recent opinion, this patient has defied all known expectations and is currently well 20 years after initial diagnosis. Finally, serial metastasectomy was well tolerated, with no deterioration in lung function. This observation was probably due to excision of nonfunctional lung tissue, allowing remaining viable lung to expand. In conclusion, pulmonary metastasis of appendiceal MCA with PMP is very rare. Moreover, recurrence can occur many years after initial presentation, suggesting that such patients should have lifelong follow-up with at least an annual chest roentgenogram. Serial lung metas-
Ann Thorac Surg 2007;83:1894 –7
Fig 3. Immunohistochemical section from recent lung metastasis specimen stained positive for cytokeratin-20 antibody, confirming the abdominal origin of the mucinous cystadenocarcinoma (⫻200 original magnification).
tasectomy is well tolerated but cannot be regarded as curative. Also, this case of prolonged survival challenges currently accepted views of the natural history of this disease.
References 1. Deans GT, Spence RA. Neoplastic lesions of the appendix. Br J Surg 1995;82:299 –306. 2. Sugarbaker PH, Chang D. Results of treatment of 385 patients with peritoneal surface spread of appendiceal malignancy. Ann Surg Oncol 1999;6:727–31. 3. Dhage-Ivatury S, Sugarbaker PH. Update on the surgical approach to mucocele of the appendix. J Am Coll Surg 2006; 202:680 – 4. 4. Mortman KD, Sugarbaker PA, Shmookler BM, DeGuzman VC, Soberman MS. Pulmonary metastases in pseudomyxoma peritonei syndrome. Ann Thorac Surg 1997;64:1434 – 6. 5. Lee BY, Kim HS, Lee SH, et al. Pseudomyxoma peritonei: extraperitoneal spread to the pleural cavity and lung. J Thorac Imaging 2004;19:123– 6.
Elastofibroma Dorsi: Clinicopathologic Review of 6 Cases Keith D. Mortman, MD, Gary M. Hochheiser, MD, Erica M. Giblin, MD, Yorell Manon-Matos, MD, and Kenneth M. Frankel, MD Division of Thoracic Surgery, Baystate Medical Center, Springfield, Massachusetts
Elastofibroma dorsi is a rare, benign lesion arising from connective tissue and usually found at the angle of the scapula. Surgical resection is often indicated in the presFig 2. Hematoxylin and eosin staining shows the tumor consists of lakes of mucin lined by goblet cells and set within a fibrous stroma (⫻200 original magnification). © 2007 by The Society of Thoracic Surgeons Published by Elsevier Inc
Accepted for publication Nov 16, 2006. Address correspondence to Dr Mortman, 2 Medical Center Dr, Suite 504, Springfield, MA 01107; e-mail: [email protected].
0003-4975/07/$32.00 doi:10.1016/j.athoracsur.2006.11.050
Ann Thorac Surg 2007;83:1894 –7
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
1895
ence of an enlarging mass or when malignancy can not be excluded. Herein we report our most recent case of elastofibroma dorsi and our review of 6 cases from the past 16 years. (Ann Thorac Surg 2007;83:1894 –7) © 2007 by The Society of Thoracic Surgeons irst described by Jarvi and Saxen [1] in 1961, elastofibroma dorsi has typically been found in elderly women and mostly on the right side. Subsequent articles note bilaterality of the lesions in as many as 66% of patients. Elastofibroma is a nonencapsulated lesion composed of fibrous tissue forming collagen bundles. It may be densely adherent to surrounding muscle and bone, and therefore the suspicion for malignancy is often raised. It is postulated that the degeneration of connective tissue is secondary to friction of the scapula against the rib cage caused by repetitive use of the shoulder girdle [1]. Thus it has often been described in patients performing repeated manual labor. Ultrasound, computed tomography, magnetic resonance imaging, and positron emission tomography have all been used to characterize elastofibroma [2– 6]. Surgical extirpation is usually curative with only one documented recurrence [7]. A 64-year-old man was referred to our institution for evaluation of an enlarging left subscapular mass. The mass was first noticed 10 years previously, but the size recently increased. The patient’s hobby is cutting wood with repetitive overhead movements of the arms. He recently noticed discomfort in the back and shoulder girdle while performing this task. Physical examination was significant for an outward displacement of the left scapula with a large ill-defined mass located at its tip. There was no tenderness or overlying skin changes. A computed tomographic scan (Fig 1) showed stranding in the fat bilaterally adjacent to the serratus and subscapularis musculature. A magnetic resonance image demonstrated an 8 ⫻ 3 cm mass deep to the left latissimus dorsi muscle, extending deep to the left serratus anterior muscle, and deep to the inferior scapular margin. There was a similar, but smaller, mass in the right chest wall. A percutaneous biopsy was consistent with elastofibroma dorsi. As the left chest wall mass was growing and the patient was increasingly symptomatic, surgical resection was recommended. Through a left lateral, musclesparing approach, the chest wall mass was completely excised (Fig 2). Final histology revealed a 13.5 ⫻ 12.5 ⫻ 4.0 cm elastofibroma. Verhoeff’s elastic tissue stain highlighted the branching and serrated elastic fibers (Fig 3). The patient had an uneventful postoperative course and is doing well 18 months after surgery with no clinical evidence of recurrence. A review of our pathology database found 5 additional cases of elastofibroma dorsi from the previous 16 years. Our first patient presented in 1989 with a 6-month history of a painless, enlarging mass at the tip of the left scapula. She underwent local excision through a posterolateral
Fig 1. Computed tomographic scan demonstrating bilateral soft tissue masses (arrows). The larger lesion on the left is displacing the serratus anterior muscle and the angle of the scapula posterolaterally.
incision and was lost to follow-up soon thereafter. Four additional patients were found to have incidental elastofibromas at the time of thoracotomy and pulmonary resection for cancer; three of these patients had nonsmall cell lung cancer and 1 patient had metastatic colon cancer. These 4 patients respectively had presenting symptoms of chest pain, cough, weight loss, and confusion that were attributed to their cancer diagnosis, and not to the elastofibroma. Although the present patient of this case report had bilateral elastofibromas, all previous patients in our series had unilateral lesions (left, 3; right, 2). In all 6 of our patients (3 males), the surgically resected elastofibromas were located at the tip of the scapula. Excluding the first patient who was lost to follow-up, there was no evidence of recurrent chest wall mass at a median length of follow-up of 36 months.
Comment Although 99% of the cases of elastofibroma are localized to the lower subscapular region, deep in the rhomboid and latissimus dorsi muscles, other unusual locations have been noted in the literature [8]. These locations include the deltoid muscle, ischial tuberosity, greater trochanter, olecranon, thoracic wall, axilla, foot, stomach, rectum, spinal canal, mediastinum, and cornea [9]. These “pseudotumors” can be firmly adherent to the thoracic fascia, periosteum, or ligaments in the regions of the seventh and eighth ribs. On physical examination these masses are wellcircumscribed and nonadherent to the overlying skin. The scapula may overlie the lesion thereby masking it. In
FEATURE ARTICLES
F
1896
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
FEATURE ARTICLES
a case report, Dalal and colleagues [3] noted that on an initial physical examination of a 78-year-old man there was no palpable subscapular mass noted. Also a magnetic resonance image and an ultrasound failed to identify the lesion until the patient abducted his arm [3]. This allowed the mass to emerge from where it was being compressed deep to the scapula. The differential diagnosis of such a soft tissue mass includes lipoma, liposarcomas, sarcoma, fibromatosis, neurofibroma, histiocytoma, desmoid, and hemangioma. Because the presence of a malignancy can not be completely excluded, these benign soft tissue masses are often excised. Recurrence after incomplete surgical excision has been reported only once [7]. There are no reports of malignant transformation of elastofibroma. A right-sided predominance has been previously described; however 10% to 66% of cases are bilateral. Synchronous lesions in the infra-olecranon area have been reported [7]. This pseudotumor is most often diagnosed in the elderly population. Age ranges are typically 35 to 94 years old; however elastofibroma has been described in patients as young as 6 years of age [10]. The literature quotes upward of 93% of individuals affected as being female. However, several studies (including this one) have suggested an equal gender predisposition. Pathologic examination of elastofibroma reveals a fibrous, nonencapsulated mass with streaks of fat within the tissue. Some elastofibromas contain cysts. Histologically the mass is hypocellular and contains a mixture of collagen, elastic fibers, and fat. The elastic fibers have a characteristic beaded appearance. The elastic fibers may also be fragmented into small globules. Ultrasound examination of elastofibroma has demonstrated characteristic findings. The masses are noted to have a fasiculated structure with regular alternating hyperechoic and hypoechoic striae of varying thickness [2]. The lesions are typically not adherent to the surrounding musculature. The masses may have irregular margins and structurally appear to be similar to the surrounding musculature [2].
Ann Thorac Surg 2007;83:1894 –7
Fig 3. Verhoeff’s elastic stain highlighting the elastic fibers (⫻200 original magnification).
The typical appearance of elastofibroma on computed tomographic scan reveals a poorly circumscribed mass which is isodense to surrounding muscle [11]. The prevalence of elastofibroma on computed tomographic scan is 2% [12]. These masses are often difficult to delineate from surrounding muscle planes. Magnetic resonance imaging will show soft tissue masses with streaks of fat and signal intensities similar to skeletal muscle [11]. Some elastofibromas have been described as having low signal intensity on T2 weighted images and this is believed to be secondary to a decrease in cellularity and increased collagen formation. Elastofibroma dorsi is a benign connective tissue tumor that is characteristically found at the angle of the scapula. Early reports of female and right-sided preponderance are contradicted by more recent case series. Although patients with elastofibroma are often asymptomatic, the most common symptom is a painless, enlarging mass. Oftentimes elastofibroma is found incidentally either during a radiographic evaluation (such as for lung cancer) or at the time of thoracotomy. Its resection is considered curative with only one documented recurrence.
References
Fig 2. Intraoperative photograph depicting left elastofibroma measuring 13.5 ⫻ 12.5 ⫻ 4.0 cm, which was excised through a musclesparing incision made along the anterior margin of the latissimus muscle.
1. Jarvi OH, Saxen AE. Elastofibroma dorsi. Acta Pathol Microbiol Scand 1961;144(Suppl 51):83– 4. 2. Solivetti FM, Bacaro D, Sidozzi AD, Cecconi P. Elastofibroma dorsi: ultrasound pattern in three patients. J Exp Clin Cancer Res 2003;22:565–9. 3. Dalal A, Miller TT, Kenan S. Sonographic detection of elastofibroma dorsi. J Clin Ultrasound 2003;31:375– 8. 4. Abe S, Miyata, N, Yamamoto Y, et al. Elastofibroma dorsi: CT, MRI, and pathologic findings. Plast Reconstr Surg 1999; 104:2121– 6. 5. Patrikeos A, Breidahl W, Robins P. F-18 FDG uptake associated with elastofibroma dorsi. Clin Nucl Med 2005;30:617– 8.
6. Pierce J, Henderson R. Hypermetabolism of elastofibroma dorsi on PET-CT. AJR 2004;183:35–7. 7. Nagamine N, Nohara Y, Ito E. Elastofibroma in Okinawa: a clinicopathologic study of 170 cases. Cancer 1982;50:1794 – 805. 8. Nielsen T, Sneppen O, Myhre-Jensen O, Daugaard S, Norback J. Subscapular elastofibroma: a reactive pseudotumor. J Shoulder Elbow Surg 1996;5:209 –13. 9. Briccoli A, Casadei R, Di Renzo M, Favale L, Bacchini P, Bertoni F. Elastofibroma dorsi. Surg Today 2000;30:147–52. 10. Marin ML, Perzin KH, Markowitz AM. Elastofibroma dorsi: benign chest wall tumor. J Thorac Cardiovasc Surg 1989;98: 234 – 8. 11. Kransdorf MJ, Meis JM, Montgomery E. Elastofibroma: MR and CT appearance with radiologic-pathologic correlation. Am J Roentgenol 1992;159:575–9. 12. Brandser EA, Goree JC, El-Khoury GY. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. AJR 1998;171:977– 80.
Tracheal Stenting of Iatrogenic Tracheal Injury: A Novel Management Approach Adam C. Yopp, MD, Jeremy G. Eckstein, MD, Richard H. Savel, MD, and Sunil Abrol, MD Department of Surgery, Division of Cardiothoracic Surgery, Maimonides Medical Center, Brooklyn, New York
We report the case of a patient who had an intubationrelated tracheal injury who we treated by deployment of a covered tracheal stent. This approach may be preferable to other alternatives in patients with a prohibitive risk of mortality with surgical repair or in an injury with sequelae not suitable for conservative management. (Ann Thorac Surg 2007;83:1897–9) © 2007 by The Society of Thoracic Surgeons
I
atrogenic injury to the tracheobronchial tree after intubation is an extremely rare occurrence that historically has been managed either expectantly or with a highly morbid surgical reconstruction. We report of the successful use of a tracheal stent to repair an intubationassociated tracheal injury.
CASE REPORT YOPP ET AL TRACHEAL STENTING OF IATROGENIC INJURY
1897
the patient was extubated without incident in the operating room. Approximately 2 hours after the conclusion of the procedure, progressively worsening respiratory distress developed. Physical examination revealed minimal subcutaneous emphysema, and a chest roentgenogram revealed no abnormalities. Computed chest tomography revealed a defect of the posterior trachea, approximately 1.5 cm proximal to the carina, as well as widespread pneumomediastinum (Fig 1). Bronchoscopic examination of the trachea confirmed a 4.0 cm tear of the posterior membranous trachea, 1.5 cm proximal to the carina (Fig 2). Given the worsening subcutaneous emphysema and increasing respiratory compromise requiring mechanical ventilation, the patient was immediately taken to the operating room. Because of the patient’s severe underlying pulmonary comorbidity, a decision was made to use a tracheal stent to repair the injury rather than a more invasive surgical reconstruction. A 6-cm ⫻ 14-mm selfexpanding, covered metallic stent (Microinvasive, Natick, MA) was deployed over the tracheal injury and 0.5 cm proximal to the carina under bronchoscopic and fluoroscopic guidance. The patient was started on broad-spectrum antibiotics and was successfully extubated 2 days later. After an unremarkable further hospital course, the patient was discharged home on postoperative day 5. The patient underwent monthly flexible bronchoscopic surveillance, which documented only minimal formation of granulation tissue. At 4 months, excess respiratory secretions developed, and flexible bronchoscopy revealed significant granulation tissue at the proximal end of the covered stent. The tracheal stent was removed using rigid forceps through rigid bronchoscopy, with only slight difficulty. The underlying trachea was well healed (Fig 3). During the procedure, ventilation and oxygenation were ensured by high-frequency jet ventilation. The patient was discharged home the next day and remains asymptomatic 1 year later.
A 75-year-old woman with a history of chronic obstructive pulmonary disease, idiopathic pulmonary hypertension, and severe cervical-thoracic spine scoliosis underwent repair of a humeral head fracture. The procedure was initially done with a scalene nerve block, but owing to ipsilateral phrenic nerve paralysis, the patient went into respiratory distress and required intubation. The intubation was difficult because of aberrant cervical anatomy in the setting of her spinal disease. A fiberoptic bronchoscopic intubation was performed after multiple unsuccessful attempts at intubation via direct laryngoscopy. The intraoperative course was unremarkable, and Accepted for publication Dec 13, 2006. Address correspondence to Dr Yopp, Maimonides Medical Center, 4802 Tenth Ave, Brooklyn, NY 11219; e-mail: [email protected].
© 2007 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Computed chest tomography reveals a defect in the posterior trachea, 1.5 cm proximal to the carina (white arrow). 0003-4975/07/$32.00 doi:10.1016/j.athoracsur.2006.12.040
FEATURE ARTICLES
Ann Thorac Surg 2007;83:1897–9
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
FEATURE ARTICLES
tumor (mucocele) appears malignant and appendicectomy if it appears benign. This could be a difficult decision to make. More recent protocols consist of aggressive cytoreductive surgery, including total omentectomy, and often splenectomy, rectosigmoid colectomy, and resection of other involved organs, followed by heated intraperitoneal chemotherapy. This treatment has been pioneered and developed by Sugarbaker and colleagues [2], who have just reported a 63% survival at 5 years from a series of 385 patients treated with this approach. The natural history of this disease is such that they state “in the absence of this [cytoreductive surgery and intraperitoneal chemotherapy] approach no long term survivors have ever been reported in the medical literature” [3]. Mortman and colleagues [4] have also reported 3 patients with metastatic pulmonary MCA appendix. All underwent initial cytoreductive surgery with heated intraperitoneal chemotherapy. On discovering their lung metastases on follow-up, 2 patients underwent further lobectomy with lymphadenectomy and 1 was treated with wedge resection. All were reported as being disease-free at 2 to 8 years of follow-up. The case presented here reports recurrent pulmonary metastatic MCA appendix and is one of a handful of cases of pulmonary metastases reported in the literature to date. Also, despite receiving suboptimal treatment at presentation compared with recent opinion, this patient has defied all known expectations and is currently well 20 years after initial diagnosis. Finally, serial metastasectomy was well tolerated, with no deterioration in lung function. This observation was probably due to excision of nonfunctional lung tissue, allowing remaining viable lung to expand. In conclusion, pulmonary metastasis of appendiceal MCA with PMP is very rare. Moreover, recurrence can occur many years after initial presentation, suggesting that such patients should have lifelong follow-up with at least an annual chest roentgenogram. Serial lung metas-
Ann Thorac Surg 2007;83:1894 –7
Fig 3. Immunohistochemical section from recent lung metastasis specimen stained positive for cytokeratin-20 antibody, confirming the abdominal origin of the mucinous cystadenocarcinoma (⫻200 original magnification).
tasectomy is well tolerated but cannot be regarded as curative. Also, this case of prolonged survival challenges currently accepted views of the natural history of this disease.
References 1. Deans GT, Spence RA. Neoplastic lesions of the appendix. Br J Surg 1995;82:299 –306. 2. Sugarbaker PH, Chang D. Results of treatment of 385 patients with peritoneal surface spread of appendiceal malignancy. Ann Surg Oncol 1999;6:727–31. 3. Dhage-Ivatury S, Sugarbaker PH. Update on the surgical approach to mucocele of the appendix. J Am Coll Surg 2006; 202:680 – 4. 4. Mortman KD, Sugarbaker PA, Shmookler BM, DeGuzman VC, Soberman MS. Pulmonary metastases in pseudomyxoma peritonei syndrome. Ann Thorac Surg 1997;64:1434 – 6. 5. Lee BY, Kim HS, Lee SH, et al. Pseudomyxoma peritonei: extraperitoneal spread to the pleural cavity and lung. J Thorac Imaging 2004;19:123– 6.
Elastofibroma Dorsi: Clinicopathologic Review of 6 Cases Keith D. Mortman, MD, Gary M. Hochheiser, MD, Erica M. Giblin, MD, Yorell Manon-Matos, MD, and Kenneth M. Frankel, MD Division of Thoracic Surgery, Baystate Medical Center, Springfield, Massachusetts
Elastofibroma dorsi is a rare, benign lesion arising from connective tissue and usually found at the angle of the scapula. Surgical resection is often indicated in the presFig 2. Hematoxylin and eosin staining shows the tumor consists of lakes of mucin lined by goblet cells and set within a fibrous stroma (⫻200 original magnification). © 2007 by The Society of Thoracic Surgeons Published by Elsevier Inc
Accepted for publication Nov 16, 2006. Address correspondence to Dr Mortman, 2 Medical Center Dr, Suite 504, Springfield, MA 01107; e-mail: [email protected].
0003-4975/07/$32.00 doi:10.1016/j.athoracsur.2006.11.050
Ann Thorac Surg 2007;83:1894 –7
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
1895
ence of an enlarging mass or when malignancy can not be excluded. Herein we report our most recent case of elastofibroma dorsi and our review of 6 cases from the past 16 years. (Ann Thorac Surg 2007;83:1894 –7) © 2007 by The Society of Thoracic Surgeons irst described by Jarvi and Saxen [1] in 1961, elastofibroma dorsi has typically been found in elderly women and mostly on the right side. Subsequent articles note bilaterality of the lesions in as many as 66% of patients. Elastofibroma is a nonencapsulated lesion composed of fibrous tissue forming collagen bundles. It may be densely adherent to surrounding muscle and bone, and therefore the suspicion for malignancy is often raised. It is postulated that the degeneration of connective tissue is secondary to friction of the scapula against the rib cage caused by repetitive use of the shoulder girdle [1]. Thus it has often been described in patients performing repeated manual labor. Ultrasound, computed tomography, magnetic resonance imaging, and positron emission tomography have all been used to characterize elastofibroma [2– 6]. Surgical extirpation is usually curative with only one documented recurrence [7]. A 64-year-old man was referred to our institution for evaluation of an enlarging left subscapular mass. The mass was first noticed 10 years previously, but the size recently increased. The patient’s hobby is cutting wood with repetitive overhead movements of the arms. He recently noticed discomfort in the back and shoulder girdle while performing this task. Physical examination was significant for an outward displacement of the left scapula with a large ill-defined mass located at its tip. There was no tenderness or overlying skin changes. A computed tomographic scan (Fig 1) showed stranding in the fat bilaterally adjacent to the serratus and subscapularis musculature. A magnetic resonance image demonstrated an 8 ⫻ 3 cm mass deep to the left latissimus dorsi muscle, extending deep to the left serratus anterior muscle, and deep to the inferior scapular margin. There was a similar, but smaller, mass in the right chest wall. A percutaneous biopsy was consistent with elastofibroma dorsi. As the left chest wall mass was growing and the patient was increasingly symptomatic, surgical resection was recommended. Through a left lateral, musclesparing approach, the chest wall mass was completely excised (Fig 2). Final histology revealed a 13.5 ⫻ 12.5 ⫻ 4.0 cm elastofibroma. Verhoeff’s elastic tissue stain highlighted the branching and serrated elastic fibers (Fig 3). The patient had an uneventful postoperative course and is doing well 18 months after surgery with no clinical evidence of recurrence. A review of our pathology database found 5 additional cases of elastofibroma dorsi from the previous 16 years. Our first patient presented in 1989 with a 6-month history of a painless, enlarging mass at the tip of the left scapula. She underwent local excision through a posterolateral
Fig 1. Computed tomographic scan demonstrating bilateral soft tissue masses (arrows). The larger lesion on the left is displacing the serratus anterior muscle and the angle of the scapula posterolaterally.
incision and was lost to follow-up soon thereafter. Four additional patients were found to have incidental elastofibromas at the time of thoracotomy and pulmonary resection for cancer; three of these patients had nonsmall cell lung cancer and 1 patient had metastatic colon cancer. These 4 patients respectively had presenting symptoms of chest pain, cough, weight loss, and confusion that were attributed to their cancer diagnosis, and not to the elastofibroma. Although the present patient of this case report had bilateral elastofibromas, all previous patients in our series had unilateral lesions (left, 3; right, 2). In all 6 of our patients (3 males), the surgically resected elastofibromas were located at the tip of the scapula. Excluding the first patient who was lost to follow-up, there was no evidence of recurrent chest wall mass at a median length of follow-up of 36 months.
Comment Although 99% of the cases of elastofibroma are localized to the lower subscapular region, deep in the rhomboid and latissimus dorsi muscles, other unusual locations have been noted in the literature [8]. These locations include the deltoid muscle, ischial tuberosity, greater trochanter, olecranon, thoracic wall, axilla, foot, stomach, rectum, spinal canal, mediastinum, and cornea [9]. These “pseudotumors” can be firmly adherent to the thoracic fascia, periosteum, or ligaments in the regions of the seventh and eighth ribs. On physical examination these masses are wellcircumscribed and nonadherent to the overlying skin. The scapula may overlie the lesion thereby masking it. In
FEATURE ARTICLES
F
1896
CASE REPORT MORTMAN ET AL ELASTOFIBROMA DORSI
FEATURE ARTICLES
a case report, Dalal and colleagues [3] noted that on an initial physical examination of a 78-year-old man there was no palpable subscapular mass noted. Also a magnetic resonance image and an ultrasound failed to identify the lesion until the patient abducted his arm [3]. This allowed the mass to emerge from where it was being compressed deep to the scapula. The differential diagnosis of such a soft tissue mass includes lipoma, liposarcomas, sarcoma, fibromatosis, neurofibroma, histiocytoma, desmoid, and hemangioma. Because the presence of a malignancy can not be completely excluded, these benign soft tissue masses are often excised. Recurrence after incomplete surgical excision has been reported only once [7]. There are no reports of malignant transformation of elastofibroma. A right-sided predominance has been previously described; however 10% to 66% of cases are bilateral. Synchronous lesions in the infra-olecranon area have been reported [7]. This pseudotumor is most often diagnosed in the elderly population. Age ranges are typically 35 to 94 years old; however elastofibroma has been described in patients as young as 6 years of age [10]. The literature quotes upward of 93% of individuals affected as being female. However, several studies (including this one) have suggested an equal gender predisposition. Pathologic examination of elastofibroma reveals a fibrous, nonencapsulated mass with streaks of fat within the tissue. Some elastofibromas contain cysts. Histologically the mass is hypocellular and contains a mixture of collagen, elastic fibers, and fat. The elastic fibers have a characteristic beaded appearance. The elastic fibers may also be fragmented into small globules. Ultrasound examination of elastofibroma has demonstrated characteristic findings. The masses are noted to have a fasiculated structure with regular alternating hyperechoic and hypoechoic striae of varying thickness [2]. The lesions are typically not adherent to the surrounding musculature. The masses may have irregular margins and structurally appear to be similar to the surrounding musculature [2].
Ann Thorac Surg 2007;83:1894 –7
Fig 3. Verhoeff’s elastic stain highlighting the elastic fibers (⫻200 original magnification).
The typical appearance of elastofibroma on computed tomographic scan reveals a poorly circumscribed mass which is isodense to surrounding muscle [11]. The prevalence of elastofibroma on computed tomographic scan is 2% [12]. These masses are often difficult to delineate from surrounding muscle planes. Magnetic resonance imaging will show soft tissue masses with streaks of fat and signal intensities similar to skeletal muscle [11]. Some elastofibromas have been described as having low signal intensity on T2 weighted images and this is believed to be secondary to a decrease in cellularity and increased collagen formation. Elastofibroma dorsi is a benign connective tissue tumor that is characteristically found at the angle of the scapula. Early reports of female and right-sided preponderance are contradicted by more recent case series. Although patients with elastofibroma are often asymptomatic, the most common symptom is a painless, enlarging mass. Oftentimes elastofibroma is found incidentally either during a radiographic evaluation (such as for lung cancer) or at the time of thoracotomy. Its resection is considered curative with only one documented recurrence.
References
Fig 2. Intraoperative photograph depicting left elastofibroma measuring 13.5 ⫻ 12.5 ⫻ 4.0 cm, which was excised through a musclesparing incision made along the anterior margin of the latissimus muscle.
1. Jarvi OH, Saxen AE. Elastofibroma dorsi. Acta Pathol Microbiol Scand 1961;144(Suppl 51):83– 4. 2. Solivetti FM, Bacaro D, Sidozzi AD, Cecconi P. Elastofibroma dorsi: ultrasound pattern in three patients. J Exp Clin Cancer Res 2003;22:565–9. 3. Dalal A, Miller TT, Kenan S. Sonographic detection of elastofibroma dorsi. J Clin Ultrasound 2003;31:375– 8. 4. Abe S, Miyata, N, Yamamoto Y, et al. Elastofibroma dorsi: CT, MRI, and pathologic findings. Plast Reconstr Surg 1999; 104:2121– 6. 5. Patrikeos A, Breidahl W, Robins P. F-18 FDG uptake associated with elastofibroma dorsi. Clin Nucl Med 2005;30:617– 8.
6. Pierce J, Henderson R. Hypermetabolism of elastofibroma dorsi on PET-CT. AJR 2004;183:35–7. 7. Nagamine N, Nohara Y, Ito E. Elastofibroma in Okinawa: a clinicopathologic study of 170 cases. Cancer 1982;50:1794 – 805. 8. Nielsen T, Sneppen O, Myhre-Jensen O, Daugaard S, Norback J. Subscapular elastofibroma: a reactive pseudotumor. J Shoulder Elbow Surg 1996;5:209 –13. 9. Briccoli A, Casadei R, Di Renzo M, Favale L, Bacchini P, Bertoni F. Elastofibroma dorsi. Surg Today 2000;30:147–52. 10. Marin ML, Perzin KH, Markowitz AM. Elastofibroma dorsi: benign chest wall tumor. J Thorac Cardiovasc Surg 1989;98: 234 – 8. 11. Kransdorf MJ, Meis JM, Montgomery E. Elastofibroma: MR and CT appearance with radiologic-pathologic correlation. Am J Roentgenol 1992;159:575–9. 12. Brandser EA, Goree JC, El-Khoury GY. Elastofibroma dorsi: prevalence in an elderly patient population as revealed by CT. AJR 1998;171:977– 80.
Tracheal Stenting of Iatrogenic Tracheal Injury: A Novel Management Approach Adam C. Yopp, MD, Jeremy G. Eckstein, MD, Richard H. Savel, MD, and Sunil Abrol, MD Department of Surgery, Division of Cardiothoracic Surgery, Maimonides Medical Center, Brooklyn, New York
We report the case of a patient who had an intubationrelated tracheal injury who we treated by deployment of a covered tracheal stent. This approach may be preferable to other alternatives in patients with a prohibitive risk of mortality with surgical repair or in an injury with sequelae not suitable for conservative management. (Ann Thorac Surg 2007;83:1897–9) © 2007 by The Society of Thoracic Surgeons
I
atrogenic injury to the tracheobronchial tree after intubation is an extremely rare occurrence that historically has been managed either expectantly or with a highly morbid surgical reconstruction. We report of the successful use of a tracheal stent to repair an intubationassociated tracheal injury.
CASE REPORT YOPP ET AL TRACHEAL STENTING OF IATROGENIC INJURY
1897
the patient was extubated without incident in the operating room. Approximately 2 hours after the conclusion of the procedure, progressively worsening respiratory distress developed. Physical examination revealed minimal subcutaneous emphysema, and a chest roentgenogram revealed no abnormalities. Computed chest tomography revealed a defect of the posterior trachea, approximately 1.5 cm proximal to the carina, as well as widespread pneumomediastinum (Fig 1). Bronchoscopic examination of the trachea confirmed a 4.0 cm tear of the posterior membranous trachea, 1.5 cm proximal to the carina (Fig 2). Given the worsening subcutaneous emphysema and increasing respiratory compromise requiring mechanical ventilation, the patient was immediately taken to the operating room. Because of the patient’s severe underlying pulmonary comorbidity, a decision was made to use a tracheal stent to repair the injury rather than a more invasive surgical reconstruction. A 6-cm ⫻ 14-mm selfexpanding, covered metallic stent (Microinvasive, Natick, MA) was deployed over the tracheal injury and 0.5 cm proximal to the carina under bronchoscopic and fluoroscopic guidance. The patient was started on broad-spectrum antibiotics and was successfully extubated 2 days later. After an unremarkable further hospital course, the patient was discharged home on postoperative day 5. The patient underwent monthly flexible bronchoscopic surveillance, which documented only minimal formation of granulation tissue. At 4 months, excess respiratory secretions developed, and flexible bronchoscopy revealed significant granulation tissue at the proximal end of the covered stent. The tracheal stent was removed using rigid forceps through rigid bronchoscopy, with only slight difficulty. The underlying trachea was well healed (Fig 3). During the procedure, ventilation and oxygenation were ensured by high-frequency jet ventilation. The patient was discharged home the next day and remains asymptomatic 1 year later.
A 75-year-old woman with a history of chronic obstructive pulmonary disease, idiopathic pulmonary hypertension, and severe cervical-thoracic spine scoliosis underwent repair of a humeral head fracture. The procedure was initially done with a scalene nerve block, but owing to ipsilateral phrenic nerve paralysis, the patient went into respiratory distress and required intubation. The intubation was difficult because of aberrant cervical anatomy in the setting of her spinal disease. A fiberoptic bronchoscopic intubation was performed after multiple unsuccessful attempts at intubation via direct laryngoscopy. The intraoperative course was unremarkable, and Accepted for publication Dec 13, 2006. Address correspondence to Dr Yopp, Maimonides Medical Center, 4802 Tenth Ave, Brooklyn, NY 11219; e-mail: [email protected].
© 2007 by The Society of Thoracic Surgeons Published by Elsevier Inc
Fig 1. Computed chest tomography reveals a defect in the posterior trachea, 1.5 cm proximal to the carina (white arrow). 0003-4975/07/$32.00 doi:10.1016/j.athoracsur.2006.12.040
FEATURE ARTICLES
Ann Thorac Surg 2007;83:1897–9