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CASE REPORT
Year : 2023 | Volume
: 17
| Issue : 1 | Page : 94-96
Combined neuraxial-general anesthesia in opsoclonus–myoclonus syndrome: A case report
Joao B Abreu1, Catarina R Cordeiro2, Ana I Amorim1, Tiago G Catanho3, Karina D Gama1
1 Department of Anesthesiology, Hospital Central do Funchal, Funchal, Portugal
2 Department of Psychiatry Critical Care, Centro Hospitalar Universitário Lisboa Norte, Faculdade de Medicina da Universidade de Lisboa, Lisboa, Portugal
3 Department of Critical Care, Hospital Central do Funchal, Funchal, Portugal
Correspondence Address:
Joao B Abreu
Department of Anesthesia Hospital Central do Funchal Av. Luís de Camões 6180, 9000-177 Funchal
Portugal
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/sja.sja_558_22
| Date of Submission | 31-Jul-2022 |
| Date of Decision | 01-Aug-2022 |
| Date of Acceptance | 02-Aug-2022 |
| Date of Web Publication | 02-Jan-2023 |
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Opsoclonus-myoclonus syndrome (OMS) is a very rare neurological disorder thought to be the result of autoimmune responses in the nervous system. The relationship between this disorder and anesthesia procedures has not been studied in detail. To our knowledge, there are only 4 case reports, none of them with epidural-general combined anesthesia.
We present a 9-year-old female with OMS due to low-grade neuroblastoma, for 7 years, who underwent tumor remotion due to the large size. Intravenous induction was done with alfentanil, lidocaine, propofol and rocuronium and ropivacaine was administered via lumbar epidural catheter. Adding to the sparse anesthetic management information in OMS, we now show one more possible approach, that can be valuable in high-risk cases, where general anesthesia can be involved with higher risk for the patient.
Keywords: Anesthesia, care, Kinsbourne, neuroblastoma, operative, opsoclonus–myoclonus, pain, pediatrics, perioperative, post, regional, surgery, syndrome
How to cite this article:
Abreu JB, Cordeiro CR, Amorim AI, Catanho TG, Gama KD. Combined neuraxial-general anesthesia in opsoclonus–myoclonus syndrome: A case report. Saudi J Anaesth 2023;17:94-6 |
How to cite this URL:
Abreu JB, Cordeiro CR, Amorim AI, Catanho TG, Gama KD. Combined neuraxial-general anesthesia in opsoclonus–myoclonus syndrome: A case report. Saudi J Anaesth [serial online] 2023 [cited 2023 Mar 31];17:94-6. Available from: https://www.saudija.org/text.asp?2023/17/1/94/364874 |
Opsoclonus–myoclonus syndrome (OMS) is a condition that goes by the name of Kinsbourne syndrome or dancing-eye syndrome. This is a very rare neurological disorder that is thought to be the result of autoimmune responses in the nervous system.[1] Clinically, as the name suggests, it is characterized by opsoclonus (rapid, multidirectional, conjugate eye movements), myoclonus, ataxia, irritability, and sleep disturbance. It is a rare neuroimmunological disorder typically presenting in previously healthy infants and toddlers. About half of the cases are associated with underlying neuroblastoma. Historically, many children had relatively poor long-term outcomes, with residual neurologic and/or neuropsychiatry sequelae.
OMS is a rare disease and its relationship with anesthesia procedures has not been studied in detail. To our knowledge, there are only four case reports describing anesthesia management of OMS,[2],[3],[4],[5] none of them with epidural-general anesthesia.
| Case Report | |  |
We present a 9-year-old female patient with learning disabilities and OMS, weighing 27 kg. Written informed consent to publication was obtained from the guardian.
At 2 years of age, she presented with opsoclonus and myoclonus after metoclopramide therapy. She was medicated with human immunoglobulin 400 mg/kg/day for 5 days and corticotherapy (dexamethasone and prednisolone). She maintained ataxia and residual tremor. Image and blood tests were inconclusive.
At 5 years of age, she presented with distal dystonia of upper limbs and slight ataxia. She was medicated with propranolol and her condition improved.
At 7 years of age, she had secondary enuresis, and abdominal ultrasound showed paravertebral/right suprarenal mass, and low-grade neuroblastoma diagnosis was confirmed with meta-iodobenzylguanidine scintigraphy and magnetic resonance imaging. Due to the mass size (51 × 32 × 50 mm), it was decided for remotion.
During the preoperative examination, the airway showed normal features (Mallampati I), and cardiopulmonary examination and laboratory parameters were unremarkable.
On the day of the surgery, the child was premedicated with 7 mg syrup midazolam. Intravenous induction was done with alfentanil 500 μg, lidocaine 25 mg, propofol 85 mg, and rocuronium 15 mg. A lumbar epidural catheter was put in the L3–L4 level. Ropivacaine 0.375% was administered in two 15 mg boluses. Anesthesia maintenance was achieved with sevoflurane. Dexamethasone and ondansetron were also administered, 2.5 mg/each. Intraoperative analgesia was supplemented with intravenous ketorolac 10 mg and paracetamol 420 mg. Blood loss was around 50 mL and replaced with 200 mL of Ringer's lactate solution. At the end of the surgery, the patient was warm, hemodynamically stable, neuromuscular block was reversed with sugammadex 60 mg, extubated, and the child was shifted to a level-two critical care unit for postoperative monitoring. As postoperative analgesia, epidural ropivacaine 0.2% 16 mg was administered followed by an epidural perfusion of ropivacaine 0.15% 3 mL/h. This perfusion was adequate for postoperative analgesia in the first 48 h, with intravenous paracetamol 420 mg 8/8 h and ketorolac 10 mg 8/8 h.
The postoperative period was uneventful, and the child was discharged 4 days later. Follow-up after 6 months showed neurologic improvement, without any tremor or other complaints.
| Discussion | | |
Despite being a case report, with the limitations associated with this type of scientific evidence, this article comprises one of the five published cases[2],[3],[4],[5] about anesthesia management in OMS and is the first one to describe a successful combined neuraxial-general approach.
Because neuroblastomas are located along the sympathetic chain or in the adrenals, including the neck, thoracic, abdominal, or pelvic cavities[6]; neuraxial anesthesia may help blunting sympathetic symptoms during surgery.[7]The adjunctive use of regional anesthesia during general anesthesia may effectively decrease the intraoperative requirements for intravenous and volatile agents, thereby providing a more rapid awakening, earlier tracheal extubation, effective blunting of the surgical stress response, and limiting the need for parenteral opioids. Additionally, many of these reports demonstrate a reduction in the intraoperative requirements for volatile anesthetic agents.[8] Likewise, this type of approach allows for improved hemodynamic stability, lowering the risk for cardiopulmonary failure and respiratory depression.[9] Nowadays, nearly one-quarter of anesthetic procedures performed today involve regional anesthesia[10] and this seems to be a secure and effective perioperative analgesic.[11] The additive properties of general and regional anesthetic techniques are brought together in combined anesthesia to minimize the side effects of individual techniques.[12]
Due to the information given in the literature[13] and confirmed firsthand in a case report,[4] ketamine seems to be an anesthetic to avoid, because it may cause further sympathetic stimulation in patients with neuroendocrine tumors, similarly to etomidate and meperidine.[5] As a safe option, inhalational agents, opioids, non-depolarizing neuromuscular blockers, nonsteroidal anti-inflammatory drugs, propofol, anticholinergics, and anticholinesterase should be considered instead.
Adding to the sparse anesthetic management information in OMS, we now show one more possible approach that can be valuable in high-risk cases, where general anesthesia can be involved with a higher risk for the patient.
Informed consent
Informed consent to publication was obtained from the patient or guardian.
Author contributions
João B. Abreu – Conceptualization, investigation, and writing.
Catarina R. Cordeiro – Conceptualization and writing.
Ana I. Amorim – Writing.
Tiago G. Catanho – Writing.
Karina D. Gama – Review.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Kinsbourne M. Myoclonic encephalopathy of infants. J Neurol Neurosurg Psychiatry 1962;25:271-6.  |
| 2. | Maranhão M, Holanda A, Tavares F. Kinsbourne syndrome: Case report. Braz J Anesthesiol 2013;63:287-9. |
| 3. | Lee J, Kim D, Jeon B, Oh J, Han Y. Anesthesia in a young adult with opsoclonus-myoclonus syndrome. Korean J Anesthesiol 2014;67:5-6. |
| 4. | Burrows F, Seeman R. Ketamine and myoclonic encephalopathy of infants (Kinsbourne syndrome). Anesth Analg 1982;61:873-5. |
| 5. | Nisa N, Talawar P, Vasudevan B. Anesthesia in a child with Kinsbourne syndrome: Does anesthesia technique matters? Saudi J Anaesth 2016;10:468-70. |
| 6. | Rossor T, Yeh E, Khakoo Y, Angelini P, Hemingway C, Irani S, et al. Diagnosis and management of opsoclonus-myoclonus-ataxia syndrome in children: An international perspective. Neurol Neuroimmunol Neuroinflamm 2022;9:8-9. |
| 7. | Chamberlain D, Chamberlain B. Changes in the skin temperature of the trunk and their relationship to sympathetic blockade during spinal anesthesia. Anesthesiology 1986;65:139-43. |
| 8. | Goeller J, Bhalla T, Tobias J. Combined use of neuraxial and general anesthesia during major abdominal procedures in neonates and infants. Paediatr Anaesth 2014;24:553-60. |
| 9. | Marhofer P, Lönnqvist P. The use of ultrasound-guided regional anaesthetic techniques in neonates and young infants. Acta Anaesthesiol Scand 2014;58:1049-60. |
| 10. | Ecoffey C, Lacroix F, Giaufré E, Orliaguet G, Courrèges P. Epidemiology and morbidity of regional anesthesia in children: A follow-up one-year prospective survey of the French-Language Society of Paediatric Anaesthesiologists (ADARPEF). Paediatr Anaesth 2010;20:1061-9. |
| 11. | Baird R, Guilbault M, Tessier R, Ansermino J. A systematic review and meta-analysis of caudal blockade versus alternative analgesic strategies for pediatric inguinal hernia repair. J Pediatr Surg 2013;48:1077-85. |
| 12. | Litz R, Bleyl J, Frank M, Albrecht D. Combined anesthesia procedures. Anaesthesist 1999;48:359-72. |
| 13. | Watso J, Huang M, Moralez G, Cramer M, Hendrix J, Cimino F, et al. Low dose ketamine reduces pain perception and blood pressure, but not muscle sympathetic nerve activity, responses during a cold pressor test. J Physiol 2021;599:67-81. |
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