Takashi Furuse

Chromosomes 4q28.3 and 7q31.2 as New Susceptibility Loci for Comitant Strabismus

PURPOSE This study was designed to localize chromosomal susceptibility loci for comitant strabismus among Japanese families by genome-wide linkage analyses. METHODS Fifty-five Japanese families, with at least two members with comitant strabismus (esotropia and/or exotropia), were subject to full ophthalmic examination, careful ocular history, and review of medical records. DNA was obtained and genotyping was performed with PCR amplification of 400 microsatellite markers. Parametric and nonparametric linkage (NPL) analyses scores were calculated. Linkage analysis was performed for the whole set of families (55 families), and then a second analysis was performed for two subgroups with the phenotypes, esotropia and exotropia. RESULTS A multipoint parametric heterogeneity logarithm of the odds (HLOD) score of 3.62 was obtained at marker D4S1575 under a dominant model, with a NPL score of 2.68 (P=0.001). Testing under different penetrances and disease allele frequencies revealed two other susceptibility loci at 7q31.2 under a recessive model (HLOD scores=3.93 and 4.40 at 125.2 cM and 107.28 cM, respectively). Analysis of the subgroups revealed new susceptibility loci for esotropia; one locus at 8q24.21 is worthy of further investigation. CONCLUSIONS This study suggests multiple susceptibility loci for comitant strabismus. The loci at chromosomes 4q28.3 and 7q31.2 show a significant evidence of linkage.

Strabismus Surgery in Ocular Myasthenia gravis

We evaluated the efficacy of eye muscle surgery in 4 patients with ocular myasthenia gravis with troublesome diplopia. All patients were in remission and had shown a consistently stable angle of deviation for at least 6 months preoperatively. The extent of eye muscle surgery was based on the degree of deviation in the primary position, and conventional recession and resection procedures were performed in 3 patients and hang-loose recession with an adjustable recession in 1. Single binocular vision was obtained in 3 patients in the primary position without prism correction or compensatory head posture. Electron-microscopic studies on the eye muscle specimens obtained at strabismus surgery revealed non-specific degenerative findings for the muscle fibers and sarcomeric disorganization compared to that in a control extraocular muscle.

Extraocular muscle surgery in a rabbit model: site of reattachment following hang-back and conventional recession.

To determine the precise site of reattachment of recessed muscles, 4-mm conventional and hang-back recessions of the inferior rectus muscle were performed in 18 albino rabbits. Six weeks later, the distance from the anterior border of the reattached muscle to the insertion was measured both grossly and microscopically. In all cases the operated muscles had advanced minimally from the site of surgical placement. Gross observation showed that the mean forward creep was significantly greater for those rabbits which underwent hang-back recession (1.81±0.67 mm) than for those that were submitted to the conventional technique (0.83±0.38 mm). Measurements done on histological sections revealed that the mean distance of the anterior border of the muscle fiber from the reference suture was larger for conventional recession (2.73±0.75 mm) than for hang-back recession (1.91±0.72 mm).

Critical Period for Restoration of Normal Stereoacuity in Acute-onset Comitant Esotropia

We conducted a retrospective study of 25 patients with acute-onset comitant esotropia to evaluate whether the timing of the start of treatment is a critical factor in the development of normal stereopsis. The mean age at onset was 12 years 4 months, and mean age at the start of treatment was 17 years 9 months. Bifixation was defined as a stereoacuity threshold score that was numerically lower than 60 seconds of arc on stereotesting. An operation was performed on the nonfixating eye for the prism-adapted angle. At the final examination, bifixation was observed in four patients (16%) with the Randot test and in 15 patients (60%) with the Titmus test. No relationship was found between the time of the start of treatment and the postoperative development of stereopsis, nor was there a significant (P > .10) difference between the two groups with early and delayed start of treatment in the proportion of patients with bifixation.

Intraoperative adjustable suture surgery for bilateral superior oblique palsy.

BACKGROUND The modified Harada-Ito procedure has been reported to be an effective treatment for correction of cyclotorsion in bilateral superior oblique palsy. However, there are no reports regarding its use in intraoperative adjustable suture surgery. METHODS The authors performed a retrospective study of 12 patients with traumatic bilateral superior oblique palsy who were classified as having either symmetric or asymmetric palsy according to the symmetry of the alternate hyperdeviation on side gazes. Cyclotorsion and vertical and horizontal deviation in the nine diagnostic positions were measured preoperatively and postoperatively. RESULTS Of the 12 patients, 6 were determined to have symmetric palsy and 6 asymmetric palsy. Intraoperative adjustable suture surgery with the modified Harada-Ito procedure was performed bilaterally in the six patients with symmetric palsy and unilaterally in those with asymmetric palsy. The median measured value of extorsion in the primary position was reduced from 14.5 degrees to 2.5 degrees in patients with symmetric palsy and from 9.5 degrees to 2.0 degrees in those with asymmetric palsy. In downgaze, some degree of residual extorsion remained, and there was no significant change in esodeviation after surgery. In five patients with symmetric palsy and in all of those with asymmetric palsy, normal single binocular vision in the primary position but did not that in downgaze was restored after surgery. CONCLUSION Intraoperative adjustable suture surgery is an effective treatment in correcting torsion, but may not be as effective for esodeviation in downgaze.

Morphological Changes in the Orbital Surface Layer Muscle of the Rabbit Eye Produced by Botulinum Toxin

We quantitated the morphological changes in the orbital surface layer muscles in the rabbit following the single injection of botulinum A toxin. Experiments were performed in 9 white rabbits (1.6–3.0 kg). They were administered 5 units (5 rabbits) or 10 units (4 rabbits) of botulinum toxin injected into the superior rectus muscle of one eye. The diameter of myofibers of the orbital and intermediate layer zones was measured with an ocular micrometer on histological sections 3 days and 1, 3 and 5 weeks after injection. Quantitative changes were noted in the muscle fibers of the orbital surface layer zone following the injection of 10 units of botulinum toxin. At 1 week, the diameter of myofibers in the orbital layer was reduced, but it was increased at 5 weeks compared to that in the control eyes; in contrast, no change in the diameter of muscle fibers was found in the intermediate layer zone.

Transposition of the anterior superior oblique insertion as a treatment for excyclotorsion induced from limited macular translocation.

PURPOSE To evaluate the transposition of the anterior superior oblique insertion as a treatment for cyclovertical diplopia accompanied by an awareness of tilted image perceived with the affected eye induced from limited macular translocation (LMT). DESIGN Observational case series. METHODS Transposition of the anterior part of the superior oblique tendon combined with or without vertical muscle surgery on the affected eye was retrospectively studied in seven patients. Clinical outcome was assessed for binocular and monocular vision. A successful result was defined as restoration of single binocular vision (SBV) at distance and near examined with the Bagolini test with disappearance of a tilted image perceived in the affected eye. RESULTS Six of seven patients (86%) became unaware of tilted image, and three patients (43%) obtained successful results after the strabismus surgery. Of these three patients with successful results, one (33%) patient recognized metamorphopsia, whereas two (67%) of the three patients with unfavorable results reported metamorphopsia. Patients with successful results showed a visual acuity of 20/25 or better in the affected eye and a significantly smaller difference in visual acuity between the two eyes than those patients with unfavorable surgical results (0.133 logarithm of the minimal angle of resolution for SBV(+) vs 0.675 logarithm of the minimal angle of resolution for SBV(-); P =.0255). CONCLUSIONS The relatively low success for restoration of SBV indicates that strabismus surgery is recommended for patients whose difference in visual acuity between the two eyes is small and who have a high level visual acuity of the affected eye.

Case Report and Literature Review of Inferior Rectus Muscle Aplasia in 16 Japanese Patients

Purpose: To describe clinical features of inferior rectus muscle aplasia in Japanese patients and to gain insight into its pathogenesis. Methods: Case presentation and literature review of 16 Japanese patients with inferior rectus muscle aplasia. Results: The age at presentation of 16 patients (9 females, 4 males, and 3 with unknown gender) varied from 7 months to 73 years (mean, 21.8 years). The inferior rectus muscle was absent on the right side in 10 patients, on the left side in 4 patients, and on both sides in 2 patients. Structural anomalies of the eye globe, such as microphthalmos, microcornea, and coloboma of varying degrees, were present on the ipsilateral side of the inferior rectus muscle aplasia in 4 patients and on the contralateral side in 1 patient. As surgical findings, anomalous inferiorly located insertion of the medial rectus muscle was discovered in 5 patients: 1 patient in association with microcornea and iris coloboma. Frequent surgical procedures were recession or tenotomy of the superior rectus muscle, combined with downward transposition of the entire or partial tendons of the medial rectus and lateral rectus muscle to the medial and lateral end, respectively, of the putative inferior rectus muscle insertion. Conclusions: The association of coloboma with inferior rectus muscle aplasia suggests that abnormal optic fissure closure during embryogenesis might underlie the muscle aplasia.

Relationship between static ocular counterroll and Bielschowsky head tilt phenomenon.

PURPOSE This study was conducted to assess how hyperdeviation of a paretic eye during ipsilesional head tilt-the Bielschowsky head tilt phenomenon (BHP)-can be explained by decreased compensatory ocular counterrolling (OCR) due to the depressed torque of the paretic superior oblique (SO) muscle. METHODS Thirty-three patients with clinically diagnosed SO palsy and 11 control subjects were studied. With a head-mounted video camera, static ocular counterrolling (s-OCR) was determined by measuring the inclination of a line connecting the two centroids of the characteristic iris pattern and corneal reflex. The BHP was measured with the alternate prism and cover test. RESULTS The mean (SD) amplitude of s-OCR in paretic eyes based on the fit of the regression sine curve against the ipsilesional head tilt angle was significantly decreased compared with that for contralesional head tilt, 6.3 (3.5) degrees for ipsilesional and 11.3 (3.9) degrees for contralesional (P < 0.001), and was significantly smaller than that in normal subjects: 10.9 (2.6) degrees (P < 0.001). No significant linear relation was noted between hyperdeviation on ipsilesional head tilt and the amplitude of s-OCR in paretic eyes (r(2) = 0.04; P = 0.29). However, the differences between the hyperdeviation with ipsilesional 30 degrees head tilt and with head-upright position correlated significantly with the amplitudes of s-OCR in paretic eyes (r(2) = 0.19, P = 0.01). CONCLUSIONS The absolute value of the hypertropia on ipsilesional head tilt in clinically diagnosed SO palsy does not directly assess the function of the SO muscle. The difference in hypertropia between ipsilesional head tilt and the upright position, however, may be a better indicator of SO function.

Contribution of vergence adaptation to difference in vertical deviation between distance and near viewing in patients with superior oblique palsy

PURPOSE To evaluate the adaptive vertical vergence aftereffect and determine whether it contributes to a difference of vertical deviation with respect to gaze distance in patients with vertical strabismus. DESIGN Prospective noncomparative studies. METHODS Eighty-four patients with unilateral superior oblique palsies were enrolled and classified into three types-A, B, and C-based on the difference in vertical deviation between distant and near viewing. The prism adaptation test was performed for 2 to 3 hours to correct vertical deviation and the response of vertical deviation to the prism adaptation test was compared among the three types RESULTS Adaptive vertical vergence aftereffect, defined as an increase of deviation by 5 prism diopters or more with the prism adaptation test, was identified in 13 patients (16%) at distance and in 23 patients (27%) at near viewing. Among the three types, the adaptive vergence aftereffect contributed mostly to the type B, in which distance deviation exceeds near deviation. Nine patients (39%) of type B changed to type A category with the prism adaptation test; and of these, 7 increased near deviation so that the deviation difference between distant and near viewing decreased. CONCLUSION The adaptive vertical vergence aftereffect contributes to a difference in vertical deviation between distant and near viewing. The vertical prism adaptation test is specifically useful to determine the extent of surgery by breaking fusional vergence in patients with hypertropia in whom deviation differs with respect to viewing distance.