A 16-year-old girl presented with left eye large-angle exotropia. On examination, we found bilateral limitation of adduction. CT orbit showed hypoplastic medial rectus bilaterally, but intraoperatively we found absent medial recti on both sides. This case report explains discrepancy between the imaging and the intraoperative findings and discusses the management dilemma in view of the risk of anterior segment ischaemia and how marked exodeviation and adduction limitation was tackled by the new technique of transposition myopexy, a modification of the procedure described by Nishida along with recession of lateral rectus to achieve good alignment. This procedure changes the vector forces of the vertical rectus without splitting or tenotomy of the muscles.

/pdf/bilateral-medial-rectus-aplasia-and-a-modified-surgical-34261q501h.pdf

Bilateral medial rectus aplasia and a modified surgical approach of transposition myopexy of vertical recti.

stitution resulting in a Glu213Lys amino acid change. The exact effect of the amino acid substitution, from glutamic acid (which has a negative charge at physiologic pH) to lysine (which has a positive charge), is unclear. However, a similar missense mutation in hemoglobin (specifically a glutamic acid to valine substitution) retards ionic cross-linking and results in altered tertiary protein structure to yield, most famously, the “sickling” of erythrocytes characteristic of sickle cell anemia. Dysfunction of bestrophin likely indirectly impairs apical fluid transport. This then indirectly impairs RPE phagocytosis of photoreceptor outer segments, lysosomal function, and regulation of subretinal fluid, yielding the characteristic vitelliform lesions and serous retinal detachments characteristic of BVMD. Similarly, the phenotypic severity of the siblings we describe, particularly serous retinal detachments and retinoschisis, suggests the mutation they harbor grossly affects chloride transport and Ca 2 signaling, both thought to underlie RPE ionic transport and fluid homeostasis. In summary, we herein present 2 sibl ings with BVMD, both exhibiting a previously unreported missense mutation in BEST1 as well as the novel findings of retinoschisis and a fullthickness macular hole.

/pdf/congenital-bilateral-aplasia-of-medial-recti-in-a-family-4x9brtcl72.pdf

Congenital bilateral aplasia of medial recti in a family.

Reports of isolated anomalies of the medial rectus (MR) muscle in literature are sparse. It has been identified as a subtype of congenital cranial dysinnervation disorder that affects the normal development of brainstem motor neurons. Herein, we report a 37-year-old male presented with large-angle exotropia since the birth of right eye with palpebral fissure widening. On examination of ocular movements, there was -6 limitation of adduction. There was no limitation in other ocular movements. In the preoperative CT scan, all extraocular muscles were present. He underwent surgery in right eye. Intraoperatively in the site of medial rectus, we found an empty sheath without muscle fibers indicating medial rectus hypoplasia. The width of muscle insertion was normal. Surgery consisted of lateral rectus muscle recession 10 mm in hang-back method and vertical muscle transposition procedure, by a modification of Nishida technique, in which the vector of superior and inferior recti was transposed medially by inserting non-absorbable sutures at nasal margins of muscles secured to sclera 8 mm posterior to medial rectus site without tenotomy or splitting. The deviation was decreased to less than 10 PD exotropia in primary position. The adduction was improved from -6 to -4. The palpebral fissure asymmetry was also corrected. Here, we also reviewed clinical features of all cases of medial rectus hypoplasia/aplasia in the literature and discussed surgical approaches. For vertical rectus transposition and horizontal muscle weakening, this technique has the advantages of being simpler and less traumatic to ocular tissues and unlike the traditional transposition procedures, there is no need for tenotomy and splitting.

Minimal invasive vertical muscle transposition for the treatment of large angle exotropia due to congenital medial rectus hypoplasia: Case Report and Literature Review.

Abnormalities in extraocular muscle insertions, such as bilateral bifid superior rectus insertion, have been reported in albinism; however, abnormalities of the inferior oblique insertion have not been described previously. We report abnormal inferior oblique insertions in 4 patients with oculocutaneous albinism. The oblique muscle insertions were more anterior than expected, and traversed the pathway of the lateral rectus recession. We also observed the distance between the limbus and horizontal rectus muscle insertions to be consistently shorter than the distance in patients without albinism.

Abnormal insertional pattern of the inferior oblique in four cases of oculocutaneous albinism and abnormal anterior insertions of horizontal rectus muscles in albinism.

ABSTRACT Introduction: We report a case of large angle exotropia in a child with limitation of adduction in the left eye with a radiological finding of hypoplastic medial rectus. Methods: A 3- year- old male child presented with left eye large angle exotropia, left face turn, -4 adduction limitation and severe amblyopia. Orbital imaging revealed hypoplasia of the medial rectus and intraoperatively a thin medial rectus was noted. The surgical procedure planned was lateral rectus recession combined with Modified Nishida’s technique in the left eye. In this technique the superior and inferior recti were transposed medially by inserting non-absorbable sutures in the sclera posteriorly, closer to the upper and lower borders of the medial rectus muscle. Result: There was improvement in adduction of left eye and reduction of original deviation following maximal lateral rectus recession and a modified Nishida’s approach. The early and optimal correction of exotropia also improved the compliance to patching with subsequent gain in visual acuity of the amblyopic eye. Conclusion: Modified Nishida’s technique has the advantage of no muscle splitting and no tenotomy, remains a less invasive surgical procedure to correct large deviations. The modification of placing the bellies closer to medial rectus augments the effect and further improves adduction in cases with severe limitation of adduction. This technique can thus be considered as a possible surgical approach in young children with large angle exotropia due to hypoplastic medial rectus.

Modified Nishida’s procedure in a case of medial rectus hypoplasia

Large-angle congenital exotropia due to ‘absent’ medial recti: a case of congenital cranial dysinnervation disorder

/pdf/large-angle-congenital-exotropia-due-to-absent-medial-recti-2nfalimoij.pdf

Large-angle congenital exotropia due to 'absent' medial recti: a case of congenital cranial dysinnervation disorder.

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Large-angle congenital exotropia due to 'absent' medial recti: a case of congenital cranial dysinnervation disorder.