Case report severe postoperative negative pressure pulmonary edema

Case report severe postoperative negative pressure pulmonary edema | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Case report severe postoperative negative pressure pulmonary edema Philipp Kazuo Omuro, David Sander, Dominique Hart This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-5018334/v1 This work is licensed under a CC BY 4.0 License Status: Published Journal Publication published 01 Nov, 2024 Read the published version in BMC Anesthesiology → Version 1 posted 11 You are reading this latest preprint version Abstract Background : Postoperative negative pressure pulmonary edema (NPPE) can occur in any patient undergoing general anesthesia. There are several risk factors for it, especially postoperative laryngospasm. The disease is usually benign and quickly reversible. In our case the severity and need for advanced critical care therapy was unusual. Case : We report of severe case of postoperative negative pressure pulmonary edema in a 62-year-old male patient undergoing elective right sided retroperitoneoscopic adrenalectomy. The patient developed a severe case of acute respiratory distress syndrome (ARDS) after postoperative laryngospasm, possibly in conjunction with a suspected anaphylactic reaction. The patient was consequently treated with a combination of invasive airway pressure release ventilation (APRV) and a prone positioning regimen. After drastic improvement of respiratory function, the patient was discharged from intensive care unit after 10 days and from hospital after 14 days. Conclusion : NPPE is a rare, but relevant complication of anesthesia and laryngospasm. The disease can basically occur in any patient undergoing general anesthesia and therefore should be considered. laryngospasm pulmonary edema intraoperative complications ARDS APRV Ventilation Mode Case We report the case of a 62-year-old male undergoing elective right sided retroperitoneoscopic adrenalectomy due to pheochromocytoma. The patient bodyweight was 75 kg at 175 cm body-height. Arterial hypertension was the only finding in his medical history. Previous he underwent uneventful general anesthesia twice. A preoperative treatment of the pheochromocytoma-induced mild arterial hypertension by alpha receptor blockade was not recommended by the endocrinologists. Following the uneventful induction of anesthesia by intravenous (i.v.) application of 30 µg Sufentanil, 400 mg Propofol and 50 mg Rocuronium bougie-guided endotracheal intubation was performed in Cormack & Lehane III conditions upon direct laryngoscopy. Balanced general anesthesia was maintained by inhalative Sevoflurane (endexpiratory concentrations 1,6-1,8 Vol%) and continuous i.v. infusion of Remifentanil (0,3 µg/kg/min). Intraoperative arterial hypotension necessitated hemodynamic support employing Noradrenaline at i.v. doses up to 0,3 µg/kg/min. An unexplained development of facial exanthema led to the clinical diagnosis of an allergic reaction of unknown cause which was subsequently treated by administration of 250mg Prednisolone i.v. and 8mg Dimetindene. There were no signs of accompanied hemodynamic instability or bronchospasm. Upon termination of surgery neuromuscular blockade was reversed by 200 mg Sugammadex i.v. and a Train-of-Four count (TOF) of 100 % was achieved. Following extubation the patient immediately developed clinical signs of a severe case of laryngospasm (inspiratory stridor, orthopnea and hyperventilation) resulting in a dip in the peripheral oxygen saturation (SpO 2 ) to 88%. In face of lacking a patent airway due to laryngospasm and the imminent threat of hypoxia, general anesthesia was reinstituted using propofol and remifentanil. The airway was secured via the placement of a laryngeal mask airway, thus reestablishing a controlled ventilation and oxygenation. After the return of sufficient spontaneous breathing with adequate oxygenation anesthesia was terminated and the laryngeal mask was removed. During the transfer from the operation room to the post anesthesia care unit the patient developed a rapidly deteriorating hypoxia with a decrease in SpO 2 to 65%, paO 2 < 40 mmHg, hypercapnia (paCO 2 60 mmHg) and progressive signs of respiratory decompensation (Table 1). During the immediate preparations for reintubation the SpO 2 degraded further to 35%. Preoxygenation with manual mask-CPAP and inspiratory O2 concentration of 100% (FiO 2 1,0) a maximum SpO 2 of 88% was achieved. After i.v. induction (10 mg Midazolam, 100 mg Ketamine, 100 mg Rocuronium) intubation was performed using a videolaryngoscope (hyper-angulated blade size 4). Initially the laryngeal view was obstructed by small amounts of a foamy, lightly red tinged fluid which was removed via suction and the trachea was successfully intubated using a Magill # 8.0 tube. The consecutive bronchoscopy showed no pathological findings. Consecutively an emergency CT-scan (thorax, abdomen) was performed (Figure 1). The CT-scan showed massive pulmonary alveolar edema of both lungs, worsening from apical to basal and from anteriorly to posteriorly. After initial stabilization of oxygenation following reintubation, a continuous degradation of gas exchange parameters over the next hours was noted. Despite employing a ventilatory strategy using APRV (peak airway pressure 32 mbar, positive end expiratory pressure 0 mbar, inspiratory to expiratory ratio 7:1, FiO2 1,0) paO2 dropped below 60 mmHg (Table 1). Ultimately the patient rapidly developed the clinical finding of ARDS. In preparation for an eventual extracorporeal pulmonary support the patient was transferred to another in-house intensive care unit. Upon arrival prone positioning was instituted. Approximately 1000 ml of light red pulmonary edema was mobilized spontaneously during the prone positioning maneuver and drained from the endotracheal tube. Following the secrete mobilization oxygenation improved significantly (Horovitz index from 56 to 150). Intermittent prone positioning was terminated after two days. In course of the next 24 hours oxygenation indices continuously improved, leading to a successful extubation on the third postoperative day. In the following period respiration was stabilized by use of high flow nasal oxygen and non-invasive ventilation. Sonography and a follow-up CT-scan on the 7 th postoperative day (Figure 2) showed relevant bilateral pleural effusion which were drained on the 7 th and 8 th postoperative day. Echocardiography on postoperative day 10 revealed a possible pulmonary hypertension (systolic pulmonary artery pressure 50 mmHg incl. central venous pressure). Critical care therapy encompassed an empirical i.v antibiotic with Piperacillin/Tazobactam from day 0 to day 6 and Meropenem from day 6 to day 10. The patient was discharged from the intensive care unit on the 10 th postoperative day and discharged from hospital on postoperative day 14 with good cardiopulmonary function despite showing residual symptoms of exertional dyspnea and feeling of general weakness. After discharge written informed consent was obtained by the patient for publication of this case report. Discussion and Conclusion Pulmonary edema can be a symptom or complication of various diseases. The somewhat fragile alveolar architecture is prone to injury, e.g. due to mechanical stress, hydrostatic or hemodynamic changes and/or capillary leakage. Factors contributing to the development of a pulmonary edema are a low osmotic pressure, high capillary permeability and a high negative pressure gradient and vice versa [1]. The pathophysiological changes ultimately lead to an increase of the alveolar diffusion distance, a decrease in gas exchange area and an increased work of breathing. Negative pressure pulmonary edema (NPPE) is typically caused by a rapid and extreme drop in intrathoracic pressure. Ma et al. report a pressures as low as -140 cmH 2 O during forced inspiration [2]. These low intrathoracic pressures may usually occur during spontaneous breathing efforts with an obstructed/occluded airway. Low intrathoracic pressure also transduces to the pulmonary vascular system. As venous return increases pulmonary blood volume and right ventricular workload increase as well as resulting in an increased transcapillary hydrostatics pressure [2]. Considering hypoxemia being present in many cases of laryngospasm and NPPE, hypoxemic pulmonary vasoconstriction can also contribute to altered capillary permeability. Those mechanisms favor the formation of severe alveolar edema. Furthermore, mechanical stress induced by negative pulmonary pressure increases capillary permeability [2]. Several situations during general anesthesia bear the risk of NPPE. One of the most common settings associated with a high risk for developing a NPPE is the occurrence of a laryngospasm during the post-extubation phase [1],[2]. Furthermore biting of the unconscious patient under general anesthesia onto the endotracheal tube or laryngeal mask can lead to an airway obstruction with a relevant risk of NPPE development [3]. Ma et al. described sleep apnea and anatomical intubation difficulties as individual risk factors for perioperative NPPE [2]. In the non-perioperative setting the disease can occur due to various pathologic changes ranging from severe infections such as epiglottitis or pseudocroup, strangulation injury and vocal cord paralysis due to tumors of the upper airway [3]. Alb et al. describe a type I and type II NPPE. Type I is typically induced by laryngospasm, type II is caused by de-obstruction of chronically obstructed airway. The type II reaction is due to a sudden reduction of chronically enhanced intrinsic PEEP after extubation, e.g. after naso-pharyngeal tumor surgery [3]. The therapeutic options for NPPE are focused on the treatment of the pathophysiological cause. Therefore positive airway pressures through non-invasive or invasive ventilation are employed to counteract the initial negative transpulmonary pressure and thereby to reduce pulmonary edema [1]. Additional supportive therapy by means of inhalative beta-mimetic drugs, steroids and diuretics have been discussed in literature but clear evidence for their benefits is lacking [4],[5]. While the clinical findings in NPPE may impose as a severe case of ARDS, pulmonary dysfunction in most patients resolves within 48 h [5]. Our patient developed an extreme case of NPPE with the rapid degradation of pulmonary function resulting in severe ARDS. Despite the typical mechanical cause of pulmonary edema, anaphylactic capillary disorder might have been a cofounding factor for the formation of NPPE [6]. Successful extubation was achieved after 72 hours of invasive ventilatory therapy in conjunction with prone-positioning, followed by respiratory support via high flow nasal oxygen therapy and non-invasive ventilation until pulmonary recovery. Considering the available literature this case showed a rather severe and prolonged impairment of pulmonary function. This might be due to the suspected multifactorial etiology of NPPE coinciding with an allergic reaction. Alb et al. suggest a sufficient depth of anesthesia, especially in management of head-neck surgery cases and whenever any airway manipulation under general anesthesia may be warranted. Airway devices might also be protected from biting by various means such as placement of a Guedel tube [3]. In our case a more delicate termination of anesthesia paired with a higher vigilance for the timepoint of full emergence from general anesthesia prior to extubation might have minimized the risk for NPPE in our patient. However, NPPE may potentially develop in any patient undergoing general anesthesia and should be carefully considered as a possible cause of sudden pulmonary dysfunction occurring after extubation. “Intubation is a skill, extubation is an art [7].” In conclusion negative pressure pulmonary edema is a usually benign, rare, but typical complication of laryngospasm under/after general anesthesia and other clinical scenarios presenting as a severe case of acute respiratory dysfunction occurring after extubation. The pathophysiological mechanisms are complex. Treatment options are aimed at counteracting these underlying mechanisms. Depending on the severity of clinical symptoms invasive ventilation may be necessary for rapid and successful treatment. In most cases the pulmonary dysfunction resolves quickly after initiation of appropriate ventilatory therapy. Abbreviations APRV airway pressure release ventilation ARDS acute respiratory distress syndrome cm Centimeter cmH2O Centimeter of water FiO2 inspiratory Oxygen fraction i.v. intravenous kg Kilogram mmHg Millimeter of Mercury NPPE negative pressure pulmonary edema Paco arterial partial pressure of Carbon dioxide Pao2 arterial partial pressure of Oxygen PEEP positive endexspiratory pressure SpO2 Oxygen saturation TOF Train-of-Four Declarations Ethics approval and consent to participate: No applicable for case report. Consent to participate granted by the patient. Consent for publication: Consent for publication granted by the patient. Availability of data and materials: Only the individual data set of clinical routine data of one patient of used in this publication. Competing interests: No competing interests by any author. Fundings: No specific fundings for this research project. Authors’ contributions: All authors were involved in direct patient care, development and writing of the manuscript. Dr. Philipp Kazuo Omuro is the corresponding author. Acknowledgements: We thank the patient for the trust in our therapy and the consent to participate in this case report. References Bhattacharya M, Kallet RH, Ware LB, Matthay MA. Negative-Pressure Pulmonary Edema. Chest . 2016; 50:4:927-933. Ma J, Tiantian W, Qiang X, Xiaohua G, Zhiqiang F, Yan Y, et al. Negative pressure pulmonary edema, Exp. Ther. Med. 2023; 26:455. Alb M, Tsagorgiorgas C, Meinhardt J. Negative-pressure pulmonary edeme (NPPE). Anästhesiol Intensivmed Notfallmed Schmerzther . 2006; 41:64-78. Liu R, Wang J, G. Zhao G, Su Z. Negative pressure pulmonary edema after general anesthesia: A case report and literature review. Med. (United States) . 2019; 98:17 Krodel DJ, Bittner EA, Abdulnour R, Brown R, Eikermann M. Case scenario: Acute postoperative negative pressure pulmonary edema. Anesthesiology . 2010; 113:1: 200-207. Ebo DG, Clarke RC, Mertes PM, Platt PR, Sabato V, Sadleir PHM. Molecular mechanisms and pathophysiology of perioperative hypersensitivity and anaphylaxis: a narrative review. Br. J. Anaesth. 2019; 123:1:38-49. Armin&Gordon. Folge #005 -Intubation is a skill, extubation is an art!. Kinderanästhesie-Talk [Podcast]. URL: https://kinderanaesthesie-talk.podigee.io/7-005_isea Table Table 1 is available in the Supplementary Files section. Additional Declarations No competing interests reported. Supplementary Files Table1.png Cite Share Download PDF Status: Published Journal Publication published 01 Nov, 2024 Read the published version in BMC Anesthesiology → Version 1 posted Editorial decision: Revision requested 11 Oct, 2024 Reviews received at journal 10 Oct, 2024 Reviews received at journal 09 Oct, 2024 Reviewers agreed at journal 02 Oct, 2024 Reviewers agreed at journal 30 Sep, 2024 Reviewers agreed at journal 29 Sep, 2024 Reviewers invited by journal 29 Sep, 2024 Editor invited by journal 09 Sep, 2024 Editor assigned by journal 03 Sep, 2024 Submission checks completed at journal 03 Sep, 2024 First submitted to journal 02 Sep, 2024 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-5018334","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":361599610,"identity":"4225a132-4c67-4a85-adcc-c869659ec221","order_by":0,"name":"Philipp Kazuo Omuro","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAABAElEQVRIiWNgGAWjYDACCcbGAwlwXgWDAQMPkH7AwMDYgFtLA5KWM1AtCXi1MDAcgHMY24jQYi7d3HDg4Y47DPxiZ8w+fJx32JiB5/DBB4k7GGT7cWixnHOw4UDimWcMkrNzjGfO3HbYjIG3Ldkg8QyD8Uwc1hjcSARqaTvMYHA7x5iZd9thG/vzPGYSiW0MiRsOEKVlzmEbBn4e8x8gLfuJ09IAcliPGQPYFlx+mZEI9guP5Oy0YsYZx9KB3j+WDHSYhPEMHLaYS6Q/fPhzxx05funkzQwfaqwNG3iSD3742GYj24/L+yACGJs86BISOJyF0IJTwSgYBaNgFIwCBgAxnGKbKIl0jAAAAABJRU5ErkJggg==","orcid":"","institution":"University Hospital Cologne","correspondingAuthor":true,"prefix":"","firstName":"Philipp","middleName":"Kazuo","lastName":"Omuro","suffix":""},{"id":361599611,"identity":"1c201639-0a20-422d-8cba-83c0c0e377e6","order_by":1,"name":"David Sander","email":"","orcid":"","institution":"University Hospital Cologne","correspondingAuthor":false,"prefix":"","firstName":"David","middleName":"","lastName":"Sander","suffix":""},{"id":361599612,"identity":"262c73fc-2111-46d9-bec8-3be7963f3254","order_by":2,"name":"Dominique Hart","email":"","orcid":"","institution":"University Hospital Cologne","correspondingAuthor":false,"prefix":"","firstName":"Dominique","middleName":"","lastName":"Hart","suffix":""}],"badges":[],"createdAt":"2024-09-02 12:31:11","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-5018334/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-5018334/v1","draftVersion":[],"editorialEvents":[{"content":"https://doi.org/10.1186/s12871-024-02785-2","type":"published","date":"2024-11-01T15:56:55+00:00"}],"editorialNote":"","failedWorkflow":false,"files":[{"id":68206401,"identity":"d85ea398-19fd-4076-9e56-1ecd1e4dc896","added_by":"auto","created_at":"2024-11-04 16:32:10","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":212463,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-5018334/v1/04992155-776b-4aa8-9ca3-7f080d9660a9.pdf"},{"id":65927170,"identity":"ce66b61f-64d7-42f1-830d-b90cba8ba476","added_by":"auto","created_at":"2024-10-04 13:11:51","extension":"png","order_by":1,"title":"","display":"","copyAsset":false,"role":"supplement","size":649379,"visible":true,"origin":"","legend":"","description":"","filename":"Table1.png","url":"https://assets-eu.researchsquare.com/files/rs-5018334/v1/35326f0346ffbea4ac85e237.png"}],"financialInterests":"No competing interests reported.","formattedTitle":"Case report severe postoperative negative pressure pulmonary edema","fulltext":[{"header":"Case","content":"\u003cp\u003eWe report the case of a 62-year-old male undergoing elective right sided retroperitoneoscopic adrenalectomy due to pheochromocytoma. The patient bodyweight was 75 kg at 175 cm body-height. Arterial hypertension was the only finding in his medical history. Previous he underwent uneventful general anesthesia twice. A preoperative treatment of the pheochromocytoma-induced mild arterial hypertension by alpha receptor blockade was not recommended by the endocrinologists. \u003c/p\u003e\n\n\u003cp\u003eFollowing the uneventful induction of anesthesia by intravenous (i.v.) application of 30 \u0026micro;g Sufentanil, 400 mg Propofol and 50 mg Rocuronium bougie-guided endotracheal intubation was performed in Cormack \u0026amp; Lehane III conditions upon direct laryngoscopy. Balanced general anesthesia was maintained by inhalative Sevoflurane (endexpiratory concentrations 1,6-1,8 Vol%) and continuous i.v. infusion of Remifentanil (0,3 \u0026micro;g/kg/min). Intraoperative arterial hypotension necessitated hemodynamic support employing Noradrenaline at i.v. doses up to 0,3 \u0026micro;g/kg/min. An unexplained development of facial exanthema led to the clinical diagnosis of an allergic reaction of unknown cause which was subsequently treated by administration of 250mg Prednisolone i.v. and 8mg Dimetindene. There were no signs of accompanied hemodynamic instability or bronchospasm. Upon termination of surgery neuromuscular blockade was reversed by 200 mg Sugammadex i.v. and a Train-of-Four count (TOF) of 100 % was achieved. \u003c/p\u003e\n\n\u003cp\u003eFollowing extubation the patient immediately developed clinical signs of a severe case of laryngospasm (inspiratory stridor, orthopnea and hyperventilation) resulting in a dip in the peripheral oxygen saturation (SpO\u003csub\u003e2\u003c/sub\u003e) to 88%. In face of lacking a patent airway due to laryngospasm and the imminent threat of hypoxia, general anesthesia was reinstituted using propofol and remifentanil. The airway was secured via the placement of a laryngeal mask airway, thus reestablishing a controlled ventilation and oxygenation. After the return of sufficient spontaneous breathing with adequate oxygenation anesthesia was terminated and the laryngeal mask was removed. \u003c/p\u003e\n\u003cp\u003eDuring the transfer from the operation room to the post anesthesia care unit the patient developed a rapidly deteriorating hypoxia with a decrease in SpO\u003csub\u003e2\u003c/sub\u003e to 65%, paO\u003csub\u003e2\u003c/sub\u003e \u0026lt; 40 mmHg, hypercapnia (paCO\u003csub\u003e2\u003c/sub\u003e 60 mmHg) and progressive signs of respiratory decompensation (Table 1). During the immediate preparations for reintubation the SpO\u003csub\u003e2\u003c/sub\u003e degraded further to 35%. Preoxygenation with manual mask-CPAP and inspiratory O2 concentration of 100% (FiO\u003csub\u003e2\u003c/sub\u003e 1,0) a maximum SpO\u003csub\u003e2\u003c/sub\u003e of 88% was achieved. After i.v. induction (10 mg Midazolam, 100 mg Ketamine, 100 mg Rocuronium) intubation was performed using a videolaryngoscope (hyper-angulated blade size 4). Initially the laryngeal view was obstructed by small amounts of a foamy, lightly red tinged fluid which was removed via suction and the trachea was successfully intubated using a Magill # 8.0 tube. The consecutive bronchoscopy showed no pathological findings. Consecutively an emergency CT-scan (thorax, abdomen) was performed (Figure 1). \u003c/p\u003e\n\n\u003cp\u003eThe CT-scan showed massive pulmonary alveolar edema of both lungs, worsening from apical to basal and from anteriorly to posteriorly. After initial stabilization of oxygenation following reintubation, a continuous degradation of gas exchange parameters over the next hours was noted. Despite employing a ventilatory strategy using APRV (peak airway pressure 32 mbar, positive end expiratory pressure 0 mbar, inspiratory to expiratory ratio 7:1, FiO2 1,0) paO2 dropped below 60 mmHg (Table 1). Ultimately the patient rapidly developed the clinical finding of ARDS. \u003c/p\u003e\n\n\u003cp\u003eIn preparation for an eventual extracorporeal pulmonary support the patient was transferred to another in-house intensive care unit. Upon arrival prone positioning was instituted. Approximately 1000 ml of light red pulmonary edema was mobilized spontaneously during the prone positioning maneuver and drained from the endotracheal tube. Following the secrete mobilization oxygenation improved significantly (Horovitz index from 56 to 150). Intermittent prone positioning was terminated after two days. In course of the next 24 hours oxygenation indices continuously improved, leading to a successful extubation on the third postoperative day. In the following period respiration was stabilized by use of high flow nasal oxygen and non-invasive ventilation. Sonography and a follow-up CT-scan on the 7\u003csup\u003eth\u003c/sup\u003e postoperative day (Figure 2) showed relevant bilateral pleural effusion which were drained on the 7\u003csup\u003eth\u003c/sup\u003e and 8\u003csup\u003eth\u003c/sup\u003e postoperative day. \u003c/p\u003e\n\n\u003cp\u003eEchocardiography on postoperative day 10 revealed a possible pulmonary hypertension (systolic pulmonary artery pressure 50 mmHg incl. central venous pressure). Critical care therapy encompassed an empirical i.v antibiotic with Piperacillin/Tazobactam from day 0 to day 6 and Meropenem from day 6 to day 10. The patient was discharged from the intensive care unit on the 10\u003csup\u003eth\u003c/sup\u003e postoperative day and discharged from hospital on postoperative day 14 with good cardiopulmonary function despite showing residual symptoms of exertional dyspnea and feeling of general weakness. \u003c/p\u003e\n\n\u003cp\u003eAfter discharge written informed consent was obtained by the patient for publication of this case report. \u003c/p\u003e"},{"header":"Discussion and Conclusion","content":"\u003cp\u003ePulmonary edema can be a symptom or complication of various diseases. The somewhat fragile alveolar architecture is prone to injury, e.g. due to mechanical stress, hydrostatic or hemodynamic changes and/or capillary leakage. Factors contributing to the development of a pulmonary edema are a low osmotic pressure, high capillary permeability and a high negative pressure gradient and vice versa\u0026nbsp;[1]. The pathophysiological changes ultimately lead to an increase of the alveolar diffusion distance, a decrease in gas exchange area and an increased work of breathing. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003eNegative pressure pulmonary edema (NPPE) is typically caused by a rapid and extreme drop in intrathoracic pressure. Ma et al. report a pressures as low as -140 cmH\u003csub\u003e2\u003c/sub\u003eO during forced inspiration\u0026nbsp;[2]. These low intrathoracic pressures may usually occur during spontaneous breathing efforts with an obstructed/occluded airway. Low intrathoracic pressure also transduces to the pulmonary vascular system. As venous return increases pulmonary blood volume and right ventricular workload increase as well as resulting in an increased transcapillary hydrostatics pressure\u0026nbsp;[2]. Considering hypoxemia being present in many cases of laryngospasm and NPPE, hypoxemic pulmonary vasoconstriction can also contribute to altered capillary permeability. Those mechanisms favor the formation of severe alveolar edema. Furthermore, mechanical stress induced by negative pulmonary pressure increases capillary permeability\u0026nbsp;[2]. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;Several situations during general anesthesia bear the risk of NPPE. One of the most common settings associated with a high risk for developing a NPPE is the occurrence of a laryngospasm during the post-extubation phase\u0026nbsp;[1],[2]. Furthermore biting of the unconscious patient under general anesthesia onto the endotracheal tube or laryngeal mask can lead to an airway obstruction with a relevant risk of NPPE development\u0026nbsp;[3]. Ma et al. described sleep apnea and anatomical intubation difficulties as individual risk factors for perioperative NPPE\u0026nbsp;[2].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIn the non-perioperative setting the disease can occur due to various pathologic changes ranging from severe infections such as epiglottitis or pseudocroup, strangulation injury and vocal cord paralysis due to tumors of the upper airway\u0026nbsp;[3].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAlb et al. describe a type I and type II NPPE. Type I is typically induced by laryngospasm, type II is caused by de-obstruction of chronically obstructed airway. The type II reaction is due to a sudden reduction of chronically enhanced intrinsic PEEP after extubation, e.g. after naso-pharyngeal tumor surgery\u0026nbsp;[3].\u003c/p\u003e\n\u003cp\u003eThe therapeutic options for NPPE are focused on the treatment of the pathophysiological cause. Therefore positive airway pressures through non-invasive or invasive ventilation are employed to counteract the initial negative transpulmonary pressure and thereby to reduce pulmonary edema\u0026nbsp;[1]. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAdditional supportive therapy by means of inhalative beta-mimetic drugs, steroids and diuretics have been discussed in literature but clear evidence for their benefits is lacking\u0026nbsp;[4],[5].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eWhile the clinical findings in NPPE may impose as a severe case of ARDS, \u0026nbsp;pulmonary dysfunction in most patients resolves within 48 h\u0026nbsp;[5].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eOur patient developed an extreme case of NPPE with the rapid degradation of pulmonary function resulting in severe ARDS. Despite the typical mechanical cause of pulmonary edema, anaphylactic capillary disorder might have been a cofounding factor for the formation of NPPE [6]. Successful extubation was achieved after 72 hours of invasive ventilatory therapy in conjunction with prone-positioning, followed by respiratory support via high flow nasal oxygen therapy and non-invasive ventilation until pulmonary recovery. Considering the available literature this case showed a rather severe and prolonged impairment of pulmonary function. This might be due to the suspected multifactorial etiology of NPPE coinciding with an allergic reaction.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eAlb et al. suggest a sufficient depth of anesthesia, especially in management of head-neck surgery cases and whenever any airway manipulation under general anesthesia may be warranted. Airway devices might also be protected from biting by various means such as placement of a Guedel tube [3]. In our case a more delicate termination of anesthesia paired with a higher vigilance for the timepoint of full emergence from general anesthesia prior to extubation might have minimized the risk for NPPE in our patient. However, NPPE may potentially develop in any patient undergoing general anesthesia and should be carefully considered as a possible cause of sudden pulmonary dysfunction occurring after extubation.\u0026nbsp;\u003cbr\u003e\u0026nbsp;\u0026ldquo;Intubation is a skill, extubation is an art [7].\u0026rdquo;\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eIn conclusion negative pressure pulmonary edema is a usually benign, rare, but typical complication of laryngospasm under/after general anesthesia and other clinical scenarios presenting as a severe case of acute respiratory dysfunction occurring after extubation. The pathophysiological mechanisms are complex. Treatment options are aimed at counteracting these underlying mechanisms. Depending on the severity of clinical symptoms invasive ventilation may be necessary for rapid and successful treatment. In most cases the pulmonary dysfunction resolves quickly after initiation of appropriate ventilatory therapy.\u0026nbsp;\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eAPRV airway pressure release ventilation\u003c/p\u003e\n\u003cp\u003eARDS acute respiratory distress syndrome \u003c/p\u003e\n\u003cp\u003ecm Centimeter\u003c/p\u003e\n\u003cp\u003ecmH2O Centimeter of water \u003c/p\u003e\n\u003cp\u003eFiO2 inspiratory Oxygen fraction \u003c/p\u003e\n\u003cp\u003ei.v. intravenous \u003c/p\u003e\n\u003cp\u003ekg Kilogram\u003c/p\u003e\n\u003cp\u003emmHg Millimeter of Mercury \u003c/p\u003e\n\u003cp\u003eNPPE negative pressure pulmonary edema \u003c/p\u003e\n\u003cp\u003ePaco arterial partial pressure of Carbon dioxide \u003c/p\u003e\n\u003cp\u003ePao2 arterial partial pressure of Oxygen \u003c/p\u003e\n\u003cp\u003ePEEP positive endexspiratory pressure \u003c/p\u003e\n\u003cp\u003eSpO2 Oxygen saturation \u003c/p\u003e\n\u003cp\u003eTOF Train-of-Four\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cu\u003eEthics approval and consent to participate: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eNo applicable for case report. Consent to participate granted by the patient.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003e \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003e\u003cu\u003eConsent for publication: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eConsent for publication granted by the patient. \u003c/p\u003e\n\n\u003cp\u003e\u003cu\u003eAvailability of data and materials: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eOnly the individual data set of clinical routine data of one patient of used in this publication. \u003c/p\u003e\n\n\u003cp\u003e\u003cu\u003eCompeting interests: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eNo competing interests by any author. \u003c/p\u003e\n\u003cp\u003e\u003cu\u003e \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003e\u003cu\u003eFundings: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eNo specific fundings for this research project. \u003c/p\u003e\n\n\u003cp\u003e\u003cu\u003eAuthors\u0026rsquo; contributions: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eAll authors were involved in direct patient care, development and writing of the manuscript.\u003c/p\u003e\n\u003cp\u003eDr. Philipp Kazuo Omuro is the corresponding author. \u003c/p\u003e\n\n\u003cp\u003e\u003cu\u003eAcknowledgements: \u003c/u\u003e\u003c/p\u003e\n\u003cp\u003eWe thank the patient for the trust in our therapy and the consent to participate in this case report.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eBhattacharya M, Kallet RH, Ware LB, Matthay MA. Negative-Pressure Pulmonary Edema. \u003cem\u003eChest\u003c/em\u003e. 2016; 50:4:927-933. \u003c/li\u003e\n\u003cli\u003eMa J, Tiantian W, Qiang X, Xiaohua G, Zhiqiang F, Yan Y, \u003cem\u003eet al.\u003c/em\u003e Negative pressure pulmonary edema, \u003cem\u003eExp. Ther. Med. 2023; \u003c/em\u003e 26:455.\u003c/li\u003e\n\u003cli\u003eAlb M, Tsagorgiorgas C, Meinhardt J. Negative-pressure pulmonary edeme (NPPE). \u003cem\u003eAn\u0026auml;sthesiol Intensivmed Notfallmed Schmerzther\u003c/em\u003e. 2006; 41:64-78. \u003c/li\u003e\n\u003cli\u003eLiu R, Wang J, G. Zhao G, Su Z. Negative pressure pulmonary edema after general anesthesia: A case report and literature review. \u003cem\u003eMed. (United States)\u003c/em\u003e. 2019; 98:17 \u003c/li\u003e\n\u003cli\u003eKrodel DJ, Bittner EA, Abdulnour R, Brown R, Eikermann M. Case scenario: Acute postoperative negative pressure pulmonary edema. \u003cem\u003eAnesthesiology\u003c/em\u003e. 2010; 113:1: 200-207. \u003c/li\u003e\n\u003cli\u003eEbo DG, Clarke RC, Mertes PM, Platt PR, Sabato V, Sadleir PHM. Molecular mechanisms and pathophysiology of perioperative hypersensitivity and anaphylaxis: a narrative review. \u003cem\u003eBr. J. Anaesth.\u003c/em\u003e 2019; 123:1:38-49.\u003c/li\u003e\n\u003cli\u003eArmin\u0026amp;Gordon. Folge #005 -Intubation is a skill, extubation is an art!. Kinderan\u0026auml;sthesie-Talk [Podcast]. URL: https://kinderanaesthesie-talk.podigee.io/7-005_isea\u003cu\u003e\u003c/u\u003e\u003c/li\u003e\n\u003c/ol\u003e"},{"header":"Table","content":"\u003cp\u003eTable 1 is available in the Supplementary Files section.\u003c/p\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bmc-anesthesiology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bane","sideBox":"Learn more about [BMC Anesthesiology](http://bmcanesthesiol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/bane","title":"BMC Anesthesiology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"laryngospasm, pulmonary edema, intraoperative complications, ARDS, APRV Ventilation Mode","lastPublishedDoi":"10.21203/rs.3.rs-5018334/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-5018334/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground\u003c/strong\u003e: Postoperative negative pressure pulmonary edema (NPPE) can occur in any patient undergoing general anesthesia. There are several risk factors for it, especially postoperative laryngospasm. The disease is usually benign and quickly reversible. In our case the severity and need for advanced critical care therapy was unusual.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase\u003c/strong\u003e: We report of severe case of postoperative negative pressure pulmonary edema in a 62-year-old male patient undergoing elective right sided retroperitoneoscopic adrenalectomy. The patient developed a severe case of acute respiratory distress syndrome (ARDS) after postoperative laryngospasm, possibly in conjunction with a suspected anaphylactic reaction. The patient was consequently treated with a combination of invasive airway pressure release ventilation (APRV) and a prone positioning regimen. After drastic improvement of respiratory function, the patient was discharged from intensive care unit after 10 days and from hospital after 14 days.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusion\u003c/strong\u003e: NPPE is a rare, but relevant complication of anesthesia and laryngospasm. The disease can basically occur in any patient undergoing general anesthesia and therefore should be considered.\u003c/p\u003e","manuscriptTitle":"Case report severe postoperative negative pressure pulmonary edema","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-10-04 13:11:46","doi":"10.21203/rs.3.rs-5018334/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2024-10-11T06:33:36+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-10-10T20:00:57+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2024-10-09T08:34:14+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"169011837661108491820395510674729244043","date":"2024-10-02T17:16:22+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"133802800983717744159996186928482190135","date":"2024-09-30T10:15:44+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"280274511896287650493291112978944117981","date":"2024-09-30T02:07:18+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-09-30T01:48:25+00:00","index":"","fulltext":""},{"type":"editorInvited","content":"","date":"2024-09-09T06:27:16+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-09-03T06:15:10+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2024-09-03T06:14:25+00:00","index":"","fulltext":""},{"type":"submitted","content":"BMC Anesthesiology","date":"2024-09-02T12:29:46+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bmc-anesthesiology","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bane","sideBox":"Learn more about [BMC Anesthesiology](http://bmcanesthesiol.biomedcentral.com/)","snPcode":"","submissionUrl":"https://www.editorialmanager.com/bane","title":"BMC Anesthesiology","twitterHandle":"BMC_series","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"em","reportingPortfolio":"BMC Series","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"9e7878c6-9bd7-465e-9c37-6f558a2047b2","owner":[],"postedDate":"October 4th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"published-in-journal","subjectAreas":[],"tags":[],"updatedAt":"2024-11-04T16:21:37+00:00","versionOfRecord":{"articleIdentity":"rs-5018334","link":"https://doi.org/10.1186/s12871-024-02785-2","journal":{"identity":"bmc-anesthesiology","isVorOnly":false,"title":"BMC Anesthesiology"},"publishedOn":"2024-11-01 15:56:55","publishedOnDateReadable":"November 1st, 2024"},"versionCreatedAt":"2024-10-04 13:11:46","video":"","vorDoi":"10.1186/s12871-024-02785-2","vorDoiUrl":"https://doi.org/10.1186/s12871-024-02785-2","workflowStages":[]},"version":"v1","identity":"rs-5018334","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-5018334","identity":"rs-5018334","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}