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Session 9: Brown’s syndrome and congenital fibrosis syndrome

Transactions 29th European Strabismological Association Meeting – de Faber (ed) © 2005 European Strabismological Association, ISBN 04 1537 211 9

Surgical findings in Brown’s syndrome

Serpil Akar, Birsen Gökyig˘it, Kemran Gök & Ömer Faruk Yılmaz

.

Beyog˘ lu Eye Education and Research Hospital, Istanbul, Turkey

ABSTRACT:

Purpose: Investigation of the results of the surgery applied to patients with Brown’s syndrome. Material and Methods: The files of 51 patients with Brown’s Syndrome, who were examined between 1991–2003 at the Pediatric Ophthalmology and Strabismus Department of our hospital, were retrospectively studied. This study was conducted on 18 patients who had been operated. The subjects consisted of 10 female and 8 male patients with mean follow up being 28 months. Findings: 19 eyes of 18 patients were operated. In 5 cases superior oblique tenotomy, in 4 cases elongation with superior oblique silicon expander, in 9 cases inter capsular tenotomy and elongation with non-absorbable suture were performed. When 1–10 scale was used for operation results, 17 of 19 eyes (89.5%) had successful results with 7 and above score.

Results: Operation techniques used, gave successful results. Elongation with suture had both stable successful results in controls and was simple and may be preferred.

1INTRODUCTION

In 1950, Harold Brown described superior oblique tendon sheath syndrome with a series of 8 cases which had positive traction test together with adduction elevation restraint. It was pointed out that the cause of this syndrome was the shortening of superior oblique tendon sheath (Crawford 1980).

In literature, in the surgical treatment of Brown’s syndrome, different procedures with different results are given (Dyer 1970, Parks 1987, Wright 2000). In this study, our purpose was the treatment of Brown’s syndrome with different surgical techniques, examination of the results and to determine which technique was the most effective.

2MATERIAL AND METHODS

The files of 51 patients with Brown’s syndrome, who were examined between 1991–2003 at the Pediatric Ophthalmology and Strabismus Department of our hospital, were retrospectively studied. This study covers 18 patients who had surgical treatment due to manifested head position or hypotropia (Wright 2000). One eye of the 17 patients and two eyes of the 1 patient were operated.

Prior to surgeries, all patients underwent full ophthalmologic and orthoptic examination. In 5 patients (Group I) superior oblique tenotomy, in 4 patients (Group II), elongation of superior oblique with silicone expander , in 10 eyes of 9 patients (Group III) elongation with non-absorbable suture bridge and inter capsular tenotomy were performed. Suitable horizontal surgery was done on patients with horizontal deviations before, during or after superior oblique surgery. Results were evaluated according to these groups. Surgical results were graded according to 1–10 scale (Wright 2000). Post-operative findings in their last examinations were the basis for this grading.

The mean follow-up was 28 (10–97) months. Mean follow-up for the Group III who had inter capsular tenotomy and elongation with non-absorbable suture was 9,33 (6–15) months.

We used Wilcoxon Signed Ranks tests for statistical evaluations.

121

Table I. Clinical findings Group I (Superior oblique tenotomy).

 

 

Pre

 

Pre

Pos

 

Pos

 

 

Pos

 

 

 

 

Surg

El

Pre

Dev

El

Pos

Dev

Pos

Pos

Bin

Add

 

 

No. Ind

Ad

Fd

(pd)

Ad

Fd

(pd)

Abp

Sop

(sec)

Surg

Fp

Fs

 

 

 

 

 

 

 

 

 

 

 

 

 

 

1

Face turn

4

4

Lho4E6

3/0

0

16H/4H

No

Mod

No

IO SR Rec.

96

7

2

Face turn

4

4

E40

0

0

5H

Small

Small

No

19

7

3

Chin elev

4

4

Lho2E4

2

0

ortho

Yes

No

40

No

6

3

4

Chin elev

3

4

Rho4E10

1

0

E10

Small

No

40

No

24

7

5

Face turn

4

4

Ortho

1

0

ortho

No

No

No

No

48

8

 

 

 

 

 

 

 

 

 

 

 

 

 

 

Sur Ind: Surgical Indication, Pre:Preoperative, Pos:Postoperative, El ad: Elevation adduction, Fd: Forced Duction, Dev: Deviation in primary position, ho: hypotropia, E: Esotropia, X:Exotropia, Abp: Abnormal head Position, Sop; Superior Oblique palsy, Bin; Postoperative Binocularity, Add surg; Re-operation, Fp; Follow period,

Fs: success score (1–10)

Table II. Clinical findings in Group II (Superior oblique tendon expander procedure).

 

 

Pre

 

Pre

Pos

 

Pos

 

 

Pos

 

 

 

 

Surg

El

Pre

Dev

El

Pos

Dev

Pos

Pos

Bin

Add

 

 

No.

Ind

Ad

Fs

(pd)

Ad

Fs

(pd)

Abp

Sop

(sec)

Surg

FP

FS

 

 

 

 

 

 

 

 

 

 

 

 

 

 

1

Chin elev

4

4

Lho6E45

1

0

3Ho

No

No

No

No

15

8

2

Chin elev

4

4

Ortho

0

0

Ortho

No

No

600

No

17

10

3

Face turn

4

4

Lho4

0

0

Ortho

No

No

No

*SO sut.el.

12

1

4

Chin elev

4

4

Rho7

0

0

Ortho

No

No

1200

No

15

10

 

 

 

 

 

 

 

 

 

 

 

 

 

 

*Superior Oblique suture elongation procedures*.

2.1.1 Surgical Techniques

Superior oblique tenotomy was done to superior rectus from nasal, as Parks has explained (Parks1987).

Superior oblique tendon expander procedure was done as Wright (1991) has explained. between the tips of the tendon cut from the 2–3 mm superior rectus nasally, 5.5–6 mm segment of No. 40 medical grade silicone retinal band is attached.

In intercapsular tenotomy and elongation with non-absorbable suture technique, following temporal conjunctiva incision, superior oblique tenotomy was done to superior rectus from nasal, as Parks (1987) has explained . During this procedure the base of the superior oblique capsule was protected. Between the tips of the tendon cut from the 2–3 mm superior rectus nasally, 5.5–7 mm segment was attached. This elongation procedure was done like braiding with 5–0 Dacron nonabsorbable suture. Tendon capsule and conjunctiva was stitched with 8–0 vicryl.

3FINDINGS

Surgical treatment was done on one eye of the 17 patients and two eyes of the 1 patient with the Brown’s syndrome. 12 of the 18 cases were congenital and 6 cases were acquired as patient’s story (cause of 2 case was trauma, cause of four cases was inflammation). Surgeries in these cases were done after at least a year’s follow-up. The subjects consisted of 10 female and 8 male patients with mean age being 7.96 (4–21) years.

It can be seen preoperative and postoperative clinical findings in Group I, Group II, Group III (Table I)(Table II)(Table III). There are statistically significant difference between preoperative and postoperative restriction elevation adduction and maximum hypotropia angle for Group I, Group II, Group III (p0.04, p 0.05), (p 0.05, p 0.04), (p 0.004, p 0.001).

122

Table III. Clinical findings in Group III (Superior oblique tenotomy and elongation with non absorbable suture bridge procedure).

 

 

Pre

 

Pre

Pos

 

Pos

 

 

Pos

 

 

 

 

Surg

Ad

Pre

Dev

Ad

Pos

Dev

Pos

Pos

Bin

Add

 

 

No.

Ind

El

Fd

(pd)

El

Fd

(pd)

Ahp

Sop

(sec)

Surg

Fp

Fs

 

 

 

 

 

 

 

 

 

 

 

 

 

 

1

Chin Elev

4

4

Ortho

2

0

Ortho

Small

No

No

No

7

7

2

Face turn

4

4

Rho12X30

4/0

4/0

X2

No

No

120

*Exp

6

8

 

Hypo

 

 

 

 

 

 

 

 

 

Adh.Liz

 

 

3

Face Turn

4

4

Lho4

3/0

3/0

LH4

No

No

200

No

12

7

4

Chin Elev

3

4

Ortho

3/ 2

3/0

Ortho

No

No

600

*Expl.

8

7

 

 

 

 

 

 

 

 

 

 

 

Adh Liz

 

 

5

Face Turn

4

4

Rho4E25

1

0

E4

No

No

No

No

15

9

6

Chin Elev

3

3

E10

0

0

Ortho

No

No

No

6

10

7

Face Turn

4

4

Lho2X10

0

0

Ortho

No

No

No

6

10

8

Hypo

4

4

Lho10E40

0

0

Ortho

No

No

3000

No

6

10

9

Chin Elev

4

4

E18

0

0

Ortho

No

No

600

**Acc.

14

10

 

 

4

4

 

0

0

 

 

No

 

So releas.

 

10

 

 

 

 

 

 

 

 

 

 

 

 

 

 

*Exploration and adhesion lizis, **Accessory superior oblique realesed.

When superior oblique tenotomy surgery final results in 5 patients based on 1 to 10 scale (10 being best); 4 of the 5 patients (80%) had 7 and above scores. In 3 of these 5 patients (60%) had 7 and above score with one surgery and 1 patient needed a second operation for secondary superior oblique paralysis (Table I).

When superior oblique tendon expander surgery final results in 4 patients based on 1 to 10 scale (10 being best); 3 of the 4 patients (75%) had 7 and above scores. With this surgery, results in 1 patient were unsuccessful and needed a second operation for secondary superior oblique. Silicon was removed and superior oblique tendon was elongated by non-absorbable suture bridge. (Table II).

When elongation with non-absorbable suture bridge surgery final results in 9 patients based on 1 to 10 scale (10 being best); all of the 10 eyes (100%) had 7 and above scores. In 7 of these 10 eyes (70%) had 7 and above score with one surgery and 3 patients needed a second operation. Under correction was due to adhesion in 2 cases and surgically their lizis was necessary. In case 9 which was bilateral an accessory superior oblique tendon was found in both eyes. These accessory tendons were realesed (Table III).

4DISCUSSION

Brown, in 1950, defined superior oblique tendon sheath (Brown) syndrome and since than the surgical treatment of the syndrome had many modifications and changes (Dyer 1970, Parks 1987, Wright 2000).

In early reports, in surgical treatment of Brown’s syndrome, the most preferred method was tenotomy and tenectomy. Sprunger and von Noorden(1991) defend SO tenectomy as a most effective starting procedure. Some other authors state that tenotomy and tenectomy results were successful in 44–50% ratio and additional surgery is required in 50–56% cases (Parks 1987, Sprunger 1991, von Noorden 1982, Wright 1992). In our series, we obtained 60% success after SO tenotomy with one operation. In 1 (20%) case additional surgery was needed. Uncontrolled separation of tendon tips is the major problem of tenotomy and tenectomy and cause consecutive UO paralysis. In different publications, ratio of development of post-operative consecutive superior oblique paralysis after these operations is given as 30–85% (Crawford 1980, Parks 1987, Sprunger 1991,von Noorden 1982, Wright1992). In our series ratio of development of superior oblique paralysis was 40%.

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Another method suggested in the treatment of Brown’s syndrome is superior oblique tendon elongation reported by Wright in 1991. This procedure provides controlled elongation of superior oblique tendon thus reducing the incidence of post-operative paralysis and residual over reaction (Wright 1992). By applying this procedure to his 15 Brown’s syndrome cases and follow-up 1–134 months, Wright has stated that success rate was 87% with one surgery (success result scores were 7–10 based on 1–10 grading scale). 39 pediatric ophthalmologists, members of AAPOS, reported same success based on same scoring system, on 65% of 140 Brown’s syndrome patients who had superior oblique tendon procedure (Wright 2000). We also obtained successful results with one surgery on 75% of the 4 cases using the same procedure. Clarke(1995) and Stager(1999) have obtained very successful results in their cases with silicon tendon expander procedure.

Other method suggested in the treatment of Brown’s syndrome is to place a suture bridge between the cut tips of tendon after the superior oblique tenotomy. Thus, superior oblique tendon can be loosened without causing an important superior oblique paralysis (Wilson 1995). Dyer(1970) has reported that by using suture bridge tips of the cut tendon can be held apart and the surgery results are successful. On the other hand Wright(2000) stated that suture bridge is not rigid enough to keep the cut tips apart, sutures can have scaffolding effect for fibrosis, cut tips will again come together and it would result in under correction. We have applied superior oblique tenotomy and elongation with nonabsorbable suture technique to 10 eyes of 9 patients in our series. It was successful with one surgery with 70% of patients (scored 7–10). There was under correction in 30% and re-operation was necessary. Under correction was due to adhesion in 2 cases and surgically their lizis was necessary. In case 9 which was bilateral an accessory superior oblique tendon was found in both eyes. These accessory tendons were realesed. Following re-operation their post-operative versions became normal and 7–10 success scores are obtained in all cases. In our study, we have followed our patients 9,33 (6–15) months. We think that there is need for a longer period to evaluate the stability of the results.

5CONCLUSION

All our results were successful with the surgical techniques we have applied. We suggest the use of elongation with suture technique, because of its results being favorable and also it being simple.

REFERENCES

1.Clarke MP, Bray LC, Manners T. 1995. Superior oblique tendon expansion in the management of superior oblique dysfunction. Br J Ophthalmol 79:661–3.

2.Crawford JS, Orton RB, LabowDaily L. 1980. Late resuls of superior oblique muscle tenotomy in true Brown’s syndrome. Am J Ophthalmol 89: 824–9

3.Dyer JA. 1970. Superior oblique tendon sheath syndrome. Ann Ophthalmol 2:790–2.

4.Parks MM, Eustis HS. 1987. Simultaneous superior oblique tenotomy and inferior oblique recession in Brown’s syndrome.Ophthalmology 94:1043–8.

5.Sprunger DT, von Noorden GK, Helveston EM. 1991. Surgical results in Brown syndrome. J. Pediatr Ophthalmol Strabismus 28:164–7.

6.Stager Jr DR, Parks MM, Stager Sr DR, et al. 1999. Long-term results of silicone expander for moderate and severe Brown syndrome (Brown syndrome “plus”). JAAPOS 3:328–32.

7.Von Noorden GK, Olivier P. 1982. Superior oblique tenectomy in Brown’s syndrome. Ophthalmology 89:303–8.

8.Wilson ME, Sinatra RB, Saunders RA. 1995. Downgaze restriction after placement of superior oblique tendon spacer for Brown’s syndrome. J Pediatr Ophthalmol Strabismus 32:29–34.

9.Wright KW.1991. Superior oblique silicone expander for Brown’s syndrome and superior oblique overaction. J Pediatr Ophthalmol Strabismus 28:101–7.

10.Wright KW, Min BM, Park C. 1992. Comparison of superior oblique tendon expander to superior oblique tenotomy for the management of superior oblique overaction and Brown syndrome J Pediatr Ophthalmol Strabismus 29:92–9.

11.Wright KW. 2000. Results of the superior oblique tendon elongation procedüre for severe Brown’s syndrome. Tr Am Ophth Soc 98:41–50.

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