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      Anesthesia Management of Vitrectomy in a Patient with Sturge-Weber Syndrome

      case-report

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          Abstract

          Sturge-Weber syndrome (SWS) is a neurocutaneous disorder, characterized by leptomeningeal angiomas involving the oral cavity, trachea, larynx, and face. Herein, we present a case of vitrectomy in a seven-year-old boy with SWS. The patient showed hemangioma on the left side of his face, as well as mental retardation and epilepsy. Preoperative examination revealed no apparent hemangioma in the oral cavity, pharynx, larynx, or trachea. However, he was predicted to have difficult airway intubation, as the oral cavity was smaller than the normal size. The minimum Mallampati score was 3–4 due to macroglossia. First, we applied awake intubation, but he failed to follow the commands. We proceeded to general anesthesia with propofol and did not use any muscle relaxants to maintain spontaneous breathing. A laryngeal mask airway was inserted to minimize any harm to possible oral angiomas. The patient was hemodynamically stable and extubated without any complications, such as bleeding or respiratory problems.

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          Sturge-Weber syndrome and port-wine stains caused by somatic mutation in GNAQ.

          Matthew Shirley, Hao Tang, Carol J Gallione … (2013)
          The Sturge-Weber syndrome is a sporadic congenital neurocutaneous disorder characterized by a port-wine stain affecting the skin in the distribution of the ophthalmic branch of the trigeminal nerve, abnormal capillary venous vessels in the leptomeninges of the brain and choroid, glaucoma, seizures, stroke, and intellectual disability. It has been hypothesized that somatic mosaic mutations disrupting vascular development cause both the Sturge-Weber syndrome and port-wine stains, and the severity and extent of presentation are determined by the developmental time point at which the mutations occurred. To date, no such mutation has been identified. We performed whole-genome sequencing of DNA from paired samples of visibly affected and normal tissue from 3 persons with the Sturge-Weber syndrome. We tested for the presence of a somatic mosaic mutation in 97 samples from 50 persons with the Sturge-Weber syndrome, a port-wine stain, or neither (controls), using amplicon sequencing and SNaPshot assays, and investigated the effects of the mutation on downstream signaling, using phosphorylation-specific antibodies for relevant effectors and a luciferase reporter assay. We identified a nonsynonymous single-nucleotide variant (c.548G→A, p.Arg183Gln) in GNAQ in samples of affected tissue from 88% of the participants (23 of 26) with the Sturge-Weber syndrome and from 92% of the participants (12 of 13) with apparently nonsyndromic port-wine stains, but not in any of the samples of affected tissue from 4 participants with an unrelated cerebrovascular malformation or in any of the samples from the 6 controls. The prevalence of the mutant allele in affected tissues ranged from 1.0 to 18.1%. Extracellular signal-regulated kinase activity was modestly increased during transgenic expression of mutant Gαq. The Sturge-Weber syndrome and port-wine stains are caused by a somatic activating mutation in GNAQ. This finding confirms a long-standing hypothesis. (Funded by the National Institutes of Health and Hunter's Dream for a Cure Foundation.).
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            Atelectasis in children undergoing either propofol infusion or positive pressure ventilation anesthesia for magnetic resonance imaging.

            Juergen Gieseke, G Lutterbey, H Schild … (2007)
            Atelectasis because of anesthesia is a recognized problem but may be affected by the anesthetic technique. We compared magnetic resonance images of atelectasis in children undergoing two types of anesthesia. Children requiring anesthesia for magnetic resonance imaging (MRI) had additional lung imaging sequences at the beginning and the end of anesthesia. Children had either i.v. propofol infusion (PI) without an artificial airway (n = 26) or positive pressure ventilation (PPV) via a tracheal tube (n = 20); the technique was chosen for clinical reasons. The extent of atelectasis was scored by two independent radiologists. The median ages (range) for PI and PPV groups were 45 months (1-77 months) and 18 months (2-74 months), respectively. The proportion of children with atelectasis was different in the first lung scan (42% vs 80%), but in the second scan atelectasis was seen frequently in both groups (82% vs 94%) with a greater extent in the PPV group. The atelectasis score was higher in young children, but all children had normal oxygen requirements and saturations. Many factors may influence the development of atelectasis but this study found less extensive atelectasis with PI than PPV. PI allows for sufficient motionlessness, required for high diagnostic image quality in pediatric MRI.
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              Tracheal intubation in ambulatory surgery patients: using remifentanil and propofol without muscle relaxants.

              Victor Stevens, Christine L. Wheatley (1997)
              Using alfentanil followed by an anesthetic induction dose of propofol provides adequate conditions for tracheal intubation without neuromuscular relaxants. Remifentanil, which has a clinical onset similar to that of alfentanil, has not been investigated for this indication. Accordingly, 80 ASA physical status I and II premedicated outpatients were randomly assigned to one of four groups (n = 20/group). Remifentanil 1, 2, 3, or 4 micrograms/kg (Groups I-IV, respectively) was infused intravenously over 90 s. Sixty seconds after beginning the remifentanil infusion, propofol 2 mg/kg was infused over 5 s. Ninety seconds after the administration of propofol, laryngoscopy and tracheal intubation were attempted and graded. Clinically acceptable intubating conditions (i.e., jaw relaxed, vocal cords open, and fewer than two coughs in response to intubation) were observed in 35%, 75%, 100%, and 95% of patients in Groups I-IV, respectively. Clinically acceptable intubating conditions were significantly (P < 0.05) less likely to occur in Group I compared with all other groups. Excellent intubating conditions (i.e., vocal cords open, no movement in response to intubation) were observed in 30%, 50%, 80%, 80% of patients in Groups I-IV, respectively. Overall conditions at intubation were significantly (P < 0.05) better in Groups III and IV compared with Groups I and II. The mean time to resumption of spontaneous ventilation after induction was < 5 min in all groups. No patient manifested clinically significant muscle rigidity. The mean arterial pressure decreased 16%, 20%, 28%, 26% immediately before tracheal intubation in Groups I-IV, respectively. No patient was treated for hypotension or bradycardia. In conclusion, healthy, premedicated patients with favorable airway anatomy can be reliably intubated with good or excellent conditions 90 s after the administration of remifentanil 3-4 micrograms/kg and propofol 2 mg/kg. Remifentanil 3 micrograms/kg and propofol 2 mg/kg co-administered intravenously may reliably provide adequate conditions for tracheal intubation in healthy patients without neuromuscular relaxants. This combination of drugs may allow the rapid return of spontaneous ventilation.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Tanaffos
                Journal ID (iso-abbrev): Tanaffos
                Journal ID (publisher-id): Tanaffos
                Journal ID (pmc): Tanaffos
                Title: Tanaffos
                Publisher: National Research Institute of Tuberculosis and Lung Disease
                ISSN (Print): 1735-0344
                ISSN (Electronic): 2345-3729
                Publication date (Print): January 2018
                Volume: 17
                Issue: 1
                Pages: 57-61
                Affiliations
                [1 ] Labbafinejad Medical Center, Department of Anesthesiology, Shahid Beheshti University of Medical Sciences, Tehran, Iran,
                [2 ] Department of Anesthesiology, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
                Author notes
                Correspondence to: Poor Zamany Nejat Kermany M, Address: Labbafinejad Medical Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran, Email address: drpoorzamany@ 123456yahoo.com
                Article
                Publisher ID: tanaffos-17-57
                PMC ID: 6087527
                SO-VID: dc3d82c9-b7ec-466c-b721-5c69db578f06
                Copyright © Copyright© 2018 National Research Institute of Tuberculosis and Lung Disease
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 13 May 2017
                Date accepted : 28 September 2017
                Categories
                Subject: Case Report

                Keywords: sturge-weber syndrome,propofol,difficult airway
                Data availability:
                Keywords: sturge-weber syndrome, propofol, difficult airway

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