Post-operative airway obstruction in Noonan syndrome: An unusual presentation
Identifieur interne : 000186 ( Pmc/Corpus ); précédent : 000185; suivant : 000187Post-operative airway obstruction in Noonan syndrome: An unusual presentation
Auteurs : Sheetal R. Jagtap ; Hemalata R. Iyer ; Rochana G. Bakhshi ; Hemant N. LahotiSource :
- Indian Journal of Anaesthesia [ 0019-5049 ] ; 2015.
Url:
DOI: 10.4103/0019-5049.160961
PubMed: 26257420
PubMed Central: 4523968
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Post-operative airway obstruction in Noonan syndrome: An unusual presentation</title><author><name sortKey="Jagtap, Sheetal R" sort="Jagtap, Sheetal R" uniqKey="Jagtap S" first="Sheetal R" last="Jagtap">Sheetal R. Jagtap</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Iyer, Hemalata R" sort="Iyer, Hemalata R" uniqKey="Iyer H" first="Hemalata R" last="Iyer">Hemalata R. Iyer</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Bakhshi, Rochana G" sort="Bakhshi, Rochana G" uniqKey="Bakhshi R" first="Rochana G" last="Bakhshi">Rochana G. Bakhshi</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Lahoti, Hemant N" sort="Lahoti, Hemant N" uniqKey="Lahoti H" first="Hemant N" last="Lahoti">Hemant N. Lahoti</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">26257420</idno><idno type="pmc">4523968</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4523968</idno><idno type="RBID">PMC:4523968</idno><idno type="doi">10.4103/0019-5049.160961</idno><date when="2015">2015</date><idno type="wicri:Area/Pmc/Corpus">000186</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000186</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Post-operative airway obstruction in Noonan syndrome: An unusual presentation</title><author><name sortKey="Jagtap, Sheetal R" sort="Jagtap, Sheetal R" uniqKey="Jagtap S" first="Sheetal R" last="Jagtap">Sheetal R. Jagtap</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Iyer, Hemalata R" sort="Iyer, Hemalata R" uniqKey="Iyer H" first="Hemalata R" last="Iyer">Hemalata R. Iyer</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Bakhshi, Rochana G" sort="Bakhshi, Rochana G" uniqKey="Bakhshi R" first="Rochana G" last="Bakhshi">Rochana G. Bakhshi</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author><author><name sortKey="Lahoti, Hemant N" sort="Lahoti, Hemant N" uniqKey="Lahoti H" first="Hemant N" last="Lahoti">Hemant N. Lahoti</name><affiliation><nlm:aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</nlm:aff></affiliation></author></analytic><series><title level="j">Indian Journal of Anaesthesia</title><idno type="ISSN">0019-5049</idno><idno type="eISSN">0976-2817</idno><imprint><date when="2015">2015</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Van Der Burgt, I" uniqKey="Van Der Burgt I">I van der Burgt</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hernandez, Rj" uniqKey="Hernandez R">RJ Hernandez</name></author><author><name sortKey="Stern, Am" uniqKey="Stern A">AM Stern</name></author><author><name sortKey="Rosenthal, A" uniqKey="Rosenthal A">A Rosenthal</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Samra, T" uniqKey="Samra T">T Samra</name></author><author><name sortKey="Banerjee, N" uniqKey="Banerjee N">N Banerjee</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Brogan, Tv" uniqKey="Brogan T">TV Brogan</name></author><author><name sortKey="Finn, Ls" uniqKey="Finn L">LS Finn</name></author><author><name sortKey="Pyskaty, Dj" uniqKey="Pyskaty D">DJ Pyskaty</name></author><author><name sortKey="Redding, Gj" uniqKey="Redding G">GJ Redding</name></author><author><name sortKey="Ricker, D" uniqKey="Ricker D">D Ricker</name></author><author><name sortKey="Inglis, A" uniqKey="Inglis A">A Inglis</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Do, Tb" uniqKey="Do T">TB Do</name></author><author><name sortKey="Chu, Jm" uniqKey="Chu J">JM Chu</name></author><author><name sortKey="Berdjis, F" uniqKey="Berdjis F">F Berdjis</name></author><author><name sortKey="Anas, Ng" uniqKey="Anas N">NG Anas</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Shah, Ss" uniqKey="Shah S">SS Shah</name></author><author><name sortKey="Drinkwater, Dc" uniqKey="Drinkwater D">DC Drinkwater</name></author><author><name sortKey="Christian, Kg" uniqKey="Christian K">KG Christian</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Eberlein, Mh" uniqKey="Eberlein M">MH Eberlein</name></author><author><name sortKey="Drummond, Mb" uniqKey="Drummond M">MB Drummond</name></author><author><name sortKey="Haponik, Ef" uniqKey="Haponik E">EF Haponik</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Pawar, Ss" uniqKey="Pawar S">SS Pawar</name></author><author><name sortKey="Chun, Rh" uniqKey="Chun R">RH Chun</name></author><author><name sortKey="Rao, Ar" uniqKey="Rao A">AR Rao</name></author><author><name sortKey="Kerschner, Je" uniqKey="Kerschner J">JE Kerschner</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Seear, M" uniqKey="Seear M">M Seear</name></author><author><name sortKey="Hui, H" uniqKey="Hui H">H Hui</name></author><author><name sortKey="Magee, F" uniqKey="Magee F">F Magee</name></author><author><name sortKey="Bohn, D" uniqKey="Bohn D">D Bohn</name></author><author><name sortKey="Cutz, E" uniqKey="Cutz E">E Cutz</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Indian J Anaesth</journal-id><journal-id journal-id-type="iso-abbrev">Indian J Anaesth</journal-id><journal-id journal-id-type="publisher-id">IJA</journal-id><journal-title-group><journal-title>Indian Journal of Anaesthesia</journal-title></journal-title-group><issn pub-type="ppub">0019-5049</issn><issn pub-type="epub">0976-2817</issn><publisher><publisher-name>Medknow Publications & Media Pvt Ltd</publisher-name><publisher-loc>India</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">26257420</article-id><article-id pub-id-type="pmc">4523968</article-id><article-id pub-id-type="publisher-id">IJA-59-442</article-id><article-id pub-id-type="doi">10.4103/0019-5049.160961</article-id><article-categories><subj-group subj-group-type="heading"><subject>Brief Communication</subject></subj-group></article-categories><title-group><article-title>Post-operative airway obstruction in Noonan syndrome: An unusual presentation</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Jagtap</surname><given-names>Sheetal R</given-names></name><xref ref-type="aff" rid="aff1"></xref><xref ref-type="corresp" rid="cor1"></xref></contrib><contrib contrib-type="author"><name><surname>Iyer</surname><given-names>Hemalata R</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Bakhshi</surname><given-names>Rochana G</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Lahoti</surname><given-names>Hemant N</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib></contrib-group><aff id="aff1">Department of Anaesthesiology and Paediatric Surgery, Pad. Dr. D. Y. Patil Medical College, Hospital and Research Centre, Mumbai, Maharashtra, India</aff><author-notes><corresp id="cor1"><bold>Address for correspondence:</bold> Dr. Sheetal R Jagtap, Department of Anaesthesiology and Paediatric Surgery, 504, Sayia CHS, Plot E/01, Sector 12, Kharghar, Mumbai - 410 210, Maharashtra, India. E-mail: <email xlink:href="drsheetaljagtap@gmail.com">drsheetaljagtap@gmail.com</email></corresp></author-notes><pub-date pub-type="ppub"><month>7</month><year>2015</year></pub-date><volume>59</volume><issue>7</issue><fpage>442</fpage><lpage>444</lpage><permissions><copyright-statement>Copyright: © Indian Journal of Anaesthesia</copyright-statement><copyright-year>2015</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions></article-meta></front><body><sec sec-type="intro" id="sec1-1"><title>INTRODUCTION</title><p>Noonan syndrome (NS) is an autosomal dominant disorder with craniofacial, cardiac, skeletal abnormalities, haemorrhagic diathesis, with reported incidence of 1 in 1000 to 1 in 2500 live births.[<xref rid="ref1" ref-type="bibr">1</xref>] Various lymphatic abnormalities have been observed in these patients including pulmonary and intestinal lymphangiectasia and lymphoedema.[<xref rid="ref2" ref-type="bibr">2</xref>] Craniofacial dysmorphism webbed neck and atlantoaxial instability can complicate airway management.[<xref rid="ref3" ref-type="bibr">3</xref>] The anaesthetic considerations include difficult airway, impaired cardio-pulmonary function, mental retardation, and short stature.</p><p>We report an unusual presentation of airway obstruction due to bronchial cast on 5<sup>th</sup> post-operative day in a case of NS, who underwent laparoscopic orchidopexy.</p></sec><sec id="sec1-2"><title>CASE REPORT</title><p>A 15-year-old male weighing 28 kg, 112 cm tall and diagnosed case of NS was posted for laparoscopic orchidopexy for bilateral undescended testes. He had no other complaints except undescended testes. Birth history was normal, but he had delayed milestones and low intelligence. He was short statured with short neck, high arched palate, adequate mouth opening, and Mallampati airway class II. His pulse rate (PR) was 70/min, blood pressure (BP) was 90/60 mm Hg. There was a systolic murmur in the pulmonary area and two-dimensional echo showed pulmonary stenosis with pulmonary artery systolic pressure of 40 mm Hg. Chest X-ray (CXR) showed prominent broncho-vascular markings. Coagulation profile and biochemical investigations were within normal limits.</p><p>During laparoscopic orchidopexy, there was a major vascular injury, which was repaired and blood loss was adequately replaced. The rest of the intraoperative course was uneventful. Patient was extubated and shifted to Intensive Care Unit (ICU). As ICU stay was unremarkable, he was moved to wards on 2<sup>nd</sup> post-operative day.</p><p>On 5<sup>th</sup> post-operative day, the patient suddenly developed fever with breathlessness. Breath sounds were absent on left side. CXR showed homogenous opacity in left hemi thorax with collapsed lung. He was shifted back to ICU. He was conscious, oriented with a PR of 90/min, BP of 100/60 mm Hg, respiratory rate of 35/min, and oxygen saturation (SpO<sub>2</sub>) fluctuating between 85% and 90% on room air. Patient was put on humidified oxygen 4 L/min and ipratropium and budesonide nebulisation. The cause of this sudden, event was thought to be thick mucoid secretions or aspiration of foreign body. Patient was taken up for rigid bronchoscopy.SpO<sub>2</sub> was 88–90% on oxygen, HR 92/min, and BP 98/62 mm Hg. Patient was pre-medicated with intravenous (IV) glycopyrrolate (4 μgm/kg), and induced with ketamine (2 mg/kg), sevoflurane and maintained on atracurium (0.5 mg/kg), and sevoflurane with the help of ventilating bronchoscope. On bronchoscopy, a tissue mass lining the left main bronchus was visualised. It was difficult to retrieve en masse even after multiple attempts with saline instillation. Intra-operative SpO<sub>2</sub> kept on fluctuating. Patient was then intubated with 5.5 mm cuffed oral endotracheal tube. End-tidal carbon dioxide (EtCO<sub>2</sub>) was 50–55 mm Hg and SpO<sub>2</sub>, 85–90%. Endotracheal suctioning with saline instillation brought out fibrinous material. Active physiotherapy was given. Following this, the saturation improved to 93–95% with EtCO<sub>2</sub> of 30 mm Hg. Keeping equipment ready for re-intubation, the neuromuscular block was reversed, and the patient was extubated. Immediate post-extubation, a chunk of tissue like material was coughed out by the patient<xref ref-type="fig" rid="F1">Figure 1</xref>. It was rubbery in consistency and on spreading out, took the shape of the bronchial tree <xref ref-type="fig" rid="F2">Figure 2</xref>. There was a dramatic improvement in saturation (SpO<sub>2</sub> 98–99%) and air entry. Patient was put on ipratropium and budesonide nebulisation 6 hourly, which was continued for the next 48 h with IV antibiotic cover. Post-operative CXR showed significant improvement. The rest of the post-operative stay was uneventful. Patient was discharged on day 3 after bronchoscopy. On subsequent follow-up, the patient was asymptomatic. Histopathology report of the tissue mass revealed neutrophils, alveolar macrophages, fibrinous and mucinous material, diagnosed as a bronchial cast.</p><fig id="F1" position="float"><label>Figure 1</label><caption><p>Coughed out tissue</p></caption><graphic xlink:href="IJA-59-442-g001"></graphic></fig><fig id="F2" position="float"><label>Figure 2</label><caption><p>Spread out bronchial cast</p></caption><graphic xlink:href="IJA-59-442-g002"></graphic></fig></sec><sec sec-type="discussion" id="sec1-3"><title>DISCUSSION</title><p>Lymphatic abnormality seems to be inherent in patients with NS. About 20% of NS patients may have lymphatic vessel dysplasia, hypoplasia or aplasia. This may cause generalised or peripheral lymphoedema, pulmonary, and intestinal lymphangiectasia.[<xref rid="ref2" ref-type="bibr">2</xref>] Borgan <italic>et al</italic>. reported bronchial cast formation in NS following surgery for Tetralogy of Fallot.[<xref rid="ref4" ref-type="bibr">4</xref>] In the present case, no thoracic intervention was done and the patient had no previous history of cough or respiratory difficulty. In retrospect, the reason for the cast formation could be attributed to elevated pulmonary arterial pressure. A literature search revealed that bronchial casts are formed in a condition called plastic bronchitis (PB). PB is a rare condition described more commonly in children than adults. It has been reported following surgeries for congenital heart disease, especially Fontan's procedure, asthma, cystic fibrosis, aspergillosis, and smoke inhalation.[<xref rid="ref4" ref-type="bibr">4</xref>] Due to acute and vague presentation, these patients may have a mortality rate as high as 29%.[<xref rid="ref4" ref-type="bibr">4</xref><xref rid="ref5" ref-type="bibr">5</xref>] The cause is usually asphyxiation due to airway obstruction. The composition of these casts is consistent with lymphatic fluid. Probably high pulmonary or intrathoracic lymphatic pressure and/or presence of undetected lymphobronchial fistulae may contribute to the formation of these casts.[<xref rid="ref6" ref-type="bibr">6</xref>] The most common clinical presentations include respiratory distress and coughing up of material like ‘chicken meat’ or ‘noodles’.[<xref rid="ref7" ref-type="bibr">7</xref>] Recurrence of the cast with repeated bronchoscopies has been reported.[<xref rid="ref8" ref-type="bibr">8</xref>]</p><p>Seear <italic>et al</italic>. classified these casts into type I (cellular) and type II (acellular).[<xref rid="ref9" ref-type="bibr">9</xref>] The casts are cohesive in nature and take the shape of bronchi in which they are formed. They are adherent but friable. They may be spontaneously expectorated or may require urgent bronchoscopy. Flexible bronchoscopy may help in diagnosis but rigid bronchoscopy is required for removal of the cast with the use of forceps during bronchoscopy. Irrigation with normal saline causes these casts to swell and thus easier to grasp. We irrigated with saline but still could not retrieve it and ultimately patient coughed it out after extubation.</p><p>In addition to bronchoscopy, adjuvant medical treatment with mucolytics, macrolide antibiotics, and tissue plasminogen activator have been used for cast disruption.[<xref rid="ref4" ref-type="bibr">4</xref>] Chest physiotherapy with steroid and saline nebulisation is frequently employed as an adjunct for cast mobilisation.</p></sec><sec sec-type="conclusions" id="sec1-4"><title>CONCLUSION</title><p>Awareness and ability to diagnose PB leading to airway obstruction in patients with NS are crucial to reduce morbidity and mortality with prompt and effective treatment.</p></sec></body><back><ack><title>ACKNOWLEDGEMENTS</title><p>Authors acknowledge support from Dr. Shilpa V. 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