Post-Operative Pain Management in Ehlers-Danlos Hypermobile-Type Syndrome Following Wisdom Teeth Extraction: A Case Report and Literature Review

Authors: Aikaterini Amanatidou

Abstract:

We describe the case of a 20-year-old female patient diagnosed with Ehlers-Danlos Syndrome (EDS) who was scheduled to undergo a wisdom teeth extraction in outpatient surgery. EDS is a hereditary connective tissue disorder characterized by joint hypermobility, skin hyper-extensibility, and vascular and soft tissue fragility. There are six subtypes of Ehlers-Danlos, and in our case, the patient had EDS hyper-mobility (HT) type disorder. One important clinical feature of this syndrome is chronic pain, which is often poorly understood and treated. Our patient had a long history of articular and lumbar pain when she was diagnosed. She was prescribed analgesic treatment for acute and neuropathic pain and had multiple sessions of psychotherapy and physiotherapy to ease the pain. Unfortunately, her extensive medical history was underrated by our anesthetic team, and no further measures were taken for the operation. Despite an uneventful intra-operative phase, the patient experienced several episodes of hyperalgesia during the immediate post-operative care. Management of pain was challenging for the anesthetic team: initial opioid treatment had only a temporary effect and a paradoxical reaction after a while. Final pain relief was eventually obtained with psycho-physiologic treatment, high doses of ketamine, and patient-controlled analgesia infusion of morphine-ketamine-dehydrobenzperidol. We suspected an episode of Opioid-Induced hyperalgesia. This case report supports the hypothesis that anti-hyperalgesics such as ketamine as well as lidocaine, and dexmedetomidine should be considered intra-operatively to avoid opioid-induced hyperalgesia and may be an alternative solution to manage complex chronic pain like others in neuropathic pain syndromes.

Keywords: Ehlers-Danlos, post-operative management, hyperalgesia, opioid-induced hyperalgesia, rare disease