A case of delayed-onset multiple oculogyric crisis and torticollis episodes after low dose intramuscular haloperidol in a non-neuroleptic drug overdose setting

This report documents a rare case of delayed-onset multiple acute dystonias after treatment with low dose intramuscular (IM) haloperidol lactate injection in a setting of non-neuroleptic drug overdose. The drug–drug interactions between haloperidol and high levels of paracetamol and naproxen are deliberated upon. A 25-year-old Asian female was admitted after an intentional overdose of medications (paracetamol, naproxen and pregabalin). She received 5 mg of IM haloperidol injection for agitation. 21 hours later she experienced mild intermittent ocular deviation in an upward and outward direction and generalised stiffness, which were self-resolving. An hour later, she required another 2.5 mg of IM haloperidol injection for further agitation. In the 35 hours following her first IM haloperidol (13 hours after the second IM haloperidol), she developed a total of three episodes of oculogyric crisis (OGC) with torticollis. Each episode was treated promptly with IM diphenhydramine 25 mg, and there was remission of symptoms within 15 minutes of treatment. An objective causality assessment revealed a definite relationship between the episodes of acute dystonia with IM haloperidol therapy. Where oral alternatives and IM atypical antipsychotics/benzodiazepines are unavailable, rapid tranquillisation with a high-potency typical antipsychotic is a possibility. However, consideration should be made to combine haloperidol with an anticholinergic agent as prophylaxis against acute dystonia, especially in the setting of drug overdose, even if it is that of a non-neuroleptic drug (in this case, paracetamol and naproxen).

Essential Infantile Esotropia: A Course of Treatment From Our Experience

Background: Essential infantile esotropia (EIE) is the most common type of childhood esotropia. Although its classical approach is surgical, less invasive techniques have been proposed as an adjunct or alternative to traditional surgery. Among them, chemodenervation with botulinum toxin (BT) has been investigated, showing variable and sometimes conflicting results.Objectives: To compare the outcomes of bilateral BT injection and traditional surgery in a pediatric population with EIE in order to optimize and standardize the therapeutic approach. Other purposes are to evaluate whether early intervention may prevent the onset of vertical ocular deviation (which is part of the clinical picture of EIE) and/or influence the development of fine stereopsis, and also to assess changes in refractive status over time among the enrolled population.Methods: A retrospective consecutive cohort study was conducted in 86 children aged 0–48 months who underwent correction of EIE. The primary intervention in naïve subjects was either bilateral BT injection (36 subjects, “BT group”) or strabismus surgery (50 subjects, “surgery group”).Results: Overall, BT chemodenervation (one or two injections) was effective in 13 (36.1%) subjects. With regard to residual deviation angle, the outcomes at least 5 years after the last intervention were overlapping in children receiving initial treatment with either injection or surgery; however, the success rate of primary intervention in the surgery group was higher, and the average number of interventions necessary to achieve orthotropia was smaller. Both early treatment with chemodenervation and surgery at a later age were not found to prevent the onset of vertical ocular deviation, whereas, surprisingly, the percentage of subjects developing fine stereopsis was higher in the surgery group. Finally, with regard to the change in refractive status over time, most of the subjects increased their initial hyperopia, whereas 10% became myopic.Conclusions: Our data suggest that a single bilateral BT injection by age 2 years should be considered as the first-line treatment of EIE without vertical component; whereas, traditional surgery should be considered as the first-line treatment for all other cases and in subjects unresponsive to primary single BT injection.

To measure the amount of ocular deviation in strabismus patients with an eye-tracking virtual reality headset

Purpose To investigate the accuracy of a newly developed, eye-tracking virtual reality (VR)-based ocular deviation measurement system in strabismus patients. Methods A VR-based ocular deviation measurement system was designed to simulate the alternative prism cover test (APCT). A fixation target was made to alternate between two screens, one in front of each eye, to simulate the steps of a normal prism cover test. Patient’s eye movements were recorded by built-in eye tracking. The angle of ocular deviation was compared between the APCT and the VR-based system. Results This study included 38 patients with strabismus. The angle of ocular deviation measured by the VR-based system and the APCT showed good to excellent correlation (intraclass correlation coefficient, ICC = 0.897 (range: 0.810–0.945)). The 95% limits of agreement was 11.32 PD. Subgroup analysis revealed a significant difference between esotropia and exotropia (p < 0.001). In the esotropia group, the amount of ocular deviation measured by the VR-based system was greater than that measured by the APCT (mean = 4.65 PD), while in the exotropia group, the amount of ocular deviation measured by the VR-based system was less than that of the APCT (mean = − 3.01 PD). The ICC was 0.962 (range: 0.902–0.986) in the esotropia group and 0.862 (range: 0.651–0.950) in the exotropia group. The 95% limits of agreement were 6.62 PD and 11.25 PD in the esotropia and exotropia groups, respectively. Conclusions This study reports the first application of a consumer-grade and commercial-grade VR-based device for assessing angle of ocular deviation in strabismus patients. This device could provide measurements with near excellent correlation with the APCT. The system also provides the first step to digitize the strabismus examination, as well as the possibility for its application in telemedicine.

Correlation between uniocular deviation and duction changes following different decompression surgeries in thyroid eye disease

Background Postoperative ocular imbalance is an important problem for orbital decompression surgery in thyroid eye disease (TED). The aim of this study was to evaluate the changes in unilateral ocular deviation and duction following orbital decompression and discuss the biomechanics of ocular imbalance. Methods Fifty-four TED patients who underwent unilateral orbital decompression were included. Fifteen patients underwent 1-wall (deep lateral wall) decompression, 18 patients underwent 2-wall (deep lateral and medial wall) decompression and 21 patients underwent 3-wall (deep lateral, medial and inferior wall) decompression. Objective and subjective deviation of the operated eyes were evaluated using the prism test and synoptophore, respectively. Ocular ductions were measured using Hirschberg’s method. The diameters of the extraocular rectus were measured by computed tomography. Results Ocular deviation and duction showed no significant difference after 1-wall decompression (p = 0.25–0.89). Esotropia increased after 2-wall decompression (p = 0.001–0.02), and hypotropia increased after 3-wall decompression (p = 0.02). Adduction increased but abduction decreased following 2-wall and 3-wall decompression (p < 0.05). Infraduction increased following 3-wall decompression (p < 0.001). Additionally, the increase in esotropia was significantly correlated with the increase in adduction and with the decrease in abduction (r = 0.37–0.63, p < 0.05). There were significant correlations between the diameter of the medial rectus and the increase in esotropia, the increase in adduction and the decrease in abduction postoperatively (r = 0.35–0.48, p < 0.05). Conclusions The changes in ocular deviation and duction were different after 1-wall, 2-wall and 3-wall orbital decompression. The increased contractile force of the rectus may be an important reason for strabismus changes after orbital decompression surgery.

Strabismus in Generations and Among Siblings

Introduction: Strabismus has been observed among family members in involved families. The study was conducted to document manifest strabismus running in generations and among siblings of strabismic patients. Methods: It was a hospital based prospective study conducted from May to November 2017. Strabismic patients with ocular deviation visiting Department of Paediatric Ophthalmology and strabismus were enrolled using specially designed proforma. Detailed ocular examinations were done and family history obtained regarding the presence of strabismus. Family tree was drawn for those with positive family history. Results: A total of 78 participants completed the study with 48.7% males. Family history of strabismus was present in 56.4%. Strabismus was present in two generations in 45.45%, in three generations in 18.2%, 1st and 3rd generation in 18.2%, 1st, 2nd and 4th generation in 4.5% and among siblings only in 13.6%. Among those with positive family history, exotropia was present in 75% and esotropia in 25%. Among exotropes, manifest deviation was present in 47.7% and intermittent exotropia in 27.3%. Among esotropes, congenital esotropia was present in 9% and accommodative esotropia in 16%. First degree relatives were affected in 45.45% of strabismic patients among which 55% had manifest exotropia, 20% had intermittent exotropia, 15% had accommodative esotropia and 10% had congenital esotropia. Conclusions: Strabismus was found in different generations in 56.4% strabismic patients. Two generations were involved in 45.45%, three generations in 18.2%, 1st and 3rd generation in 18.2%, 1st, 2nd and 4th generation in 4.5% and siblings only in 13.6%. Genetic factors appear to be significant in strabismus in Nepalese population and it requires to be substantiated with further larger studies.

The effect of amblyopia on clinical outcomes of children with astigmatism

Background: Amblyopia is more common in children with high astigmatism, but factors contributing to development of amblyopia and visual outcomes are not fully understood. Objective: To evaluate the effect of amblyopia on the clinical outcomes in children with ⩾1.75 diopter (D) astigmatism. Methods: We reviewed the medical records of children with ⩾1.75 D astigmatism with and without amblyopia (amblyopes group and non-amblyopes group). The mean age, gender, amount and type of ocular deviation, presence of convergence insufficiency (CI), stereopsis, time of initial spectacle use and follow-up time, differences in best-corrected visual acuity (VoD) and spherical equivalent (SE) between eyes were assessed and compared between the groups. Best-corrected visual acuity (BCVA), mean SE, astigmatism measurements were assessed and compared between amblyopic, fellow, and non-amblyopic eyes. Results: The records included 68 eyes of 34 children with amblyopia and 56 eyes of 28 children without amblyopia. The mean age, gender, amount and type of ocular deviation, presence of CI, stereopsis, time of initial spectacle use, follow-up time, and the difference in SE did not differ between groups. In amblyopes, exodeviation was more common and statistically greater in near (33 cm) than at distance (6 m) (p = 0.005). The mean BCVA and astigmatism values were statistically different between amblyopic, fellow, and non-amblyopic eyes. Conclusion: A greater near than distance exodeviation and higher mean astigmatism value were found in amblyopic children with astigmatism.

Oculogyric Crisis in a patient with PURA Syndrome

Context: PURA syndrome is a neurodevelopmental disorder characterized by neonatal hypotonia, delayed psychomotor development, early-onset feeding difficulties and an epileptic encephalopathy. Case Report: A 3-month-old Brazilian boy presented with severe neonatal hypotonia associated with feeding difficulties due to serious dysphagia requiring nasoenteral tube feeding. Excessive drowsiness, poor social interaction and repetitive episodes of involuntary abnormal upward eye movements and ocular version with short duration were also reported by parents. Neurological examination revealed severe axial and upper limb hypotonia, orofacial dyskinetic movements and episodes of abnormal eye movements with upward ocular deviation with less than 30 seconds in duration compatible with oculogyric crisis. It was performed Whole-Exome sequencing and it was identified a new pathogenic variant in PURA gene that establisehd the final diagnosis of PURA Syndrome or Autosomal Dominant Mental Retardation type 31, MDR 31 (OMIM #616158). Conclusions: PURA Syndrome emerges as one of the major differential diagnoses of neonatal hypotonia and in addition, we can consider the early manifestation of oculogyric crisis as a phenotypic expansion of the syndrome, making its diagnosis even more challenging, since epileptic encephalopathies and neurotransmitter deficiency-related diseases present with a similar clinical course.

Predictive factors of stereopsis outcomes following strabismus surgery

Purpose: To determine the predictive factors of post-operative stereopsis in patients with strabismus. Method: In this retrospective study, records of patients who received surgical treatment for strabismus were reviewed. All types of strabismus were included. Pre- and post-treatment stereoacuity were measured using the Titmus Stereo Fly test, and predictive factors of stereopsis were evaluated. Results: A total of 194 patients (132 females and 62 males) with a mean age of 14.8 ± 8.4 years were included. There was a statistically significant improvement in stereopsis following surgery ( p value < 0.001). Patients with a higher amount of deviation at baseline had poorer stereopsis on the final examination ( p value < 0.001). Stereopsis improvement was more prominent in the pure horizontal strabismus group, compared to combined horizontal and vertical deviations. Baseline and the final stereopsis were higher in the “exotropia” group as compared to the “esotropia” group ( p value = 0.003 and 0.0155, respectively); however, the within group change of stereopsis was not significantly different between these two groups ( p value = 0.144). Post surgical residual deviation was associated with a poorer stereopsis ( p value = 0.002, r = 0.251). A longer duration of strabismus before surgery was associated with poorer final stereopsis levels ( p value = 0.026). The presence of amblyopia before surgery was associated with poorer stereopsis on last examination ( p value < 0.001 for both correlations). Conclusion: Based on the result of this study, final stereopsis after strabismus surgery could be affected by the type, duration, and the amount of deviation before surgery, amblyopia, and post operative ocular deviation.