The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
deeper within the tumor. A blending of squamous cells and spindle cells can be often seen and differentiated by their different arrangement, which includes storiform, solid, and fascicular appearances. Furthermore, immunohistochemical studies of epithelial and mesenchymal markers are important to diagnose the tumor. Epithelial markers include keratin (AE1/AE3), epithelial membrane antigens, KI, and K18, whereas mesenchymal markers include vimentin, desmin, S-100, osteopontin, and bone morphogenetic protein.1 In our study population, the epithelial markers EMA and AE1/ AE3 were positive in all 5 cases. Percentage and intensity of staining in tumor cells, which were positive for epithelial markers, varied widely; in some cases, the cells showed diffuse and intense positivity, and in some cases, the staining was weak and focal. Vimentin was positive in all cases. Occasional aberrant expression of other mesenchymal markers gave rise to diagnostic confusion. S100 was positive in a case. None of the cases were positive for HMB45. The most controversial aspect of carcinosarcomas is the pathogenesis, the determination of which influences the prognosis and treatment. Because of its rarity, it is difficult to completely bring to a conclusion the controversy of their origin or derivation, but the epithelial nature of the tumor seems to be strongly supported by the histologic and immunophenotypic expression.8 This conclusion therefore influences the choice of management. Several histogenetic theories have been developed over the years, with a few principal theories, such as the “carcinosarcoma or collision tumor theory” (a separate epithelial and mesenchymal cell become each malignant), the “spindle cell carcinoma theory” (an epithelial cell differentiates into both squamous and spindle cell components), the “carcinoma with pseudosarcoma theory” (a carcinoma stimulates a benign reactive stromal response), and the “carcinosarcoma theory” (a malignant epithelial cell “dedifferentiates” into a sarcoma).8 Although it may be difficult to prove such theories, the occurrence of carcinosarcomas in the sites that normally have squamous epithelium, a preponderance of carcinomas rather than sarcomas, a superficial location, the immunoreactivity with epithelial antigens, and the direct continuity and smooth transition of the spindled cells with areas of squamous epithelium may constitute a compelling evidence in favor of an epithelial cell that differentiates into both a carcinoma and a spindle cell component.8 Surgery is the treatment of choice for spindle cell carcinoma of the larynx. Exclusive radiotherapy has also been used successfully, although only with limited lesions. In fact, histologic diagnosis of spindle cell carcinoma should not influence the decision to treat a patient with early-stage glottis disease with radiotherapy.3
CONCLUSIONS Carcinosarcoma has been considered a biphasic tumor composed of a squamous cell carcinoma and a malignant spindle cell component with a mesenchymal appearance, but of epithelial origin. However, this tumor can now be regarded as a monoclonal epithelial neoplasm, with a divergent (mesenchymal) differentiation, rather than a collision tumor or biphasic derivation. Histologically, a blending of squamous cells and spindle cells is often observed, and they may present different arrangements with storiform, solid, and fascicular appearances. Immunohistochemical studies of epithelial and mesenchymal markers (such as AE1/AE3, epithelial membrane antigens, vimentin, desmin, and S-100) are fundamental to diagnose the tumor, although they may vary widely.
REFERENCES 1. Boamah H, Ballard B. A case report of spindle cell (sarcomatoid) carcinoma of the larynx. Case Rep Med 2012;2012:370204
Brief Clinical Studies
2. Lewis JE, Olsen KD, Sebo TJ. Spindle cell carcinoma of the larynx: review of 26 cases including DNA content and immunohistochemistry. Hum Pathol 1997;28:664–673 3. Marioni G, Bottin R, Staffieri A, et al. Spindle-cell tumours of the larynx: diagnostic pitfalls. A case report and review of the literature. Acta Otolaryngol 2003;123:86–90 4. Samdhani S, Choudhary A, Mahanta VR, et al. Spindle cell sarcoma of larynx. Indian J Otolaryngol Head Neck Surg 2006;58:305–306 5. Singh P, Kanotra JP, Luthra D, et al. Sarcomatoid carcinoma of larynx. Indian J Otolaryngol Head Neck Surg 2000;52: 189–190 6. Stomeo F, Rocca PC, Bozzo C, et al. Laryngeal true malignant mixed tumor. Head Neck 2009;31:556–560 7. Viswanathan S, Rahman K, Pallavi S, et al. Sarcomatoid (spindle cell) carcinoma of the head and neck mucosal region: a clinicopathologic review of 103 cases from a tertiary referral cancer centre. Head Neck Pathol 2010;4:265–275 8. Thompson LD, Wieneke JA, Miettinen M, et al. Spindle cell (sarcomatoid) carcinomas of the larynx: a clinicopathologic study of 187 cases. Am J Surg Pathol 2002;26:153–170 9. Tulunay O, Küçük B, Yorulmaz I, et al. Sarcomatoid carcinoma of the larynx: immunohistochemical analysis in two cases. Otolaryngol Head Neck Surg 2006;134:1057–1059
Quick and Accurate Measures in Negative Pressure Pulmonary Edema: A Guideline for Orthognathic Surgeons Sung Ok Hong, DMD,* Jun-Young Chung, MD, PhD,† Deok-Won Lee, DMD, MSD* Abstract: Perioperative pulmonary edema is a rare complication of maxillofacial surgery. However, this potentially fatal complication may arise during any maxillofacial surgery. Negative pressure pulmonary edema can be caused by upper airway obstruction after operation. When this phenomenon arises, if not treated properly, it progresses rapidly causing fatal outcomes. Because orthognathic surgery is performed mostly on healthy and young patients, surgeons and anesthesiologists might neglect the possibility of such complications. Therefore, we must always take into consideration the possibility of negative pressure pulmonary edema. Careful observation of the patient; accurate knowledge; and active, quick, and noninvasive safe measures are required to treat this malady when it does occur. We report a case of negative pressure pulmonary edema after orthognathic surgery and its successful treatment. From the Departments of *Oral Maxillofacial Surgery and †Anesthesiology and Pain Medicine, School of Medicine, Kyung Hee University Dental Hospital at Gangdong, Kyung Hee University, Seoul, Republic of Korea. Received January 27, 2014. Accepted for publication February 17, 2014. Address correspondence and reprint requests to Deok-Won Lee, DMD, MSD, Department of Oral and Maxillofacial Surgery, Kyung Hee University Dental Hospital at Gangdong, Kyung Hee University, 149, Gandong Gu, Sang-il Dong, Seoul 134-727, Korea; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000944
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e433
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
Brief Clinical Studies
Key Words: Continuous positive airway pressure, negative pressure pulmonary edema, orthognathic surgery, upper airway obstruction
P
ostoperative negative pressure pulmonary edema (NPPE) can arise by several events. The most common cause is acute upper airway obstruction associated with laryngospasm after extubation.1 In maxillofacial surgery, the risk of upper airway obstruction increases because of reasons such as postoperative bleeding, swelling, or anatomic changes of airway after operation. This phenomenon arises and progresses rapidly if not treated and can cause fatal outcomes such as severe respiratory distress, hypoxia002C and even hypoxic brain damage. So, adequate diagnosis, treatment, and differential diagnosis from other causes of pulmonary edema should be made quickly for improvement of patient outcomes. We present here a case of postoperative NPPE after orthognathic surgery to help guide surgeons to take active, quick, and adequate measures, therefore preventing such fatal complications.
MATERIALS AND METHODS A 26-year-old male patient was hospitalized for orthognathic surgery, having no special medical history or abnormality on the laboratory findings and chest x-ray (Fig. 1A). A routine nasotracheal intubation was performed using a nasal right angle endotracheal tube and maintained with sevoflurane and remifentanil. Le Fort I osteotomy and both sagittal split ramal osteotomy were performed via the common method, and the total time for anesthesia was 270 minutes. After surgery, pyridostigmine and glycopyrrolate were administered as antagonists for the muscle relaxation, and extubation was performed. However, the patient began to show irregular, shallow breathing immediately after extubation, with symptoms of breathing difficulty and upper airway obstruction. The peripheral oxygen saturation (SpO2) was reduced to 87% after 3 minutes. Despite approximately 2 minutes of additional assisted ventilation with a facial mask, the spontaneous respiration was insufficient, and the symptoms became increasingly severe. The anesthesiologist injected 60 mg of lidocaine while performing positive pressure ventilation. The patient began to breathe with relative stability, and SpO2 returned back to 94% with assisted ventilation with 100% of oxygen. After recovery from upper airway obstruction, the patient had a clear consciousness, but he complained of discomfort in the chest while breathing. He was continuously excreting large amounts of pink frothy sputum; therefore, a chest x-ray was initiated (Fig. 1B). The patient was transferred
FIGURE 2. A, Chest posterior-anterior view of 12 hours after occurrence of pulmonary edema shows slight improvement with reduced haziness of hilum (A) and reduced pulmonary vasculature on both lungs (B). Most of the pink frothy sputum was removed after 12 hours because of deep and large breaths and continued suctioning while a noninvasive CPAP was maintained. The supplemented oxygen was stopped after 6 hours for 30 minutes of observation. B, Chest posterior-anterior view of 3 months after operation shows totally improved status of pulmonary edema. There are no remarkable findings. The patient received 5 days of postoperative management and was discharged without other problems.
to the intensive care unit under suspicion of pulmonary edema. Pulmonary edema was observed on the chest x-ray. In the intensive care unit, the patient was placed in the semi-Fowler position, and the pink frothy sputum was removed with assisted oral suction. There was no sign of hypoxia; therefore, 6 L/min of oxygen was applied using a noninvasive continuous positive airway pressure (CPAP) mask. Under the diagnosis of pulmonary edema, the amount of injected fluid was limited and given 10 mg of diuretics. Continued oral suction and spitting of secretions were performed to prevent disturbance of the gas exchange. Morphine was also administered and followed by furosemide afterward to reduce the patient's uneasiness and induce reduction in the overall load through extension of the systemic veins.
RESULTS Deep and large breaths were encouraged with continued suction of the pink frothy sputum to help smooth respiration. Most of the sputum was removed after 12 hours, and the pulmonary edema was significantly improved based on the chest x-ray (Fig. 2A). The CPAP mask was removed, and oxygen was supplied with a venturi mask. The patient was moved to a general ward about 24 hours after operation, and the fluid balance was continuously controlled. The SpO2 was maintained at 97% to 99%, and the chest x-ray became normal on the following day. The patient received 5 days of postoperative management and was discharged without other problems. A chest x-ray taken at 3 months after the operation confirmed total improvement in the status of pulmonary edema (Fig. 2B).
DISCUSSION
FIGURE 1. A, Chest posterior-anterior view before surgery shows no remarkable findings in the 26-year-old male patient scheduled for orthognathic surgery. The patient had no special medical history and abnormality on the laboratory results nor in the upper airway. B, Chest posterior-anterior view 30 minutes postoperatively. Chest x-ray checked in the postanesthetic care unit reveals full hazy hilum (A), prominence of the pulmonary vasculature and haziness on both lungs distribution (B), and increased heart size (C), thus implicating pulmonary edema. Clinical symptom was irregular, shallow breathing immediately after extubation. The oxygen saturation was reduced, the spontaneous respiration was insufficient, and the symptoms became increasingly severe. The patient complained of discomfort in the chest while breathing. However, he had a clear consciousness, and he was continuously excreting large amounts of pink frothy sputum.
e434
The NPPE can occur from acute or continued obstruction of the upper airway and mainly occurs during emergence from general anesthesia. Although there are differences, it has been estimated that NPPE develops in 11% of patients2 and requires active intervention for acute upper airway obstruction. The morbidity and mortality associated with NPPE is 11% to 40%, and it is primarily associated with delayed recognition and treatment. Halow and Ford3 reported that the ability to generate large intrapulmonary pressures with a very compliant chest wall may play a significant role in the pathophysiology of NPPE. Young, healthy patients are capable of generating large negative intrathoracic pressures by virtue of their increased level of fitness, which increases the risk of NPPE.4 Another risk factors are anatomically difficult intubation, nasal, oral, or pharyngeal surgical site of pathology; obesity with obstructive apnea; short neck; and acromegaly.5 Especially in case of orthognatic surgery, there are increased risks of airway obstruction and NPPE, because mandibular retropositioning can cause airway narrowing. © 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
In general, there are many pathophysiologic processes that can lead to NPPE. Excessive inspiratory force is the main cause and was reported to reach as high as −100 mm Hg during upper airway obstruction. This excessive negative pressure is thought to cause disruption of the alveolar capillaries to cause pulmonary edema and, sometimes, alveolar hemorrhage.6 Moreover, this event leads to an increase in venous return and blood flow to the right heart. However, hyperadrenergic state induced by hypoxia can increase systemic vascular resistance, pulmonary vascular resistance, and pulmonary capillary pressure and subsequently increase afterload.1, 5 This combination causes an increase in pulmonary blood volume and pulmonary venous pressure and then an increase in hydrostatic pressure and edema formation.5, 7 Most of NPPEs arise immediately after relief of an airway obstruction; however, a delayed onset of up to 24 hours has been reported.4 The clinical signs of NPPE are typical. After recovery from airway obstruction, the patient presents tachypnea, dyspnea, and decreased SpO2 levels despite of supplying oxygen. In addition, tachycardia and hypertension also can arise because of increased sympathetic activity. We can hear rales and rhonchi on chest auscultation,8 and chest radiograph shows rapid bilateral changes consistent with pulmonary edema.9 All the treatments for NPPE are supportive treatments and are able to be executed by any surgeon. 1. 2. 3. 4. 5.
Maintenance of the airway Supplying oxygen Hemodynamic stability Positive pressure ventilation Diuretics and appropriate fluid therapy
Although active treatment using intubation and positive pressure is required for severe cases of hypoxia, the use of CPAP for the treatment of acute pulmonary edema and respiratory failure is generally broadly accepted. There have been reports of successful treatment using noninvasive CPAP via facial masks or a nasal cannula.10 In this case, after recovering from airway obstruction with soft positive pressure ventilation and lidocaine administration, the patient had a clear consciousness and was able to excrete the sputum. The possibility of pulmonary aspiration was considered to be low, so CPAP could be applied through a mask while maintaining spontaneous respiration with assisted oral suctioning of the pink frothy sputum. As a result of the parallel allopathic treatments, the sputum in the lungs was removed, and the respiration sounds became clean on the ausculation of the thorax. After such treatment, the patient completely recovered without other problems. In conclusion, the patients receiving orthognathic surgery are easily exposed to the risk of NPPE because of upper airway obstruction. This event can cause fatal outcomes if quick and accurate measures are not taken. So, careful observation of the patient and accurate knowledge are required to treat this malady when it does occur. Active, quick, and adequate measures must be taken to treat the complications of NPPE.
REFERENCES 1. Lang SA, Duncan PG, Shephard DA, et al. Pulmonary oedema associated with airway obstruction. Can J Anaesth 1990;37:210–218 2. Krodel DJ, Bittner EA, Abdulnour R, et al. Case scenario: acute postoperative negative pressure pulmonary edema Anesthesiology 2010;113:200–207 3. Halow KD, Ford EG. Pulmonary edema following post-operative laryngospasm: a case report and review of the literature. Am Surg 1993;59:443–447
Brief Clinical Studies
4. McConkey PP. Postobstructive pulmonary oedema—a case series and review. Anaesth Intensive Care 2000;28:72–76 5. Udeshi A, Cantie SM, Pierre E. Postobstructive pulmonary edema. J Crit Care 2010;25:538 e1–5 6. Hirano Y, Sugawara T, Sato Y, et al. Negative pressure pulmonary edema following foramen magnum decompression for Chiari malformation type I. Neurol Med Chir (Tokyo) 2008;48:137–139 7. Guffin TN, Har-el G, Sanders A, et al. Acute postobstructive pulmonary edema. Otolaryngol Head Neck Surg 1995;112:235–237 8. Van Kooy MA, Gargiulo RF. Postobstructive pulmonary edema. Am Fam Physician 2000;62:401–404 9. Cascade PN, Alexander GD, Mackie DS. Negative-pressure pulmonary edema after endotracheal intubation. Radiology 1993;186:671–375 10. Volpicelli G, Fogliati C, Radeschi G, et al. A case of unilateral re-expansion pulmonary oedema successfully treated with non-invasive continuous positive airway pressure. Eur J Emerg Med 2004;11:291–294
Inferior Alveolar Nerve Repositioning and Orthognathic Surgery Amin Rahpeyma,* Saeedeh Khajehahmadi, DDS† Background: Inferior alveolar nerve (IAN) repositioning is a wellknown technique in implant dentistry. This invaluable technique can be combined with orthognathic techniques to enhance the effect of orthognathic surgery in some selected cases. Materials and Methods: In a retrospective study, data were obtained from the archived files of Department of Oral and Maxillofacial Surgery, Mashhad University of Medical Sciences, Iran, from 2007 to 2012. Patients undergoing mandibular orthognathic surgery alone or in combination with maxillary surgery were searched. Cases with IAN repositioning were selected. Results: In this study, in 4% of orthognathic surgical procedures involving the mandibular bone, IAN repositioning was indicated. Genioplasty, body ostectomy, total mandibular subapical alveolar osteotomy, and inferior border osteotomy for correction of asymmetric mandibular excess in hemimandibular hyperplasia were in this list. Conclusion: In carefully selected orthognathic patients, IAN repositioning can enhance the effect of orthognathic surgery and should be considered in the treatment plan strategies as an option. From the *Oral and Maxillofacial Diseases Research Center and †Dental Research Center, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran. Received January 28, 2014. Accepted for publication February 17, 2014. Address correspondence and reprint requests to Saeedeh Khajehahmadi, DDS, Dental Research Center, Faculty of Dentistry, Mashhad University of Medical Sciences, Vakilabad Blvd, Mashhad, P.O. Box: 91735-984, Iran; E-mail: [email protected] or [email protected] Supported by a grant from the Vice Chancellor of Research of Mashhad University of Medical Sciences. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000945
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e435
deeper within the tumor. A blending of squamous cells and spindle cells can be often seen and differentiated by their different arrangement, which includes storiform, solid, and fascicular appearances. Furthermore, immunohistochemical studies of epithelial and mesenchymal markers are important to diagnose the tumor. Epithelial markers include keratin (AE1/AE3), epithelial membrane antigens, KI, and K18, whereas mesenchymal markers include vimentin, desmin, S-100, osteopontin, and bone morphogenetic protein.1 In our study population, the epithelial markers EMA and AE1/ AE3 were positive in all 5 cases. Percentage and intensity of staining in tumor cells, which were positive for epithelial markers, varied widely; in some cases, the cells showed diffuse and intense positivity, and in some cases, the staining was weak and focal. Vimentin was positive in all cases. Occasional aberrant expression of other mesenchymal markers gave rise to diagnostic confusion. S100 was positive in a case. None of the cases were positive for HMB45. The most controversial aspect of carcinosarcomas is the pathogenesis, the determination of which influences the prognosis and treatment. Because of its rarity, it is difficult to completely bring to a conclusion the controversy of their origin or derivation, but the epithelial nature of the tumor seems to be strongly supported by the histologic and immunophenotypic expression.8 This conclusion therefore influences the choice of management. Several histogenetic theories have been developed over the years, with a few principal theories, such as the “carcinosarcoma or collision tumor theory” (a separate epithelial and mesenchymal cell become each malignant), the “spindle cell carcinoma theory” (an epithelial cell differentiates into both squamous and spindle cell components), the “carcinoma with pseudosarcoma theory” (a carcinoma stimulates a benign reactive stromal response), and the “carcinosarcoma theory” (a malignant epithelial cell “dedifferentiates” into a sarcoma).8 Although it may be difficult to prove such theories, the occurrence of carcinosarcomas in the sites that normally have squamous epithelium, a preponderance of carcinomas rather than sarcomas, a superficial location, the immunoreactivity with epithelial antigens, and the direct continuity and smooth transition of the spindled cells with areas of squamous epithelium may constitute a compelling evidence in favor of an epithelial cell that differentiates into both a carcinoma and a spindle cell component.8 Surgery is the treatment of choice for spindle cell carcinoma of the larynx. Exclusive radiotherapy has also been used successfully, although only with limited lesions. In fact, histologic diagnosis of spindle cell carcinoma should not influence the decision to treat a patient with early-stage glottis disease with radiotherapy.3
CONCLUSIONS Carcinosarcoma has been considered a biphasic tumor composed of a squamous cell carcinoma and a malignant spindle cell component with a mesenchymal appearance, but of epithelial origin. However, this tumor can now be regarded as a monoclonal epithelial neoplasm, with a divergent (mesenchymal) differentiation, rather than a collision tumor or biphasic derivation. Histologically, a blending of squamous cells and spindle cells is often observed, and they may present different arrangements with storiform, solid, and fascicular appearances. Immunohistochemical studies of epithelial and mesenchymal markers (such as AE1/AE3, epithelial membrane antigens, vimentin, desmin, and S-100) are fundamental to diagnose the tumor, although they may vary widely.
REFERENCES 1. Boamah H, Ballard B. A case report of spindle cell (sarcomatoid) carcinoma of the larynx. Case Rep Med 2012;2012:370204
Brief Clinical Studies
2. Lewis JE, Olsen KD, Sebo TJ. Spindle cell carcinoma of the larynx: review of 26 cases including DNA content and immunohistochemistry. Hum Pathol 1997;28:664–673 3. Marioni G, Bottin R, Staffieri A, et al. Spindle-cell tumours of the larynx: diagnostic pitfalls. A case report and review of the literature. Acta Otolaryngol 2003;123:86–90 4. Samdhani S, Choudhary A, Mahanta VR, et al. Spindle cell sarcoma of larynx. Indian J Otolaryngol Head Neck Surg 2006;58:305–306 5. Singh P, Kanotra JP, Luthra D, et al. Sarcomatoid carcinoma of larynx. Indian J Otolaryngol Head Neck Surg 2000;52: 189–190 6. Stomeo F, Rocca PC, Bozzo C, et al. Laryngeal true malignant mixed tumor. Head Neck 2009;31:556–560 7. Viswanathan S, Rahman K, Pallavi S, et al. Sarcomatoid (spindle cell) carcinoma of the head and neck mucosal region: a clinicopathologic review of 103 cases from a tertiary referral cancer centre. Head Neck Pathol 2010;4:265–275 8. Thompson LD, Wieneke JA, Miettinen M, et al. Spindle cell (sarcomatoid) carcinomas of the larynx: a clinicopathologic study of 187 cases. Am J Surg Pathol 2002;26:153–170 9. Tulunay O, Küçük B, Yorulmaz I, et al. Sarcomatoid carcinoma of the larynx: immunohistochemical analysis in two cases. Otolaryngol Head Neck Surg 2006;134:1057–1059
Quick and Accurate Measures in Negative Pressure Pulmonary Edema: A Guideline for Orthognathic Surgeons Sung Ok Hong, DMD,* Jun-Young Chung, MD, PhD,† Deok-Won Lee, DMD, MSD* Abstract: Perioperative pulmonary edema is a rare complication of maxillofacial surgery. However, this potentially fatal complication may arise during any maxillofacial surgery. Negative pressure pulmonary edema can be caused by upper airway obstruction after operation. When this phenomenon arises, if not treated properly, it progresses rapidly causing fatal outcomes. Because orthognathic surgery is performed mostly on healthy and young patients, surgeons and anesthesiologists might neglect the possibility of such complications. Therefore, we must always take into consideration the possibility of negative pressure pulmonary edema. Careful observation of the patient; accurate knowledge; and active, quick, and noninvasive safe measures are required to treat this malady when it does occur. We report a case of negative pressure pulmonary edema after orthognathic surgery and its successful treatment. From the Departments of *Oral Maxillofacial Surgery and †Anesthesiology and Pain Medicine, School of Medicine, Kyung Hee University Dental Hospital at Gangdong, Kyung Hee University, Seoul, Republic of Korea. Received January 27, 2014. Accepted for publication February 17, 2014. Address correspondence and reprint requests to Deok-Won Lee, DMD, MSD, Department of Oral and Maxillofacial Surgery, Kyung Hee University Dental Hospital at Gangdong, Kyung Hee University, 149, Gandong Gu, Sang-il Dong, Seoul 134-727, Korea; E-mail: [email protected] The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000944
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e433
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
Brief Clinical Studies
Key Words: Continuous positive airway pressure, negative pressure pulmonary edema, orthognathic surgery, upper airway obstruction
P
ostoperative negative pressure pulmonary edema (NPPE) can arise by several events. The most common cause is acute upper airway obstruction associated with laryngospasm after extubation.1 In maxillofacial surgery, the risk of upper airway obstruction increases because of reasons such as postoperative bleeding, swelling, or anatomic changes of airway after operation. This phenomenon arises and progresses rapidly if not treated and can cause fatal outcomes such as severe respiratory distress, hypoxia002C and even hypoxic brain damage. So, adequate diagnosis, treatment, and differential diagnosis from other causes of pulmonary edema should be made quickly for improvement of patient outcomes. We present here a case of postoperative NPPE after orthognathic surgery to help guide surgeons to take active, quick, and adequate measures, therefore preventing such fatal complications.
MATERIALS AND METHODS A 26-year-old male patient was hospitalized for orthognathic surgery, having no special medical history or abnormality on the laboratory findings and chest x-ray (Fig. 1A). A routine nasotracheal intubation was performed using a nasal right angle endotracheal tube and maintained with sevoflurane and remifentanil. Le Fort I osteotomy and both sagittal split ramal osteotomy were performed via the common method, and the total time for anesthesia was 270 minutes. After surgery, pyridostigmine and glycopyrrolate were administered as antagonists for the muscle relaxation, and extubation was performed. However, the patient began to show irregular, shallow breathing immediately after extubation, with symptoms of breathing difficulty and upper airway obstruction. The peripheral oxygen saturation (SpO2) was reduced to 87% after 3 minutes. Despite approximately 2 minutes of additional assisted ventilation with a facial mask, the spontaneous respiration was insufficient, and the symptoms became increasingly severe. The anesthesiologist injected 60 mg of lidocaine while performing positive pressure ventilation. The patient began to breathe with relative stability, and SpO2 returned back to 94% with assisted ventilation with 100% of oxygen. After recovery from upper airway obstruction, the patient had a clear consciousness, but he complained of discomfort in the chest while breathing. He was continuously excreting large amounts of pink frothy sputum; therefore, a chest x-ray was initiated (Fig. 1B). The patient was transferred
FIGURE 2. A, Chest posterior-anterior view of 12 hours after occurrence of pulmonary edema shows slight improvement with reduced haziness of hilum (A) and reduced pulmonary vasculature on both lungs (B). Most of the pink frothy sputum was removed after 12 hours because of deep and large breaths and continued suctioning while a noninvasive CPAP was maintained. The supplemented oxygen was stopped after 6 hours for 30 minutes of observation. B, Chest posterior-anterior view of 3 months after operation shows totally improved status of pulmonary edema. There are no remarkable findings. The patient received 5 days of postoperative management and was discharged without other problems.
to the intensive care unit under suspicion of pulmonary edema. Pulmonary edema was observed on the chest x-ray. In the intensive care unit, the patient was placed in the semi-Fowler position, and the pink frothy sputum was removed with assisted oral suction. There was no sign of hypoxia; therefore, 6 L/min of oxygen was applied using a noninvasive continuous positive airway pressure (CPAP) mask. Under the diagnosis of pulmonary edema, the amount of injected fluid was limited and given 10 mg of diuretics. Continued oral suction and spitting of secretions were performed to prevent disturbance of the gas exchange. Morphine was also administered and followed by furosemide afterward to reduce the patient's uneasiness and induce reduction in the overall load through extension of the systemic veins.
RESULTS Deep and large breaths were encouraged with continued suction of the pink frothy sputum to help smooth respiration. Most of the sputum was removed after 12 hours, and the pulmonary edema was significantly improved based on the chest x-ray (Fig. 2A). The CPAP mask was removed, and oxygen was supplied with a venturi mask. The patient was moved to a general ward about 24 hours after operation, and the fluid balance was continuously controlled. The SpO2 was maintained at 97% to 99%, and the chest x-ray became normal on the following day. The patient received 5 days of postoperative management and was discharged without other problems. A chest x-ray taken at 3 months after the operation confirmed total improvement in the status of pulmonary edema (Fig. 2B).
DISCUSSION
FIGURE 1. A, Chest posterior-anterior view before surgery shows no remarkable findings in the 26-year-old male patient scheduled for orthognathic surgery. The patient had no special medical history and abnormality on the laboratory results nor in the upper airway. B, Chest posterior-anterior view 30 minutes postoperatively. Chest x-ray checked in the postanesthetic care unit reveals full hazy hilum (A), prominence of the pulmonary vasculature and haziness on both lungs distribution (B), and increased heart size (C), thus implicating pulmonary edema. Clinical symptom was irregular, shallow breathing immediately after extubation. The oxygen saturation was reduced, the spontaneous respiration was insufficient, and the symptoms became increasingly severe. The patient complained of discomfort in the chest while breathing. However, he had a clear consciousness, and he was continuously excreting large amounts of pink frothy sputum.
e434
The NPPE can occur from acute or continued obstruction of the upper airway and mainly occurs during emergence from general anesthesia. Although there are differences, it has been estimated that NPPE develops in 11% of patients2 and requires active intervention for acute upper airway obstruction. The morbidity and mortality associated with NPPE is 11% to 40%, and it is primarily associated with delayed recognition and treatment. Halow and Ford3 reported that the ability to generate large intrapulmonary pressures with a very compliant chest wall may play a significant role in the pathophysiology of NPPE. Young, healthy patients are capable of generating large negative intrathoracic pressures by virtue of their increased level of fitness, which increases the risk of NPPE.4 Another risk factors are anatomically difficult intubation, nasal, oral, or pharyngeal surgical site of pathology; obesity with obstructive apnea; short neck; and acromegaly.5 Especially in case of orthognatic surgery, there are increased risks of airway obstruction and NPPE, because mandibular retropositioning can cause airway narrowing. © 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery • Volume 25, Number 5, September 2014
In general, there are many pathophysiologic processes that can lead to NPPE. Excessive inspiratory force is the main cause and was reported to reach as high as −100 mm Hg during upper airway obstruction. This excessive negative pressure is thought to cause disruption of the alveolar capillaries to cause pulmonary edema and, sometimes, alveolar hemorrhage.6 Moreover, this event leads to an increase in venous return and blood flow to the right heart. However, hyperadrenergic state induced by hypoxia can increase systemic vascular resistance, pulmonary vascular resistance, and pulmonary capillary pressure and subsequently increase afterload.1, 5 This combination causes an increase in pulmonary blood volume and pulmonary venous pressure and then an increase in hydrostatic pressure and edema formation.5, 7 Most of NPPEs arise immediately after relief of an airway obstruction; however, a delayed onset of up to 24 hours has been reported.4 The clinical signs of NPPE are typical. After recovery from airway obstruction, the patient presents tachypnea, dyspnea, and decreased SpO2 levels despite of supplying oxygen. In addition, tachycardia and hypertension also can arise because of increased sympathetic activity. We can hear rales and rhonchi on chest auscultation,8 and chest radiograph shows rapid bilateral changes consistent with pulmonary edema.9 All the treatments for NPPE are supportive treatments and are able to be executed by any surgeon. 1. 2. 3. 4. 5.
Maintenance of the airway Supplying oxygen Hemodynamic stability Positive pressure ventilation Diuretics and appropriate fluid therapy
Although active treatment using intubation and positive pressure is required for severe cases of hypoxia, the use of CPAP for the treatment of acute pulmonary edema and respiratory failure is generally broadly accepted. There have been reports of successful treatment using noninvasive CPAP via facial masks or a nasal cannula.10 In this case, after recovering from airway obstruction with soft positive pressure ventilation and lidocaine administration, the patient had a clear consciousness and was able to excrete the sputum. The possibility of pulmonary aspiration was considered to be low, so CPAP could be applied through a mask while maintaining spontaneous respiration with assisted oral suctioning of the pink frothy sputum. As a result of the parallel allopathic treatments, the sputum in the lungs was removed, and the respiration sounds became clean on the ausculation of the thorax. After such treatment, the patient completely recovered without other problems. In conclusion, the patients receiving orthognathic surgery are easily exposed to the risk of NPPE because of upper airway obstruction. This event can cause fatal outcomes if quick and accurate measures are not taken. So, careful observation of the patient and accurate knowledge are required to treat this malady when it does occur. Active, quick, and adequate measures must be taken to treat the complications of NPPE.
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Inferior Alveolar Nerve Repositioning and Orthognathic Surgery Amin Rahpeyma,* Saeedeh Khajehahmadi, DDS† Background: Inferior alveolar nerve (IAN) repositioning is a wellknown technique in implant dentistry. This invaluable technique can be combined with orthognathic techniques to enhance the effect of orthognathic surgery in some selected cases. Materials and Methods: In a retrospective study, data were obtained from the archived files of Department of Oral and Maxillofacial Surgery, Mashhad University of Medical Sciences, Iran, from 2007 to 2012. Patients undergoing mandibular orthognathic surgery alone or in combination with maxillary surgery were searched. Cases with IAN repositioning were selected. Results: In this study, in 4% of orthognathic surgical procedures involving the mandibular bone, IAN repositioning was indicated. Genioplasty, body ostectomy, total mandibular subapical alveolar osteotomy, and inferior border osteotomy for correction of asymmetric mandibular excess in hemimandibular hyperplasia were in this list. Conclusion: In carefully selected orthognathic patients, IAN repositioning can enhance the effect of orthognathic surgery and should be considered in the treatment plan strategies as an option. From the *Oral and Maxillofacial Diseases Research Center and †Dental Research Center, School of Dentistry, Mashhad University of Medical Sciences, Mashhad, Iran. Received January 28, 2014. Accepted for publication February 17, 2014. Address correspondence and reprint requests to Saeedeh Khajehahmadi, DDS, Dental Research Center, Faculty of Dentistry, Mashhad University of Medical Sciences, Vakilabad Blvd, Mashhad, P.O. Box: 91735-984, Iran; E-mail: [email protected] or [email protected] Supported by a grant from the Vice Chancellor of Research of Mashhad University of Medical Sciences. The authors report no conflicts of interest. Copyright © 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0000000000000945
© 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
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