Puzzling cause of a delayed recovery and persistent neurological deficit after urological surgery

OBSERVATION

A 74-year-old, ASA II female patient with past history of high blood pressure, dyslipidemia, smoking habits, osteoporosis, depression, chronic left shoulder pain, and a body mass index of 19 kg/m² was scheduled an artificial urethral sphincter placement under laparoscopy. Her surgical history encompassed several procedures under general anesthesia including tonsillectomy, appendicectomy, colic polypectomy, bilateral hallux valgus treatment, narrow lumbar canal enlargement, and complete radical hysterectomy. One year ago, she already benefited from a laparoscopic promonto-fixation, and the treatment of her urinary incontinence with sub-urethral slings. All these procedures were achieved without any relevant complication. Her chronic medications included lisinopril, pravastatine, omeprazole, risedronic acid, bromazepam, paracetamol, and piroxicam.

The patient received alprazolam 0.5 mg orally as a premedication, 60 minutes before the procedure. Induction of general anesthesia was achieved using intravenous sufentanil 5 µg, lidocaïne 60 mg, propofol 120 mg, kétamine 15 mg, atracurium 25 mg, dehydrobenzperidol 1.25mg, ketorolac 25 mg, and clonidine 150 µg. Anesthesia was maintained using sevoflurane.  The anesthetic technique, including type of drugs and dosage, was almost identical to the one used for the previous intervention one year ago. The procedure was uneventful except for a conversion from laparoscopy to laparotomy through a Pfannenstiel incision due to peritoneal adherences. Noteworthy, no major hemodynamic disturbances occurred. At the end of the procedure, after return of spontaneous breathing and airway protective reflexes, the endotracheal tube was removed, and patient was transferred to the post anesthesia care unit.

Surprisingly, after 2 hours, the patient had still not recovered full consciousness. At that time, her Glasgow Score was 8/15 (M5 V1 E3).

TABLE 1

DELAYED RECOVERY

DIAGNOSTIC TOOL

Case Report

Respiratory disturbances

Oxygen saturation, capnography, arterial blood pHmetry

 oxygen saturation, arterial gazometry in the normal range

Drugs’overdose

Signs of overdose (pupil dialeter, respiratory frequency…)

Pupils reactive and symmetric, normal respiratory frequency

Metabolic disturbances

Blood sampling

 Glycemia and blood pH in the normal range

Ionic disturbances

Blood sampling

No ionic disturbances

Hypothermia

Thermometry

Normothermia

After exclusion of all the usual causes of delayed recovery (Table 1), and discussion with a neurologist, a CT scan of the brain was performed. Conclusions of this exam were not clear cut, with a suspicion of sub-acute ischemic deep injury in the right middle cerebral artery territory.                                                                          

Finally, four hours after the completion of surgery, the patient suddenly recovered a full wake state, with a Glasgow Score of 15/15, but still with a paresis in the right lower limb. According to these symptoms and the results of the CT scan, anti-aggregation therapy (aspirin) was started. On the next morning, no improvement of the neurological symptoms was observed. Moreover, paresis in the left upper and lower limbs, along with hypoesthesia of the right side of the face was reported by the patient.

At this point, several complementary exams were performed. First, a brain and spinal MRI failed to confirm any central ischemic origin. Electrocardiography, cardiac echocardiography, and an imaging of the vessels of the neck excluded any embolic etiology.  A peripheral origin for the neurological problem was excluded by an EMG. Somatosensory and motor Evoked Potentials (SEPs) were also reported as normal.

The clinical course of the patient showed poor improvement. Several weeks after surgery, she was still unable to walk alone, the strength tests were fluctuating daily, while the topography of the symptoms remained the same.

Incidentally, during an interview with a neurologist, the patient revealed that her twin sister presented the same post-operative complication with a complete recovery. Interestingly, in this case, no explanation was found. The patient also mentioned that, for the first time during its entire lifetime, she was separated from her twin sister during a two months period. She experienced this separation as extremely stressful.

Considering the atypical delayed recovery of consciousness, the neurological symptoms, the normality of all the additional examinations, the fluctuation of clinical signs, the familial history, and the stress due to the separation from her twin sister, a psychiatric origin for the disorder was proposed.

The explanation for this quite unusual postoperative course is most likely the first episode of a conversion disorder or hysteria.

DISCUSSION

The first medical descriptions of hysteria were performed in 1880 by Jean-Martin CHARCOT: “hysteria symptoms are due to a traumatic shock, which causes consciousness dissociation, the memory of which is still unconscious or subconscious”. It laid the basis to the “trauma-dissociative theory” of neurosis. Charcot’s works have been taken up by Sigmund FREUD, who defines conversion as the transformation of a psychological conflict in physical symptoms. At the present time, the terminology of “conversion disorder” is preferred to “hysteria”.

In modern psychiatry, hysteria is a feature of hysterical disorders, in which patient experiences physical symptoms that have a psychological, rather than an organic origin. It is thought to be a defense mechanism to escape painful emotions by unconsciously transferring this distress to the body. There may be a symbolic function for this conversion. For example, a rape victim may develop paralysis of the legs. For this reason, symptoms of hysteria may mimic numerous physical (i.e. neurological) disorders. As a consequence, the diagnosis can only be evoked when all the other nosological entities have been ruled out [1].

The physiopathology underlying this somatoform disorder is still controversial. However, an abnormal activation of the parietal lobe, which reduces the activity of the prefrontal cortex, the supramarginal gyrus and the precuneus, has been recorded in patients with conversion hysteria, in comparison to a control group, suggesting a neurophysiological substratum related to this disorder [2]. Conversion disorder or hysteria is mostly but not exclusively found in young age, female patients. Social or familial instability are recognized as risk factors [3]. The prevalence of this disease in the general population is 11/100,000. Occurrence of this disorder during the immediate postoperative period is extremely rare, as evidenced by the very few case reports in the literature. Most of the time, unexplained delayed recovery was observed [4] [5] [6] [7]. Conversion hysteria has even been described following epidural anesthesia [8] [9].

CONCLUSION

We here report a rare postoperative complication, namely conversion hysteria. Its diagnosis is based on the exclusion of all the organic causes leading to the observed symptoms. Anesthesia practitioners should be aware of its potential occurrence during the peri-operative period.

REFERENCES

[1]American Psychiatric Association. 1400 K Street NW, Washington DC 20005. http://www.psych.org/

[2] Van Beilen M, De Jong BM, Gieteling EW, Renken R, Leenders KL. Abnormal parietal function in conversion paresis. PLoS One 2011;6:e25918.

[3] American Psychiatric Association. Diagnostic and statistical manual of mental disorders, 4th ed., Washington, DC: American Psychiatric Association; 1994.

[4] Chieko Nakagawa, Yoshito Shiraishi, Shigehito Sato. A case of conversion disorder showing transient hemiplegia after general anesthesia. 8 January 2010 Japanese Society of Anesthesiologists. 

[5] McClelland MRA. A case of hysteria. Anaesthesia 1956;11:353–4.

[6] Adams AP, Goroszeniuk T. Hysteria: a cause of failure to recover after anaesthesia. Anaesthesia 1991;46:923–4.

[7] MaddockH,CarleyS,McCluskeyA. An unusual case of hysterical postoperative coma. Anaesthesia 1999;54:717–8.

[8] Collier CB. Un planned unconsciousness at caesarean section: hysteria or drug reaction? Int J Obstet Anesth 2007;16:192–3.

[9] M. Bensghir a, H. Alaoui a, R. Ahtil a, H. Azendour a, K. Mouhadi b, N. Drissi Kamili

Impaired consciousness associated with a hysterical conversion after obstetrical epidural analgesia: A case report with literature review. Annales Françaises d’Anesthésie et de Réanimation 31 (2012) 919–921