Ann Thorac Surg 2006;82:1898-1900
© 2006 The Society of Thoracic Surgeons
Case Reports
Management Dilemmas Due to a Paratracheal Follicular Dendritic Cell Tumor
Louis P. Voigt, MD, FCCPa,*,
Ali Hmidi, MDa,
Stephen M. Pastores, MD, FCCPa,
Robert G. Maki, MD, PhDb,
Diane L. Carlson, MDc,
Manjit S. Bains, MD, FACSd,
Neil A. Halpern, MD, FCCPa
a Department of Anesthesiology and Critical Care Medicine, Memorial Sloan Kettering Cancer Center, New York, New York
b Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, New York
c Department of Pathology, Memorial Sloan Kettering Cancer Center, New York, New York
d Department of Surgery, Memorial Sloan Kettering Cancer Center, New York, New York
Accepted for publication March 13, 2006.
* Address correspondence to Dr Voigt, Department of Anesthesiology and Critical Care Medicine, Memorial Sloan-Kettering Cancer Center, 1275 York Avenue M-210, New York, NY 10021. (Email: voigtl{at}mskcc.org).
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Abstract
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Follicular dendritic cell tumors are rare cancers of the lymph nodes of the head and neck. Despite a relatively high rate of recurrence, these neoplasms have a limited impact on the airways because they can be potentially cured by surgical resection. We present the case of a young woman with an unresectable follicular dendritic cell tumor involving the paratracheal region causing upper airway obstruction and respiratory failure. This malignant tumor created numerous management dilemmas and therapeutic challenges related to the unstable airway and the need for tracheal stenting to bypass the airway obstruction.
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Introduction
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Follicular dendritic cell (FDC) tumors are rare neoplasms occurring predominantly in the lymph nodes and extranodal sites of the head and neck [1, 2]. Since the first description in 1986 [3], at least 68 cases of FDC tumors have been reported worldwide. We believe that our report is the first case of a paratracheal FDC tumor causing both airway obstruction and significant management dilemmas in the intensive care unit (ICU).
A previously healthy 29-year-old woman presented to an outside hospital with a month-long progression of neck swelling and pain followed by dyspnea and stridor. She was hospitalized and intubated. A chest roentgenogram revealed tracheal deviation and narrowing. Computed tomography of the neck confirmed a large subglottic paratracheal mass (5.1 x 6.0 x 8.0 cm) that invaded the esophagus and the right posterolateral wall of the trachea. The mass was also associated with intraluminal tracheal narrowing and encasement of the right common carotid artery. The biopsy suggested possible FDC tumor. She was transferred to the ICU at our institution.
Two days later, she was taken to the operating room for a planned surgical resection of the tumor. The procedure was aborted when the rigid bronchoscope could not be advanced beyond the subglottic area because the tumor protruded into the trachea, extending from 1 cm below the vocal cords to 2 cm above the carina. A repeat biopsy confirmed the diagnosis of FDC tumor through histology and immunohistochemical studies. A 7-cm anode endotracheal tube was inserted and she returned on mechanical ventilation to the ICU. One day postoperatively, bilateral pneumothoraces prompted insertion of chest tubes.
On ICU day 5, the patient returned to the operating room for airway stabilization. Under fluoroscopic guidance and flexible bronchoscopy, three 20 x 14-mm, expandable, covered Ultraflex tracheobronchial metallic stents (Boston Scientific, Natick, MA) were placed in tandem from the subglottic region to 2 cm above the carina. Postoperatively she was managed with jet ventilation for 3 days using a 14-French catheter inserted into the proximal stent. However, the anticipated recovery of the respiratory failure from upper airway obstruction did not occur, and she was orally intubated on ICU day 8 with a 7.5-cm endotracheal tube inserted through the stents.
For approximately 3 weeks she received combined chemoradiation therapy. She was transported to the radiation oncology suite on nearly a daily basis and received 5,940 cGy of radiation therapy. Concomitantly she was treated with five weekly doses of gemcitabine at 400 mg/m2 as a radiation sensitizer. On ICU day 15, doxycycline pleurodesis was performed on the right side and the chest tube was removed. The left chest tube was retained for longer term drainage.
On ICU day 20, a computed tomographic scan (see Fig 1) showed significant tumor reduction. Two days later, a No. 6 Shiley tracheostomy tube (Nellcor Tyco Healthcare, Pleasanton, CA) was inserted. Subsequently, chest roentgenograms showed intermittent distal migration of the "in-tandem" stents into the opening of the right main stem bronchus. This situation resulted in left-sided atelectasis and postobstructive bacterial pneumonia (Fig 2). Approximately 20 bronchoscopies were performed for stent repositioning and pulmonary toilet. Gradually her condition improved and she was liberated from mechanical ventilation. On ICU day 42, the stents were extracted uneventfully in the operating room. Three days later she was discharged to the ward for physical therapy and further chemoradiation. Thereafter the left chest tube was removed and she was decannulated. A computed tomographic scan of the neck revealed near-complete resolution of the FDC tumor. After approximately 2 months in the hospital, she was discharged for ongoing rehabilitation. The Institutional Review Board at Memorial Sloan-Kettering Cancer Center deemed that a waiver of authorization for this case report was not necessary. An individual consent was not obtained as no specific patient identifiers were used.
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Fig 1. Computed tomographic scan of (A) the cervical mass showing (B) the endotracheal tube passing through (C) the tracheal stents.
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Fig 2. Chest roentgenogram showing (A) the tracheostomy tube with migration of (B) the tracheal stents into the right main bronchus with resultant left-sided atelectasis.
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Comment
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Follicular dendritic cell tumors usually occur in young adults and are associated with a mortality rate of 16% [1]. These neoplasms are indolent, of intermediate malignant characteristics, and indeterminate clinical behavior. Recurrence is frequent and metastases may occur to the lungs, liver, and peritoneum [1, 2]. Follicular dendritic cell tumors can be misdiagnosed as lymphoma, thymoma, ectopic meningioma, sarcoma, and metastatic carcinoma [4]. Associations with Castleman's disease and Epstein Barr virus have been reported [1]. Poor prognostic factors include a tumor size > 6 cm, nuclear pleomorphism, necrosis, high mitotic rate, and intraabdominal location [2]. Complete surgical resection is the treatment of choice [1]. However, if resection is not an option, radiation and chemotherapy may be recommended.
Throughout our patient's ICU stay, the issue of the unstable airway remained a constant concern. Early on, the tumor infiltrated the entire trachea leaving little room to place an artifical airway. The Ultraflex expandable metallic stents (Boston Scientific) placed in the trachea provided a temporary airway solution while the patient was receiving chemoradiation. Stents have been shown to adequately restore airway patency when surgical repair of the airway obstruction is contraindicated [5]. Later, to facilitate long-term ventilation and intrahospital transport, a tracheostomy tube was inserted when the tumor diminished in size. Subsequently, intermittent migration of the stents into the right mainstem bronchus was associated with left-sided airway obstruction, atelectasis, and pneumonia. We postulated that the stents slid distally because of the constant pressure on the stents by the tracheostomy tube in the setting of improved airway patency in response to chemoradiation therapy. The stents were ultimately extracted because they were causing more harm than benefit. A recent observational study has documented safe endoscopic removal of metallic stents in cases of malignant airway obstruction [6].
Intra-hospital transports of mechanically ventilated patients tax the ICU staff and may be associated with many complications [7]. In our patient, more than 40 uneventful intra-hospital transports were arranged to the operating room, to the imaging center, and to the radiation oncology suite. These transports forced the ICU to continually seek the most stable airway possible in a dynamically changing situation. In addition, these transports necessitated extraordinary coordination and cooperation among the ICU, thoracic surgery, respiratory therapy, and radiation oncology teams.
In conclusion, this case illustrates the unique challenges faced by clinicians caring for the critically ill oncologic patient undergoing multimodality cancer therapy in the ICU. It also demonstrates that despite the presence of life-threatening airway obstruction and management dilemmas in the ICU, the application of multidisciplinary support resulted in a positive outcome in this patient with an unresectable FDC tumor.
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References
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- Chan JK, Fletcher CD, Nayler SJ, Cooper K. Follicular dendritic cell sarcoma: clinicopathologic analysis of 17 cases suggesting a malignant potential higher than currently recognized Cancer 1997;79:294-313.[Medline]
- Perez-Ordonez B, Erlandson RA, Rosai J. Follicular dendritic cell tumor: report of 13 additional cases of a distinctive entity Am J Surg Pathol 1996;20:944-955.[Medline]
- Monda L, Warnke R, Rosai J. A primary lymph node malignancy with features suggestive of dendritic reticulum cell differentiation: a report of 4 cases Am J Pathol 1986;122:562-572.[Abstract]
- Pileri SA, Grogan TM, Harris NL, et al. Tumours of histiocytes and accessory dendritic cells: an immunohistochemical approach to classification from the International Lymphoma Study Group based on 61 cases Histopathology 2002;41:1-29.[Medline]
- Madden BP, Datta S, Charokopos N. Experience with Ultraflex expandable metallic stents in the management of endobronchial pathology Ann Thorac Surg 2002;73:938-944.[Abstract/Free Full Text]
- Lunn W, Feller-Kopman D, Wahidi M, Ashiku S, Thurer R, Ernst A. Endoscopic removal of metallic airways stents Chest 2005;127:2106-2112.[Abstract/Free Full Text]
- Braman SS, Dunn SM, Amico CA, Millman RP. Complications of intrahospital transport in critically ill patients Ann Intern Med 1987;107:469-473.[Medline]
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Abstract
Introduction
