Case report
Anesthesia for bariatric surgery in a patient with Prader–Willi syndrome: Case report
Anestesia para cirugía bariátrica en paciente con síndrome de Prader-Willy: reporte de un caso
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In 1956, Prader, Labhart and Willi described nine other cases and gave the syndrome its name. In 1980, Ledbetter discovered the existence of a microdeletion of the 15q11-q13 region and three years later, Butler and Nichols observed this genomic imprinting in patients with Prader–Willi syndrome.<a class="elsevierStyleCrossRef" href="#bib0160"><span class="elsevierStyleSup">2</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">There are few reported cases in the literature on the anesthetic management of adult patients with Prader–Willi syndrome with low life expectancy as a consequence of morbid obesity.<a class="elsevierStyleCrossRefs" href="#bib0165"><span class="elsevierStyleSup">3–5</span></a> Furthermore, these patients are predisposed to sudden death from respiratory diseases or in the postoperative period.<a class="elsevierStyleCrossRefs" href="#bib0180"><span class="elsevierStyleSup">6–10</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the anesthetic management of a 31-year-old patient diagnosed with Prader–Willi syndrome who was scheduled to undergo bariatric surgery.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case description</span><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was admitted to surgery at the Manuel Uribe Ángel Hospital in Envigado, Antioquia, Colombia. The patient was 31 years old and female with a background of Prader–Willi syndrome associated with hypertension, extreme obesity (BMI 54), mental retardation, hypothyroidism and obstructive sleep apnea syndrome. She was admitted to undergo a gastric bypass.</p><p id="par0030" class="elsevierStylePara elsevierViewall">The patient is considered to be a NYHA functional class III, ASA 4.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The physical exam revealed central cyanosis and blood pressure of 140/80<span class="elsevierStyleHsp" style=""></span>mmHg, heart rate of 68<span class="elsevierStyleHsp" style=""></span>bpm, respiratory rate of 19 breaths/min, Mallampati score of II, oral aperture above 4<span class="elsevierStyleHsp" style=""></span>cm, cervical perimeter below 40<span class="elsevierStyleHsp" style=""></span>cm, 35° extension of the upper neck and thyromental distance above 6<span class="elsevierStyleHsp" style=""></span>cm.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Rhythmic cardiac sounds were auscultated, revealing P2<span class="elsevierStyleHsp" style=""></span>><span class="elsevierStyleHsp" style=""></span>A2 along with a protomesosystolic murmur at the pulmonary and tricuspid focuses, III/VI. There was a diminished bilateral vesicular murmur.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Oxygen saturation oscillated between 80 and 90% through the nasal canal at 3<span class="elsevierStyleHsp" style=""></span>l/min.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The patient did not resist any of the examiner's maneuvers and collaborated during the whole evaluation.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Presurgical exams revealed: hemoglobin 12.2<span class="elsevierStyleHsp" style=""></span>g/dl, hematocrite 41%, 371<span class="elsevierStyleHsp" style=""></span>000<span class="elsevierStyleHsp" style=""></span>platelets/mL, TSH 7.92<span class="elsevierStyleHsp" style=""></span>mIU/L, HA1G 6.75%, arterial blood gases (FiO<span class="elsevierStyleInf">2</span>) 0.21: pH 7.38, PCO<span class="elsevierStyleInf">2</span> 54<span class="elsevierStyleHsp" style=""></span>mmHg, PO<span class="elsevierStyleInf">2</span> 25<span class="elsevierStyleHsp" style=""></span>mmHg, HCO<span class="elsevierStyleInf">3</span> 29<span class="elsevierStyleHsp" style=""></span>mEq/l. Echocardiogram (TTE): FE: 60%, PSAP 52<span class="elsevierStyleHsp" style=""></span>mmHg. There were slightly insufficient tricuspid and pulmonary valves, but normal cavity size.</p><p id="par0060" class="elsevierStylePara elsevierViewall">In the operating room, a 16G cannula was placed on 2 cephalic veins; a rapid sequence induction was performed with previous oxygenation during 5<span class="elsevierStyleHsp" style=""></span>min until a maximum saturation of 95% was reached, followed by a 100<span class="elsevierStyleHsp" style=""></span>mcg bolus intravenous administration of remifentanil, 60<span class="elsevierStyleHsp" style=""></span>mg lidocaine, 150<span class="elsevierStyleHsp" style=""></span>mg propofol, 100<span class="elsevierStyleHsp" style=""></span>mg succinylcholine, laryngoscopy with valve curve #3 (Cormack II visualization), introduction of 8.0<span class="elsevierStyleHsp" style=""></span>mm orotracheal tube, maintenance with 3% sevoflurane, flow of oxygen gases at 0.3<span class="elsevierStyleHsp" style=""></span>l/min and air at 0.5<span class="elsevierStyleHsp" style=""></span>l/min, and finally an intravenous infusion of remifentanil at 0.1<span class="elsevierStyleHsp" style=""></span>mcg/kg/min.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The anesthesia machine was programmed for volume, with a tidal volume of 7<span class="elsevierStyleHsp" style=""></span>ml/kg, PEEP 5<span class="elsevierStyleHsp" style=""></span>cm H<span class="elsevierStyleInf">2</span>O, showing peak pressures higher than 35<span class="elsevierStyleHsp" style=""></span>cm H<span class="elsevierStyleInf">2</span>O with plateau pressures lower than 30<span class="elsevierStyleHsp" style=""></span>cm H<span class="elsevierStyleInf">2</span>O. The bilateral position of the orotracheal tube was immediately verified through auscultation to rule out obstruction or kinking. The monitor showed a progressive increase in the level of CO<span class="elsevierStyleInf">2</span> at the end of the expiration without presenting inclination in the curve of the capnogram in phase 2. 6<span class="elsevierStyleHsp" style=""></span>mg cisatracurium was administered intravenously, with significant improvement in the respective values.</p><p id="par0070" class="elsevierStylePara elsevierViewall">During the intraoperative period the surgical team identified severe cardiomegaly associated with hepatomegaly, which led to the decision to perform gastric sleeve surgery to then proceed with a bypass in the second surgical period.</p><p id="par0075" class="elsevierStylePara elsevierViewall">At the end of the procedure, 0.6<span class="elsevierStyleHsp" style=""></span>mg of hydromorphone was administered intravenously.</p><p id="par0080" class="elsevierStylePara elsevierViewall">There were no episodes of desaturation, severe hypotension/hypertension or arrhythmias. She was extubated after finishing surgery and transferred to the special care unit and supplemented with 50% oxygen at 10<span class="elsevierStyleHsp" style=""></span>l/min with the Ventury system. No reintubation was required.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0085" class="elsevierStylePara elsevierViewall">Prader–Willi syndrome is considered to be the leading cause of obesity associated with genetic syndromes and has a prevalence of approximately 1/25000.<a class="elsevierStyleCrossRef" href="#bib0205"><span class="elsevierStyleSup">11</span></a> The partial deletion of the long arm of the paternal chromosome 15 is a common marker of this disease,<a class="elsevierStyleCrossRef" href="#bib0210"><span class="elsevierStyleSup">12</span></a> though it can also present as a maternal uniparental disomy.<a class="elsevierStyleCrossRef" href="#bib0215"><span class="elsevierStyleSup">13</span></a></p><p id="par0090" class="elsevierStylePara elsevierViewall">The annual mortality rate was 3% for all ages, but the rate increases to 7% over the age of 30.<a class="elsevierStyleCrossRef" href="#bib0205"><span class="elsevierStyleSup">11</span></a> Patients generally suffer an early death due to secondary complications related to obesity, such as diabetes mellitus, hypertension, obstructive apnea-hypopnea, cardiovascular disease and respiratory failure.<a class="elsevierStyleCrossRef" href="#bib0220"><span class="elsevierStyleSup">14</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall">The clinical course for Prader–Willi syndrome is usually divided in two phases.<a class="elsevierStyleCrossRef" href="#bib0225"><span class="elsevierStyleSup">15</span></a> The first phase occurs during the neonatal and lactation periods and is characterized by marked hypotonia, difficulties with suction, persistent cough, crying and episodes of asphyxia.<a class="elsevierStyleCrossRefs" href="#bib0230"><span class="elsevierStyleSup">16,17</span></a> The second phase, between the ages of 2–5, is characterized by hypogonadism, mental retardation and obesity related to hyperphagia, most likely due to a hypothalamic defect in the satiety center<a class="elsevierStyleCrossRef" href="#bib0240"><span class="elsevierStyleSup">18</span></a> or an innate failure to metabolize lipids and carbohydrates.<a class="elsevierStyleCrossRef" href="#bib0245"><span class="elsevierStyleSup">19</span></a></p><p id="par0100" class="elsevierStylePara elsevierViewall">As was previously mentioned, there are few available reports on the anesthetic management of patients with Prader–Willi syndrome, and in the case at hand the patient who was schedule for a low-risk cardiovascular procedure was in her fourth decade of life with extreme obesity, obstructive sleep apnea syndrome and chronic hypoxia.</p><p id="par0105" class="elsevierStylePara elsevierViewall">Alterations in body temperature (hyperthermia and hypothermia), intraoperative arrhythmias, cor pulmonale and clinical consequences of obesity (decrease in functional residual capacity) are particular problems during the perioperative period.<a class="elsevierStyleCrossRefs" href="#bib0250"><span class="elsevierStyleSup">20–24</span></a></p><p id="par0110" class="elsevierStylePara elsevierViewall">During the intraoperative period the only difficulty with our patient was the elevated peak pressure values accompanied by normal plateau pressure levels. An elevated peak pressure can be accompanied by a concomitant elevation of plateau pressure, but when this association is not present, we are faced with an increase in the resistance of the airways (decrease of dynamic pulmonary distensibility), causing us to rule out bronchospasms, presence of secretions, obstruction, kinking or biting of the orotracheal tube.<a class="elsevierStyleCrossRef" href="#bib0275"><span class="elsevierStyleSup">25</span></a></p><p id="par0115" class="elsevierStylePara elsevierViewall">It should be noted that excessive intra-abdominal pressure decreases the pulmonary volumes in morbidly obese patients under sedation or anesthesia, notably in terms of residual functional capacity, while the alveolo-arterial oxygenation gradient is increased along with respiratory resistance.<a class="elsevierStyleCrossRef" href="#bib0280"><span class="elsevierStyleSup">26</span></a> Our patient's condition improved with the administration of a neuromuscular blocker. This technique is similar to that performed in patients with intra-abdominal hypertension syndrome, in which neuromuscular blockers are used to lower intra-abdominal and airway pressure.<a class="elsevierStyleCrossRef" href="#bib0285"><span class="elsevierStyleSup">27</span></a></p><p id="par0120" class="elsevierStylePara elsevierViewall">There was also no difficulty in performing the laryngoscopy, despite reports of difficult airways in these patients.<a class="elsevierStyleCrossRef" href="#bib0290"><span class="elsevierStyleSup">28</span></a></p><p id="par0125" class="elsevierStylePara elsevierViewall">A transfer to the special care unit was considered since these patients have an increased risk of postoperative hypoxia attributed to the altered response in consciousness level due to changes in oxygen and CO<span class="elsevierStyleInf">2</span> in blood pressure.<a class="elsevierStyleCrossRef" href="#bib0295"><span class="elsevierStyleSup">29</span></a></p><p id="par0130" class="elsevierStylePara elsevierViewall">Another issue is the documented presence of an oppositional and aggressive personality type associated with this syndrome,<a class="elsevierStyleCrossRef" href="#bib0300"><span class="elsevierStyleSup">30</span></a> although in our case the patient cooperated at all times during her hospital stay and did not require sedatives such as benzodiazepine which can possibly cause episodes of desaturation and apnea.</p><p id="par0135" class="elsevierStylePara elsevierViewall">In conclusion, the approach that an anesthesiologist takes with a Prader–Willi patient should include all the associated comorbidities, especially secondary ones like morbid obesity, taking into account the appropriate postoperative monitoring to avoid the onset of respiratory complications.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Funding</span><p id="par0140" class="elsevierStylePara elsevierViewall">Authors’ own resources.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflict of interest</span><p id="par0145" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres679500" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec685447" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres679501" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec685446" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case description" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interest" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2014-03-05" "fechaAceptado" => "2016-02-26" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec685447" "palabras" => array:5 [ 0 => "Anesthesia" 1 => "Obesity" 2 => "Bariatric surgery" 3 => "Sleep apnea, Obstructive" 4 => "Prader–Willi syndrome" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec685446" "palabras" => array:5 [ 0 => "Anestesia" 1 => "Obesidad" 2 => "Cirugía bariátrica" 3 => "Apnea del sueño obstructiva" 4 => "Síndrome de Prader Willi" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Prader–Willi syndrome is a genetic disorder characterized by hypotonia, obesity, short stature, mental retardation, hyperphagia, hypogonadism and low life expectancy.</p><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">We describe the case of a 31-year-old female patient with Prader–Willi syndrome scheduled for bariatric surgery. Anesthetic considerations are reviewed highlighting perioperative complications associated with this syndrome.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">El síndrome de Prader-Willi es un desorden genético caracterizado por hipotonía, obesidad, baja estatura, retraso mental, hiperfagia, hipogonadismo y expectativa de vida reducida.</p><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Describimos el caso de una paciente de 31 años con antecedente de síndrome de Prader -Willi, programada para realización de cirugía bariátrica. Se revisan las consideraciones anestésicas, haciendo énfasis en las complicaciones perioperatorias secundarias a este síndrome.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Rada-Ortega C, Gómez-Ramírez CF. Anestesia para cirugía bariátrica en paciente con síndrome de Prader-Willy: reporte de un caso. Rev Colomb Anestesiol. 2016;44:255–258.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:30 [ 0 => array:3 [ "identificador" => "bib0155" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Prader–Willi syndrome: consensus diagnostic criteria" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "V.A. Holm" 1 => "S.B. Cassidy" 2 => "M.G. Butler" 3 => "J.M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 4 | 2 | 6 |
| 2024 October | 14 | 4 | 18 |
| 2024 September | 22 | 2 | 24 |
| 2024 August | 19 | 5 | 24 |
| 2024 July | 22 | 5 | 27 |
| 2024 June | 27 | 3 | 30 |
| 2024 May | 29 | 4 | 33 |
| 2024 April | 36 | 5 | 41 |
| 2024 March | 57 | 5 | 62 |
| 2024 February | 18 | 8 | 26 |
| 2024 January | 20 | 1 | 21 |
| 2023 December | 28 | 11 | 39 |
| 2023 November | 16 | 4 | 20 |
| 2023 October | 24 | 8 | 32 |
| 2023 September | 12 | 2 | 14 |
| 2023 August | 25 | 8 | 33 |
| 2023 July | 10 | 4 | 14 |
| 2023 June | 28 | 3 | 31 |
| 2023 May | 37 | 4 | 41 |
| 2023 April | 32 | 2 | 34 |
| 2023 March | 18 | 4 | 22 |
| 2023 February | 13 | 17 | 30 |
| 2023 January | 11 | 20 | 31 |
| 2022 December | 9 | 14 | 23 |
| 2022 November | 30 | 6 | 36 |
| 2022 October | 10 | 10 | 20 |
| 2022 September | 23 | 8 | 31 |
| 2022 August | 15 | 3 | 18 |
| 2022 July | 8 | 6 | 14 |
| 2022 June | 7 | 10 | 17 |
| 2022 May | 13 | 10 | 23 |
| 2022 April | 15 | 9 | 24 |
| 2022 March | 19 | 10 | 29 |
| 2022 February | 15 | 5 | 20 |
| 2022 January | 15 | 10 | 25 |
| 2021 December | 12 | 4 | 16 |
| 2021 November | 17 | 6 | 23 |
| 2021 October | 7 | 13 | 20 |
| 2021 September | 11 | 6 | 17 |
| 2021 August | 11 | 4 | 15 |
| 2021 July | 17 | 5 | 22 |
| 2021 June | 8 | 5 | 13 |
| 2021 May | 8 | 5 | 13 |
| 2021 April | 34 | 12 | 46 |
| 2021 March | 10 | 4 | 14 |
| 2021 February | 4 | 7 | 11 |
| 2021 January | 5 | 4 | 9 |
| 2020 December | 5 | 6 | 11 |
| 2020 November | 6 | 5 | 11 |
| 2020 October | 5 | 4 | 9 |
| 2020 September | 4 | 4 | 8 |
| 2020 August | 7 | 6 | 13 |
| 2020 July | 8 | 9 | 17 |
| 2020 June | 1 | 1 | 2 |
| 2020 May | 2 | 7 | 9 |
| 2020 April | 4 | 3 | 7 |
| 2020 March | 5 | 1 | 6 |
| 2020 February | 5 | 6 | 11 |
| 2020 January | 5 | 3 | 8 |
| 2019 December | 7 | 10 | 17 |
| 2019 November | 3 | 3 | 6 |
| 2019 October | 0 | 0 | 0 |
| 2019 September | 1 | 5 | 6 |
| 2019 August | 0 | 1 | 1 |
| 2019 July | 5 | 12 | 17 |
| 2019 June | 0 | 1 | 1 |
| 2019 May | 5 | 13 | 18 |
| 2019 April | 1 | 0 | 1 |
| 2018 September | 1 | 0 | 1 |
| 2018 June | 3 | 5 | 8 |
| 2018 May | 23 | 7 | 30 |
| 2018 April | 31 | 11 | 42 |
| 2018 March | 26 | 12 | 38 |
| 2018 February | 19 | 9 | 28 |
| 2018 January | 25 | 7 | 32 |
| 2017 December | 17 | 7 | 24 |
| 2017 November | 8 | 7 | 15 |
| 2017 October | 18 | 12 | 30 |
| 2017 September | 21 | 11 | 32 |
| 2017 August | 16 | 8 | 24 |
| 2017 July | 15 | 3 | 18 |
| 2017 June | 25 | 8 | 33 |
| 2017 May | 18 | 16 | 34 |
| 2017 April | 31 | 16 | 47 |
| 2017 March | 19 | 11 | 30 |
| 2017 February | 9 | 6 | 15 |
| 2017 January | 12 | 8 | 20 |
| 2016 December | 35 | 14 | 49 |
| 2016 November | 21 | 13 | 34 |
| 2016 October | 40 | 15 | 55 |
| 2016 September | 68 | 16 | 84 |
| 2016 August | 61 | 11 | 72 |
| 2016 July | 37 | 9 | 46 |
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