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From the Département d’anesthésie-réanimation, Centre hospitalier affilié universitaire de Québec, Université Laval, Québec, Québec, Canada.
Address correspondence to: Dr. Hélène G. Pellerin, Département d’anesthésie-réanimation, CHA (Hôpital de l’Enfant-Jésus), 1401, 18e rue, Québec, Québec G1J 1Z4, Canada. Phone: 418-649-5807; Fax: 418-649-5918; E-mail: helenpellerin{at}gmail.com
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Abstract |
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Methods: Medical records of all OPMD patients who had either LPR or CPM between 1997 and 2002 were reviewed. The following complications were collected: death, pneumonia, aspiration pneumonitis, airway obstruction, reintubation, and severe coughing and choking caused by profuse secretions.
Results: One hundred and fourteen surgeries on 92 OPMD patients were studied. Fifty-nine were LPR under general anesthesia (LPR-GA) and 22 were LPR under local anesthesia (LPR-LA). Thirty-three surgeries were CPM, all under general anesthesia. There was no death or reintubation. Patients who had LPR-LA had shorter postanesthesia care unit (P < 0.001) and ambulatory surgery unit (P < 0.001) stays than those who had LPR-GA. Complications were more frequent after CPM than LPR-GA (P < 0.001).
Conclusion: The complication rate after LPR was low. Both local and general anesthesia are safe alternatives in OPMD patients operated for LPR. Patients having CPM presented more respiratory complications than those having LPR.
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Introduction |
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The diagnosis of OPMD is usually made on clinical findings: a family history and the successive appearance of bilateral eyelid ptosis and dysphagia, usually between 50 and 60 yr of age. Oculopharyngeal muscular dystrophy is a progressive disease for which no curative treatment is known. Two types of surgery are often done to alleviate the symptoms of OPMD: levator palpebrae resection (LPR) for correction of bilateral ptosis and cricopharyngeal myotomy (CPM) to improve dysphagia.
Only a few publications about the anesthetic considerations for OPMD patients can be found in the literature and none about perioperative risks. The objectives of this retrospective chart review were to describe the postoperative complications of OPMD patients undergoing LPR or CPM and to determine their incidence.
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Methods |
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Comparisons were made between LPR done under local anesthesia (LPR-LA) with those done under general anesthesia (LPR-GA) and between LPR-GA and CPM, which were all done under general anesthesia. Data are presented as mean with SD or proportions. Statistical analysis was done with the Student’s t test or the Welch’s test for continuous variables and the Fisher’s exact test for proportions. Ninety-five percent confidence intervals (CI95%) were calculated with respect to the incidence of patients presenting complications following LPR or CPM. P < 0.05 was considered significant.
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Results |
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). Six patients had LPR prior to CPM while seven others had CPM prior to LPR. All LPR-LA and 51 LPR-GA were done on an outpatient basis while eight LPR-GA and all CPM required hospitalization. One patient had both surgeries on the same day and was considered and analyzed as a CPM.
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. Compared to patients having LPR-GA, patients undergoing CPM were older (P < 0.001), thinner (P < 0.001), had a higher percentage of women (P < 0.005) and a higher incidence of preoperative dysphagia (P < 0.001). Duration of surgery was comparable for LPR-GA and LPR-LA (P = 0.43), and for LPR-GA and CPM (P = 0.71). However, patients undergoing CPM had a longer delay before extubation at the end of surgery (P < 0.01) and had a longer stay in the postanesthesia care unit (PACU) (P < 0.001) than LPR-GA patients. Patients undergoing LPR-GA had a longer stay in the PACU and in the ambulatory surgery unit (ASU) than LPR-LA patients (P < 0.001). Eight LPR-GA patients required an overnight hospital stay, while all CPM patients had a hospital stay averaging 3.3 ± 2.1 days.
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. No complication was observed in patients having LPR-LA (CI95%: 0.0%–14.9%). In the LPR-GA group, there were five complications occurring after four surgical procedures (6.7%, CI95%: 2.0%–16.0%). In the CPM group, 18 complications occurred following 12 surgical procedures (36.4%, CI95%: 20.0%–55.0%). There was no difference in the incidence of postoperative complications between LPR-LA and LPR-GA (P = 0.57), but CPM was associated with more complications than LPR-GA (P < 0.001). The most frequent complication was severe coughing and choking caused by profuse secretions, occurring both in the PACU and on the ward. There was no death or reintubation.
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Discussion |
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Although there are cases of OPMD reported worldwide, the prevalence of OPMD in the province of Quebec (Canada) is estimated at 1:1000, among the highest in the world.5 In the 17th century, three French sisters with OPMD immigrated to the province of Quebec. All three had a lot of descendants thus spreading the disease in the French Canadian population. The area of L’Islet-Montmagny, on the south shore of the St. Lawrence River East of Quebec City, is where this founder effect is the most evident. Similar clusters of OPMD have also been reported in Israel, and New Mexico.6,7
Oculopharyngeal muscular dystrophy patients have a normal life expectancy. The degree of dysphagia is the main factor determining prognosis. Dysphagia is caused by progressive weakness of the esophagopharyngeal muscles. These muscles help to propel the food bolus down the esophagus. Frequent choking when swallowing increases the duration of mealtime, eventually leading to malnutrition. Respiratory problems, such as aspiration pneumonitis, also have a negative influence on the quality of life of these patients. Cricopharyngeal myotomy becomes necessary when dysphagia is accompanied by marked weight loss, severe choking, or recurrent pneumonia.8,9 The eyelid ptosis has to be corrected when it interferes with vision or when cervical pain appears secondary to constant dorsiflexion of the neck.10
The literature on the anesthesia management of OPMD patients is scarce. Landrum et al. and Chun et al. have both described their anesthetic management of one OPMD patient, reporting the safe use of vecuronium and mivacurium respectively, with no postoperative complication.11,12 Christopher et al. have reported a case of difficult postoperative extubation in a patient with OPMD.13 The patient presented airway obstruction and aspiration pneumonitis, failed two extubation attempts and finally required a tracheostomy. In a prospective pharmacodynamic study in OPMD patients, Caron et al. reported a delayed onset of action with a normal duration of action of cisatracurium.14
The retrospective nature of our study carries some limitations, the most obvious being the dependence upon the quality of the data recorded in the medical records. Furthermore, a selection bias cannot be excluded and for these reasons the true incidence of postoperative complications may have been underestimated. It could also be argued that severe coughing and choking should not be considered as a complication. Although it did not result in long term consequences, many patients experienced drop in oxygen saturation during these spells, and it was a major factor in their prolonged PACU and hospital stays. There is also a possibility of a type 2 error when the incidence of complications between LPR-LA and LPR-GA were compared.
This study shows that some anesthetic considerations should be kept in mind for patients with OPMD. Respiratory complications are frequent in the postoperative period of OPMD patients undergoing CPM and these patients should be carefully monitored. Although we did not have access to data on the severity of dysphagia, it is most likely that patients with severe dysphagia and an important weight loss have a higher risk of postoperative complications. However OPMD patients can be treated safely on an outpatient basis after general anesthesia for minor procedures such as LPR. Two patients who had received rocuronium presented airway obstruction in the PACU which was relieved by administration of reversal agents. However, we did not find any case of prolonged postoperative weakness or prolongation in the time for extubation in patients who received muscle relaxants. It remains unclear whether OPMD patients have an increased risk of complications with muscle relaxants since Caron et al. and Plaud et al. have reported normal duration of action for cisatracurium and rocuronium respectively.14,15 Obviously, recovery of neuromuscular function must be carefully assessed at the end of surgery. Oculopharyngeal muscular dystrophy patients have difficulty swallowing and retain an increased amount of pharyngeal secretions which should not be mistaken for gastroesophageal reflux. Therefore the anesthesiologist should carefully aspirate pharyngeal secretions before anesthesia induction, but indications for rapid sequence induction should not be different than in the normal surgical population.
In conclusion, in patients with OPMD, general and local anesthesia are safe alternatives for LPR but local anesthesia allows shorter PACU and ASU stays. Cricopharyngeal myotomy is associated with a significant risk of postoperative complications mostly affecting the respiratory system and should be planned and managed accordingly.
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Footnotes |
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Presented in part at the 61st Annual Meeting of the Canadian Anesthesiologists’ Society, Vancouver, BC, June 17–21, 2005.
Accepted for publication January 16, 2007. Revision accepted February 1, 2007.
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References |
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2 Amyot R. Hereditary, familial and acquired ptosis of late-onset. Can Med Assoc J 1948; 59: 434–8.
3 Victor M, Hayes R, Adams RD. Oculopharyngeal muscular dystrophy. A familial disease of late life characterized by dysphagia and progressive ptosis of the eyelids. N Engl J Med 1962; 267: 1267–72.[Medline]
4 Bouchard JP. André Barbeau and the oculopharyngeal muscular dystrophy in French Canada and North America. Neuromuscul Disord 1997; 7(Suppl 1): S5–11.[Medline]
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6 Blumen SC, Nisipeanu P, Sadeh M, et al. Epidemiology and inheritance of oculopharyngeal muscular dystrophy in Israel. Neuromuscul Disord 1997; 7(Suppl 1): S38–40.[Medline]
7 Becher MW, Morrison L, Davis LE, et al. Oculopharyngeal muscular dystrophy in Hispanic New Mexicans. JAMA 2001; 286: 2437–40.
8 Duranceau AC, Beauchamp G, Jamieson GG, Barbeau A. Oropharyngeal dysphagia and oculopharyngeal muscular dystrophy. Surg Clin North Am 1983; 63: 825–32.[Medline]
9 Fradet G, Pouliot D, Robichaud R, St-Pierre S, Bouchard JP. Upper esophageal sphincter myotomy in oculopharyngeal muscular dystrophy: long-term clinical results. Neuromuscul Disord 1997; 7(Suppl 1): S90–5.[Medline]
10 Rodrigue D, Molgat YM. Surgical correction of blepharoptosis in oculopharyngeal muscular dystrophy. Neuromuscul Disord 1997; 7(Suppl 1): S82–4.[Medline]
11 Landrum AL, Eggers GW. Oculopharyngeal dystrophy : an approach to anesthetic management. Anesth Analg 1992; 75: 1043–5.
12 Chun R, Ananthanarayan C. Safe use of mivacurium in oculopharyngeal muscular dystrophy. Can J Anaesth 1998; 45: 1134.[Medline]
13 Christopher K, Horkan C, Patterson RB, Yodice PC. Oculopharyngeal muscular dystrophy complicating airway management. Chest 2001; 120: 2101–3.[Medline]
14 Caron MJ, Girard F, Girard DC, et al. Cisatracurium pharmacodynamics in patients with oculopharyngeal muscular dystrophy. Anesth Analg 2005; 100: 393–7.
15 Plaud B, Duranceau A, Donati F. Dystrophie oculopharyngée (DOP) et blocage neuromusculaire avec le rocuronium. Ann Fr Anesth Réanim 2003; 22: S195 (abstract).
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