Case Report- ECT for Autism-related Stereotyped Emesis

Out on PubMed, from clinicians at the Department of Psychiatry, Penn State Medical School, Hershey, PA is this LTE case report:  

Electroconvulsive Therapy for Autism-related Stereotyped Emesis After Chiari Malformation Type 1 Craniectomy.

Mormando C, Mikoluk C, Garman JC, Rapp M, Murray M, Francis A.J ECT. 2020 Dec 17;Publish Ahead of Print. doi: 10.1097/YCT.0000000000000738. Online ahead of print.PMID: 33337647

Excerpts from the text:
A 21-year-old White man with a history of autism was referred to our ECT service after a 4-year history of unexplained repetitive emesis reaching over 200 episodes per day. Prior studies revealed that CM1, which was treated with craniectomy at age 17 years, partially aimed at relief of the emesis. This surgery consisted of partial resection of the occipital bones and removal of some cerebellar tissue. Despite this procedure, the emesis persisted. He developed tonic-clonic and possibly absence seizures postoperatively, and was known to have had at least 3 witnessed motor seizures. An electroen- cephalogram obtained at age 19 years showed generalized spike-and-wave fea- tures. His seizures were well controlled on lamotrigine. A brain magnetic resonance imaging without contrast at age 19 years showed right-sided cerebellar gliosis and occipital craniectomy but was otherwise normal. Several behavioral interventions, antiemetics, and psychotropic agents proved ineffective, including an extended longer than 12 months hospitalization at a special- ized neurobehavioral treatment center for individuals with developmental disorders. Ultimately, management consisted of com- plete daytime restriction of oral food and water, with enteral nutrition from night-time gastrojejunal tube feedings, and polypharmacy with high-dose benzodiazepines. De- spite these interventions, the patient had lost weight and developed symptomatic hypokalemia, leading to referral for ECT.
...in light of identified catatonic symp- toms and framing the frequent emesis as a catatonic stereotypy, we posited a possible benefit of ECT. Bitemporal ECT was initi- ated 3 times per week with consent of the parental guardians. Because of aversion to needles, the patient was sedated with nitrous oxide, followed by intravenous flumazenil, methohexital or etomidate, and remifentanil. Electroconvulsive therapy was performed with a Thymatron System IV (0.5 ms pulse width, 70 Hz). Initial ECT dosing used the “half-age” method. Seizures were typically 30 to 40 seconds on electroencephalogram and were well formed.
...The patient showed marked improvement in emesis frequency over a treatment course of 7 months. Daily emesis counts dropped from 216 ± 7 (mean ± SEM) over the 2 months pre-ECT to 30 ± 7 and then 5 ± 1 in the following 2-month intervals totaling 4 months, while mean daily aggressive episodes dropped from 18 ± 2 to <1. Electrolytes normalized, he gained weight, and overall functioning was markedly im- proved. Daily lorazepam doses were reduced, and haloperidol, escitalopram, and propranolol were discontinued. He continued with maintenance ECT at varying intervals.




This is an interesting case report with 2 messages: 1) that repetitive stereotyped behaviors (including frequent emesis, as in this case) in the context of ASD may usefully be conceptualized as catatonic symptoms and 2) that ECT can be safely performed in patients with Chiari malformations (either with, or without, prior surgical repair.)
In addition, the pre-anesthesia sedation with nitrous oxide is innovative.
From personal communication with Dr. Mormando, the patient has remained stable for 2 years, with maintenance ECT q 6-8 weeks.
This LTE adds to the growing literature on ECT for the treatment of self-injurious and related catatonic symptoms in patients with ASD.


Comments

  1. We describe a 21-year-old male post-craniectomy for CM1 and autism-related stereotyped emesis successfully treated with ECT. This study affords quantitative data that autism-related stereotyped behavior may dramatically improve with ECT if recognized and treated as catatonic stereotypy. A broader definition of catatonic behaviors in individuals with neurodevelopmental disorders should be considered.

    Furthermore, ECT appears to be safe in patients with Chiari malformations (with or without surgical repair). A table summarizing our literature review of ECT in patients with Chiari malformation is included in Dr. Kellner's above post.

    - Charles Mormando

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