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Wednesday, June 8, 2011

Clinical outcome of shoulder muscle transfer for shoulder deformities in obstetric brachial plexus palsy: A study of 150 cases

Clinical outcome of shoulder muscle transfer for shoulder deformities in obstetric brachial plexus palsy: A study of 150 cases Thatte MR, Agashe MV, Rao A, Rathod CM, Mehta R - Indian J Plast Surg Background: Residual muscle weakness, cross-innervation (caused by misdirected regenerating axons), and muscular imbalance are the main causes of internal rotation contractures leading to limitation of shoulder joint movement, glenoid dysplasia, and deformity in obstetric brachial plexus palsy. Muscle transfers and release of antagonistic muscles improve range of motion as well as halt or reverse the deterioration in the bony architecture of the shoulder joint. The aim of our study was to evaluate the clinical outcome of shoulder muscle transfer for shoulder abnormalities in obstetric brachial plexus palsy. Materials and Methods: One hundred and fifty patients of obstetric brachial plexus palsy with shoulder deformity underwent shoulder muscle transfer along with anterior shoulder release at our institutions from 1999 to 2007. Shoulder function was assessed both preoperatively and postoperatively using aggregate modified Mallet score and active and passive range of motion. The mean duration of follow-up was 4 years (2.5-8 years). Results: The mean preoperative abduction was 45? ? 7.12, mean passive external rotation was 10? ? 6.79, the mean active external rotation was 0?, and the mean aggregate modified Mallet score was 11.2 ? 1.41. At a mean follow-up of 4 years (2.5-8 years), the mean active abduction was 120? ? 18.01, the mean passive external rotation was 80? ? 10.26, while the mean active external rotation was 45? ? 3.84. The mean aggregate modified Mallet score was 19.2 ? 1.66. Conclusions: This procedure can thus be seen as a very effective tool to treat internal rotation and adduction contractures, achieve functional active abduction and external rotation, as well as possibly prevent glenohumeral dysplasia, though the long-term effects of this procedure may still have to be studied in detail clinico-radiologically to confirm this hypothesis. Level of evidence: Therapeutic level IV"> 1, Mandar V Agashe1, Aamod Rao2, Chasanal M Rathod3, Rujuta Mehta2"> Indian Journal of Plastic Surgery An open access publication of Association of Plastic Surgeons of India Users Online: 34   Home | Subscribe | Feedback | Login  About Current Issue Ahead of print Archive Search Instructions Online Submission Etcetera Contact   Navigate here    Search       Resource links  ?   Similar in PUBMED  ?  Search Pubmed forThatte MRAgashe MVRao ARathod CMMehta R  ?  Search in Google Scholar for Thatte MR Agashe MV Rao A Rathod CM Mehta R  ?Related articlesBrachial plexus palsycontracturemallet score  ?   Article in PDF (2,295 KB)  ?   Citation Manager  ?   Access Statistics  ?   Reader Comments  ?   Email Alert *  ?   Add to My List * * Registration required (free)     In this article  ?  Abstract ?  Introduction ?  Materials and Me... ?  Results ?  Discussion ?  References ?  Article Figures ?  Article Tables
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   Table of Contents    ORIGINAL ARTICLE Year : 2011  |  Volume : 44  |  Issue : 1  |  Page : 21-28  

Clinical outcome of shoulder muscle transfer for shoulder deformities in obstetric brachial plexus palsy: A study of 150 cases

Mukund R Thatte1, Mandar V Agashe1, Aamod Rao2, Chasanal M Rathod3, Rujuta Mehta2
1 Department of Reconstructive and Plastic Surgery, Bai Jerbai Hospital for Children; Bombay Hospital and Medical Research Institute, Mumbai, India
2 Department of Paediatric Orthopaedics, Bai Jerbai Hospital for Children, Mumbai, India
3 Department of Plastic Surgery, Bombay Hospital and Medical Research Institute, Mumbai, India

Date of Web Publication21-May-2011

Correspondence Address:
Mukund R Thatte
167-F, Dr. Ambedkar Road, Dadar, Mumbai - 400 014
India
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DOI: 10.4103/0970-0358.81441

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 ? Abstract Background: Residual muscle weakness, cross-innervation (caused by misdirected regenerating axons), and muscular imbalance are the main causes of internal rotation contractures leading to limitation of shoulder joint movement, glenoid dysplasia, and deformity in obstetric brachial plexus palsy. Muscle transfers and release of antagonistic muscles improve range of motion as well as halt or reverse the deterioration in the bony architecture of the shoulder joint. The aim of our study was to evaluate the clinical outcome of shoulder muscle transfer for shoulder abnormalities in obstetric brachial plexus palsy. Materials and Methods: One hundred and fifty patients of obstetric brachial plexus palsy with shoulder deformity underwent shoulder muscle transfer along with anterior shoulder release at our institutions from 1999 to 2007. Shoulder function was assessed both preoperatively and postoperatively using aggregate modified Mallet score and active and passive range of motion. The mean duration of follow-up was 4 years (2.5-8 years). Results: The mean preoperative abduction was 45? ? 7.12, mean passive external rotation was 10? ? 6.79, the mean active external rotation was 0?, and the mean aggregate modified Mallet score was 11.2 ? 1.41. At a mean follow-up of 4 years (2.5-8 years), the mean active abduction was 120? ? 18.01, the mean passive external rotation was 80? ? 10.26, while the mean active external rotation was 45? ? 3.84. The mean aggregate modified Mallet score was 19.2 ? 1.66. Conclusions: This procedure can thus be seen as a very effective tool to treat internal rotation and adduction contractures, achieve functional active abduction and external rotation, as well as possibly prevent glenohumeral dysplasia, though the long-term effects of this procedure may still have to be studied in detail clinico-radiologically to confirm this hypothesis. Level of evidence: Therapeutic level IV


Keywords: Brachial plexus palsy, contracture, mallet score


How to cite this article:
Thatte MR, Agashe MV, Rao A, Rathod CM, Mehta R. Clinical outcome of shoulder muscle transfer for shoulder deformities in obstetric brachial plexus palsy: A study of 150 cases. Indian J Plast Surg 2011;44:21-8
How to cite this URL:
Thatte MR, Agashe MV, Rao A, Rathod CM, Mehta R. Clinical outcome of shoulder muscle transfer for shoulder deformities in obstetric brachial plexus palsy: A study of 150 cases. Indian J Plast Surg [serial online] 2011 [cited 2011 May 23];44:21-8. Available from: http://www.ijps.org/text.asp?2011/44/1/21/81441


 ? Introduction Top

Obstetric brachial plexus palsy occurs due to trauma to the roots of the brachial plexus during difficult delivery, [1] or as suggested by some authors, even during intrauterine life. [2] It has been documented in earlier literature that about 75-90% of all obstetric brachial plexus palsies recover spontaneously and completely during the first year of life. However the patients who do not recover spontaneously usually have some permanent damage and the brunt of the damage is borne by the shoulder. [3] Almost all patients with obstetric palsy have involvement of the fifth and sixth cervical nerves and almost 48% of them have involvement of C5-C6 alone. [1] In fact, in the last few decades, it has been proved by many authors that shoulder abnormalities are a rule in patients with chronic brachial plexopathy. [3] These abnormalities develop very early in infancy and are caused by muscle imbalances due to the developing cross-innervation in a growing child. The imbalance is between the more powerful adductor-internal rotator and the relatively less powerful abductor-external rotator group. The resultant contracture results in assymetrical forces acting on the glenohumeral articulation leading to grossly deformed and posteriorly subluxated humeral head with an excessively retroverted glenoid if allowed to act unchecked over the years. [3]

Surgery for the shoulder in brachial plexus palsy is aimed at treating the internal rotation contracture, increasing the range of abduction and external rotation as well as to prevent secondary structural changes in the glenohumeral articulation. [4] Most of the surgeries are modifications of Sever's original description of release of the subscapularis and the pectoralis major. [5] L'Episcopo described a surgery in which he divided the tendinous origin of the teres major, latissimus dorsi, and repositioned them under the lateral head of the triceps thus converting them into external rotators. [6] Various modifications of this technique have been described over the years, such as those by Zachary and Leeds [7] and Phipps and Hoffer, [8] though the basic principle of all the surgeries has remained the same, that is, either releasing, sectioning, or transferring the overacting internal rotators to convert them into external rotators. A newer modification of this technique was suggested by Chuang et al. in 1998 which has since then been widely used at various centers of the world. [9] More recently, the role of injecting botulinum toxin in the hyperacting muscle has been investigated by some authors (Grosmann et al., 2007). It has been shown that injecting the hyper-agonist muscle with botulinum toxin reduces spastic co-contractions and improves resting posture of the muscles. [10],[11]

The present study describes a series of 150 consecutive patients operated at our institutes for secondary shoulder deformities due to obstetric brachial plexus palsy by a combination including anterior shoulder release and tendon transfer. The aims of the present study were to determine the effect of these soft tissue rebalancing procedures on glenohumeral function based on shoulder movements as well as functional grading.


 ? Materials and Methods Top

Between January 1999 to June 2007, 305 patients were evaluated and treated for obstetric brachial plexus palsy at our institute. Of these, 35 had a complete recovery and were treated conservatively, and 169 patients were operated primarily at around 3 months for a primary nerve surgery. One hundred and sixty-three children with obstetric brachial plexus palsy with shoulder deformities and contractures were treated with soft tissue rebalancing surgeries of which 62 had been operated previously for primary nerve surgeries. Thirteen were lost to follow-up, making a total of 150 patients who were included in the study. Most patients were operated on by the senior surgeon (MRT) and about 15 by the other senior author (RM). There were 81 boys and 69 girls. The average age at the time of presentation to our institute was 3 months and the average age for shoulder muscle transfer was 40 ? 20.06 months (range 15 months to 8 years). Of the 150 children, 80 were C5-C6 lesions, 34 were C5-C7 lesions, 32 were C5-T1 lesions, and 4 were C5-T1 lesions with Horner's sign. Exploration, neurotisation, and nerve grafting were performed around the age of 3 months in 62 patients; these included 18 patients with C5-C6 lesions, 15 C5-C7 lesions, 25 C5-T1 lesions, and all 4 patients with global plexus palsy with Horner's sign [Table 1]. The right shoulder was affected in 78 of these patients while the left was affected in 72 patients. Table 1: Distribution of the lesions of brachial plexus palsy

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All patients were evaluated preoperatively according to a fixed protocol designed by the senior author (MRT), which included active and passive shoulder range of motion, presence or absence of Putti's sign, presence or absence of contractures, and an aggregate modified Mallet score (total 25). The aggregate Mallet [4] score is calculated by finding the individual Mallet score for the five components (global abduction, global external rotation, hand-to-spine, hand-to-mouth, and hand-to-neck) and then summing up the individual components. For each component, grade I is given for no motion while grade V denotes normal motion (Equal to contralateral side). The patients were also examined preoperatively for co-contractions between the muscles.

The surgeries were aimed at releasing the contracted muscle and augmenting the weaker but more useful muscle. The surgical technique was therefore a combination of existing historical techniques tailored to the situation in the individual child based on three factors - muscle strength, co-contractions, and concomitant contractures. All patients had shoulder abduction and external rotation weakness with a concomitant internal rotation contracture.

Surgical technique

During the surgical procedure, lateral position was given with the affected arm draped free and able to move in all directions. The surgical incision was infiltrated with 1:1,00,00,00 (one million) concentration of saline adrenaline for a bloodless operative field. This concentration allows much larger volumes to be injected without reducing the quality of vasoconstriction. The first incision was taken on the lateral margin of the scapula in the axilla going across the axillary skin fold to expose and detach the tendons of the conjoined muscles, teres major, and latissimus dorsi from their insertion on the humerus. These conjoined tendons were then elevated upto their neurovascular pedicle found proximally along the lateral border of the scapula. Through a second incision posteriorly over the spine of the scapula, the tendon of the infraspinatus was exposed below the scapular spine after retracting the posterior fibers of the deltoid away. A wide subcutaneous tunnel between the first and second incision was created through which the detached tendon of the teres major was tunneled, and woven on to the tendon of the infraspinatus using 2-0 nylon, keeping the upper limb abducted and externally rotated after the anterior shoulder release was completed as described in the next paragraph.

Keeping the arm abducted, an anterior incision is taken in the delto-pectoral groove to release the shoulder joint. Through this incision, the coraco humeral ligament and if necessary the capsule of the shoulder joint was cut to obtain full external rotation and reduction of the subluxated head in those cases where it was fully subluxated. In some cases, partial excision of the hypertrophied tip of the coraocoid process was done if found to be impinging on the humerus in rotation or abduction. At this stage, botulinum toxin was injected into the subscapularis and the pectoralis major in selected cases depending on the degree of co-contraction felt pre-operatively and a presence of a strong Putti's sign.

After achieving hemostasis, the wound was closed in layers. A plaster spica was applied keeping the arm in abduction above 90? and external rotation with the elbow slightly flexed to hold the rotation more easily and the forearm supinated. This spica was worn for 4-6 weeks after which a controlled rehabilitation program was started by a well-trained physiotherapist which included active and passive range of motion exercises along with strengthening exercises. An extension elbow-guard was used to prevent flexion of the elbow during active shoulder abduction. This brace was removed while doing physiotherapy for external rotation.

After the surgery, the patients were asked to follow-up every 6 weeks for the first 3 months followed by 3 monthly for the first year and half yearly thereafter. During every follow-up visit, the patients were assessed as to their active and passive range of motion, Putti's sign, as well as their aggregate modified Mallet score.

Statistical analysis

Data were entered using Microsoft Excel 2007 and analyzed using SPSS version 16. Since the data were not distributed normally (Kolmogorov-Smirnov test for normality), Wilcoxon signed rank test was used for before and after comparison of different variables. P value of less than 0.05 was taken as significant.


 ? Results Top

All patients showed improvement in shoulder joint function and movement after the surgical procedure as measured by the aggregate modified Mallet score after a mean follow-up of 4 years (range 2.5-8 years) [Figure 1]a-e and [Figure 2]a-e. The pre-operative Mallet score ranged from 8 to 13 with a mean of 11.2 ? 1.41. This score improved to a mean aggregate Mallet score of 19.2 ? 1.66 (range 15-20). No patient had worsening of shoulder function. All factors of the Mallet score (global abduction, global external rotation, hand-to-mouth, hand-to-spine, and hand-to-neck) improved with the maximum improvement being seen in the global abduction and global external rotation (pre-operative mean of 2.0 to a post-operative mean of 4.1 for abduction and pre-operative mean of 2.0 to a post-operative mean of 4.0 for external rotation). The active abduction improved from a pre-operative value of 45 o ? 7.12 to a post-operative value of 120 o ? 18.01, while the passive external rotation improved from 10 o ? 6.79 pre-operatively to 80 o ? 10.26 post-operatively . The active external rotation also showed a similar increase from a pre-operative value of 0 o to 45 o ? 7.12 post-operatively. The details of the pre-operative and post-operative range-of-motion and Mallet scores are provided in [Table 2]. A total of 10 patients had botulinum toxin A injected into their subscapularis and sternal head of pectoralis major. The post-operative Mallet scores of these 10 patients was marginally better than the other patients (19.5 ? 0.52) (P>0.05). However this is not statistically significant at this time.Table 2: Preoperative and postoperative mallet scores and values of abduction and external rotation

Click here to viewFigure 1: (a) Seven years post-operative clinical photograph of a girl operated with anterior shoulder release and latissimus dorsi and teres major transfer. Active external rotation was 80? (Mallet score-IV). Note the healed scar of the anterior shoulder release (blue arrow). (b) Active abduction of around 130? (Mallet score IV). (c) Hand-to-neck Mallet score V. (d) Hand-to-mouth Mallet score V. (e) Hand-to-spine Mallet score IV.

Click here to viewFigure 2: (a) Preoperative clinical photograph of a 14-month-old child with untreated right-sided obstetric brachial plexus palsy (C5-6 lesion) showing inability to abduct the right shoulder with a concomitant internal rotation contracture, (b) Three years post-operative clinical photograph of the same patient depicting excellent clinical function showing abduction (2b), external rotation (2c), hand-to-head (2d), and hand-to-mouth (2e) activities, (c) Three years postoperative clinical photograph of the same patient depicting excellent clinical function showing abduction (2b), external rotation (2c), hand-to-head (2d), and hand-to-mouth (2e) activities, (d) Three years post-operative clinical photograph of the same patient depicting excellent clinical function showing abduction (2b), external rotation (2c), hand-to-head (2d), and hand-to-mouth (2e) activities, (e) Three years postoperative clinical photograph of the same patient depicting excellent clinical function showing abduction (2b), external rotation (2c), hand-to-head (2d), and hand-to-mouth (2e) activities.

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All the increases in the Mallet scores of the respective components were statistically significant at P<0.05. No patient had a decrease or weakness in internal rotation with the hand-to-spine score improving from a mean of 2.4 to a post-operative of 3.8. No post-operative complications like wound infection, axillary nerve, or radial nerve palsy were noted.

Statistical analysis performed using the Wilcoxon Signed rank test with P<0.05.


 ? Discussion Top

It has been well documented in literature [1] that almost 90% of all patients of obstetric brachial plexus palsy recover completely and thus can be treated conservatively. However, this figure is different in this study group with only 35 of the 305 patients recovering completely. The reason for this discrepancy may be the fact that ours is a tertiary referral center and only those patients who do not recover sufficiently are sent to us for further management.

Shoulder deformities in obstetric brachial plexus palsies have been an object of study and debate over a number of years. The cross-innervations of the regenerating nerve fibers lead to significant internal rotation and adduction contractures with profound effect on the glenohumeral development and functional ability of the child. The range of soft tissue and bony implications of this condition is so vast that a "cafeteria" approach has to be adopted for the management of a particular deformity. Current treatment options include anterior shoulder release, tendon transfers, and soft tissue balancing procedures, [3],[4],[12] concomitant use of botulinum toxin A [11] and humeral and glenoid osteotomies. [13] However, there are no clear guidelines for the selection of any of these procedures as well as the ideal timing at which they should be used.

The average age of our patients at the time of surgery was 44 months (range 18 months to 7 years). This was slightly more as compared to that quoted in western literature. In a series by Waters et al., [4] the average age at the time of tendon transfers was 27 months while that in a series by Hoffer et al., [8] it was 28 months. This finding may be due to lack of awareness among other specialties and the fact that there are no nationally or regionally circulated guidelines for the management as well as referral of patients of obstetric plexus palsy to a specialist clinic such as ours.

In our series, the aggregate modified Mallet score was used to evaluate the patients pre- and post-operatively. The intra- and interobserver reliability of the Mallet classification system have been adequately established. [14],[15] All our patients showed satisfactory improvement in the Mallet scores with the score improving from a pre-operative mean of 11 to a post-operative mean of 19.2. This compares well with other similar series which have been previously reported. Waters et al.[4] reported on 23 patients of obstetric palsy treated with tendon transfers and musculotendinous lengthening, in which the average Mallet score improved from 10 to 18.

In recent times, there has been a general trend toward performing bony surgeries in the form of humeral [13],[14] and glenoid [16] osteotomies as salvage procedures for late obstetric palsies with secondary glenohumeral dysplasia. This inference has been put forth due to the assumption that soft tissue procedures may not be able to stop or reverse bony changes in the glenohumeral joint. However this statement may not be true as we have been able to match the results of glenoid as well as humeral osteotomies irrespective of the amount of glenohumeral dysplasia in the form of clinical outcome (Details of some of the important studies mentioned in literature are given in [Table 3]). Pre-operative and post-operative magnetic resonance imaging scanning was possible in only a few of our patients due to financial constraints. However the few in which this was possible showed good improvement in parameters of glenohumeral dysplasia like glenoid version and percentage of humeral head anterior to the transverse scapular line [Figure 3]a and b.Table 3: Summary of various articles showing results of various modifications of tendon transfers and bony procedures for obstetric palsy

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Also there has been a fear that anterior shoulder release as well as transfer of the powerful internal rotators without a compensatory bony procedure would weaken internal rotation. No patient in our series developed weakness in internal rotation. On the contrary, the average hand-to-spine Mallet score improved from 2.4 to 3.8 postoperatively. This improvement may be attributed to better compliance of the patient to physiotherapy and rehabilitative care [4] as well as to meticulous surgical technique to prevent overzealous anterior release and over-lengthening of the subscapularis.Figure 3: (a) Preoperative MRI of a patient with left brachial plexus palsy showing a subluxed humeral head with a glenoid version of 22? on the affected side as compared to a version of 6? on the normal side, (b) MRI of the same patient 3 years after shoulder muscle transfer showing relocated humeral head with a normalized glenoid version

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In a few of our patients, we have used injection of botulinum toxin type A into the pectoralis major and subscapularis as an adjunct to the formal surgical release. The clinical results of this technique were better in these patients though the difference was not statistically significant. It has been suggested by some authors that benefits of botulinum toxin are sustained even beyond the period for which the toxin is recognized to be active. [11] This observation was found to be true in our study also as the results were sustained even 3 years after surgery. This is supposed to be due to improved cortical recruitment of the injured nerves caused by the toxin. [16] It thus appears that botulinum toxin may be effective in improving long-term results though its exact indications, dosage, as well as efficacy may require randomized trials for confirmation.

The points in favor of this study include a large number of patients with a varied age of presentation and surgery, who have been followed up for a significant amount of time with a low attrition rate (less than 10%). All surgeries were performed by a single senior surgeon with a similar technique and a constant treatment protocol was followed. A classification with intra- and interobserver reliability (the Mallet score) [11] was used for evaluation and a well-defined physiotherapy regimen and rehabilitative care was given to all patients.

However the limitations of the present study include the unblinded and noncomparative nature of its treatment modality as well as certain other limitations which are common to other previously described methods. Clinical examination of infants and children is difficult and children's fear of examination, inability to comprehend instructions, and in-coordination due to poorly developed motor function are all hindrances toward accurate clinical examination. Thus clinical examination alone can only be considered as an approximation of the true motor deficit. As a result, any study based solely on clinical findings without any radiological correlation is bound to have some shortcomings. The authors are currently undertaking a study taking into account the clinicoradiological status of the glenohumeral joint as a whole using CT and MRI scans. This study is however limited by the fact that CT and MRI scans preoperatively and postoperatively prove to be a significant financial burden to our patients, many of whom belong to low socioeconomic strata.

In summary, this study indicates that latissimus dorsi and teres major tendon transfers are useful in improving function in chronic brachial plexopathy. Injection of botulinum toxin type A has proved to be an effective adjunct to surgery, though its exact indications still need to be defined. Though bony procedures have found support in recent times, it can be said that glenohumeral changes would still be best treated early with muscle-rebalancing procedures before irreversible changes develop. A CT/MRI based study would be an ideal tool to check the efficacy of such procedures in treating these complex problems of glenohumeral articulation.













   ? References Top
1.Kirkos JM, Kyrkos MJ, Kapetanos GA, Haritidis JH. Brachial Plexus Palsy secondary to birth injuries: Long term results of anterior release and tendon transfers around the shoulder. J Bone Joint Surg Br 2005;87:231-5.  Back to cited text no. 1
[PUBMED]  [FULLTEXT]  2.Alfonso I, Papazian O, Shuhaiber H, Yaylali I, Grossman JA. Intrauterine shoulder weakness and obstetric brachial plexus palsy. Pediatr Neurol 2004;31:225-7.  Back to cited text no. 2
[PUBMED]  [FULLTEXT]  3.Waters PM. Management of shoulder deformities in brachial plexus birth injuries. J Pediatr Orthop 2010;30:S53-6.  Back to cited text no. 3
    4.Waters PM, Bae DS. The early effects of tendon transfers and open capsulorrhaphy on Glenohumeral deformity in Brachial plexus Birth palsy. J Bone Joint Surg Am 2008;90:2171-9.  Back to cited text no. 4
[PUBMED]  [FULLTEXT]  5.Sever JW. Obstetric paralysis, its etiology, pathology, clinical aspects and treatment with a report of four hundred and seventy cases. Am J Dis Child 1916;12:541-78.  Back to cited text no. 5
    6.L'Episcopo J. Tendon transplantation in Obstetric paralysis. Am J Surgery 1934;25:122-5.  Back to cited text no. 6
    7.Zachary RB. Transplantation of teres major and latissimus dorsi for loss of external rotation at shoulder. Lancet 1947;2:757.  Back to cited text no. 7
[PUBMED]    8.Hoffer MM, Phipps GJ. Closed reduction and tendon transfers for transfer for treatment of dislocation of the glenohumeral joint secondary to brachial plexus birth palsy. J Bone Joint Surg Am 1998;80:997-1001.  Back to cited text no. 8
[PUBMED]  [FULLTEXT]  9.Chuang DC, Hae-Shya M, Wei F. A new strategy of muscle transposition for shoulder deformity caused by Obstetric brachial plexus palsy. Plast Recontr Surg 1998;101:686-94.  Back to cited text no. 9
    10.Gracies JM. Physiological effects of Botulinum toxin on spasticity. Mov Disord 2004;19:120-8.  Back to cited text no. 10
    11.Price AE, DiTaranto P, Yaylali I, Tidwell MA, Grossmann JA. Botulinum toxin type A as an adjunct to the surgical treatment of the medial rotation deformity of the shoulder in birth injuries of the brachial plexus. J Bone Joint Surg Br 2007;89:327-9.  Back to cited text no. 11
    12.Nath RK, Paizi M. Improvement in abduction of the shoulder after reconstructive soft-tissue procedures in obstetric brachial plexus palsy. J Bone Joint Surg Br 2007;89:620-6.  Back to cited text no. 12
[PUBMED]  [FULLTEXT]  13.Waters PM, Peljovich AE. Shoulder reconstruction in patients with chronic brachial plexus birth palsy. Clin Orthop Relat Res 1999;364:144-52.  Back to cited text no. 13
[PUBMED]    14.Waters PM, Bae DS. The effect of derotational humeral osteotomy on global shoulder function in brachial plexus birth palsy. J Bone Joint Surg Am 2006;88:1035-42.  Back to cited text no. 14
[PUBMED]  [FULLTEXT]  15.Nath RK, Lyons AB, Melcher BA, Paizi M. Surgical correction of medial rotation contracture in obstetric brachial plexus palsy. J Bone Joint Surg Br 2007;89:1638-44.  Back to cited text no. 15
    16.Bae DS, Waters PM, Zurakowski D. Reliability of three classification systems measuring active motion in brachial plexus birth palsy. J Bone Joint Surg Am 2003;85:1733-8.  Back to cited text no. 16
[PUBMED]  [FULLTEXT]  

    Figures
  [Figure 1], [Figure 2], [Figure 3]
 
      Tables
 [Table 1], [Table 2], [Table 3]

 

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Monday, June 6, 2011

Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals

Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals

Jyotsna Murthy
Department of Plastic Surgery, Sri Ramachandra Medical College and Research Institute, Porur, Chennai, India

Correspondence Address:
Jyotsna Murthy
Department of Plastic Surgery, Sri Ramachandra Medical College and RI, Porur, Chennai-600 116, Tamil Nadu
India
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DOI: 10.4103/0970-0358.81447

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Objective: Palatal fistula is a significant complication following cleft palate repair. The guidelines of management of the palatal fistula is dependent on the type of cleft, site of fistula, condition of surrounding tissue and associated problem. We studied the management and outcome of 194 cleft palate fistula in our institute. Design: We present the descriptive hospital-based study of management of palatal fistula in 194 cleft patients. We have excluded all the syndromic children and children whose anterior palate was not operated as per protocol. Settings : Of 194 cleft palate fistula, 37 had palate repair in our hospital and 157 were refereed with fistula following palate repair.The patients were evaluated by interdisciplinary team and plan of management was decided. Result : Various parameters like types of cleft, site of fistula and management of fistula were studied in all the patients. Fifty-two percent were in unilateral CLP and 30% in bilateral CLP because unilateral CLP is the commonest type of cleft. Postalveolar and hard palate region contributing to 67% of all fistulae, followed by junctional in (9%). Seventy-two percent of fistula were amenable for repair by local available tissue, 28% needed tongue flap due to shortage of tissue. Minor numbers have failure of procedure for fistula closure needing further management. Conclusions : This descriptive study present analysis of management of fistula in our institute. It also reinforces that patient with bilateral cleft lip and palate more likely to have shortage of local tissue needing the local flaps like tongue flap compare to other cleft types. The surgical management of fistula can be combined to tackle the associated problems.

Keywords: Palatal fistula; tongue flap; post alveolar fistula


How to cite this article:
Murthy J. Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals. Indian J Plast Surg 2011;44:41-6
How to cite this URL:
Murthy J. Descriptive study of management of palatal fistula in one hundred and ninety-four cleft individuals. Indian J Plast Surg [serial online] 2011 [cited 2011 May 23];44:41-6. Available from: http://www.ijps.org/text.asp?2011/44/1/41/81447

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One of the expected outcomes of palate repair is to achieve complete partition between nasal and oral cavity in addition to good speech. Any failure of achieving complete structural integrity of palate is labeled as an oronasal (palatal) fistula with persistent passage between oral and nasal cavity. The term, palatal fistula, is normally used for residual non-repaired cleft palate or result of breakdown of repaired palate. [1]

Every visible palatal fistula does not need surgical repair. The indications for a fistula repair depend on the associated symptoms, which are in turn related to the size and location of the fistula. The common symptoms which have been extensively discussed in the literature are: 1. Nasal emission causing speech distortions, 2. Leakage of fluid and food into the nasal cavity leading to poor oral hygiene and foul smell. The timing of the fistula repair depends on symptoms like nasal regurgitation, effect on speech and concerned of the patient. It is generally agreed that fistulae causing disturbances in speech should be repaired as soon as possible. A fistula leading to significant regurgitation of fluid/food into the nose, resulting in inflammation of nasal lining and malodor also needs to be repaired early. However, the repair of a small fistula with no effect on speech and occasional regurgitation of fluid/food into nose, can be delayed to be combined with other procedures. Many attempts have been made to classify palatal fistulae according to the site and is the best presented by Cohen et al. [2] Smith et al, presented modified classification and presented fistula in VII categories according to its site with more details. [3]

The speech evaluation by the speech pathologist is essential to diagnose if the fistula is contributing to the speech problems. The speech pathologist also provides information regarding the associated Velo-pharyngeal incompetence (VPI) which may or may not be due to the fistula after closing the fistula with chewing gum temporarily. [4] Similarly, evaluation and suggestions by an orthodontist are essential for a perialveolar fistula. In certain cases, orthodontic treatment e.g., expansion of arches and removal of deciduous or supernumerary teeth may be necessary. This additional information is vital for combining fistula closure with another procedure like VPI correction, alveolar bone graft or lip revision. The factors which need to be studied and examined before fistula repair are presented in [Table 1]. The concerns and complaints of the patients need to be elicited in detail and to be taken into consideration while planning the management.

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We analyzed the management of palatal fistula in 194 fistulae in our institute since 2002. Of total 194 fistulae, 37 patients had palate repair in our institute, 157 patients were referred to our institute for fistula repair. Of referral patients, 80% were either detected during camps in rural area or referred by the health workers from rural areas and 20% approached directly to our center for help. Our data were collected as alveolar (which also includes prealveolar and postalveolar), hard palate, combined postalveolar extending to hard palate, junction, soft palate and combination of hard and soft palate fistula. In view of this being a retrograde study, we presented the data according to these sites mentioned above. A small uvular notching or bifid uvula was not considered as fistula, but uvular breakdown was considered as soft palate fistula because majority of them will need repair of soft palate. Of 2387 palate repairs in our institute, 2067 patients were followed up at least once postoperatively within 1 year and 75 had fistula. Out of 75 patients, we have excluded 38 patients with small, non-symptomatic postalveolar defect which was not repaired as the protocol (no anterior palate repair with lip repair) in our institute between 2001 and 2003. However, the significant postalveolar fistulae in these patients which needed closure were considered as a fistula and included in the study. This protocol was changed thereafter due to higher percentage of postalveolar fistula needing closure. Hence, 37 patients had cleft palate repair in our institute, needing fistula repair were included in this study. We also excluded syndromic cleft lip and palate patients having associated with cardiac anomalies, ectodermal dysplasia and Pierre Robin sequence with compromised airway. Eight patients with total breakdown of palate with severe shortage of tissue, who needed the simultaneous tongue flap and pharyngeal flaps, [5] were excluded from this study.

As discussed above, the decision for surgical repair of palatal fistula was done after proper evaluation [Table 1]. The previous surgical techniques, scarring and shortage of tissue, inflammation of tissue and oral hygiene, availability of local tissue and concomitant planned procedures were considered to decide the timing and the surgical procedure for the fistula repair. For fistula in hard palate or junctional area, if adequate local tissue was available, it was closed by the mucoperiosteal flaps with releasing incisions like in Von Langenbeck palatoplasty. [6] In large fistulae and/or fistula extending in postalveolar and alveolar region were closed by two-flap technique and preferably with alveolar extended palatoplasty. [7],[8] Alveolar extension palatoplasty (AEP) flaps were extremely useful [Figure 1]a for fistula in postalveolar region. The AEP flaps could be raised cautiously even in the presence of the previous scars between mucoperiosteum flaps and its extension into alveolus [Figure 1]b. However, an interval of 6 months or more between the palate repair and the fistula repair by AEP flaps is necessary. Figure 1a: Incision for alveolar extension palatoplasty
Figure 1b: Fistula repair by alveolar extended palatoplasty

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For the large fistulae with a shortage of tissue and/or severe scarring preventing mobilization of the palatine tissue, an additional tissue was imported as local flaps according to the site of fistula. For postalveolar or anterior hard palate fistulae, where additional tissue was needed, an anteriorly based tongue flap were done [9] [Figure 2]. The tongue flaps provided oral lining, while the nasal lining was repaired with turn-over flaps from the fistula edges (hinge flaps) and this step was of paramount importance to prevent recurrent fistulae. The tongue flaps were routinely divided between 10 and 12 days, postoperatively.

Patients needing VPI correction and/or with a scarred/broken soft palate with fistula had nasoendoscopy for the structural evaluation. In severely scarred and broken soft palate with large nasopharyngeal defect on endoscopy, a superiorly based pharyngeal flap was used to add tissue for the nasal lining for the soft palate which also helps to improve velopharyngeal competence.

In a situation with failure of multiple attempts of fistula repair, refusal for surgery by patients and associated demands for a tooth prosthesis, prosthetic cover for a fistula was used.

We have analyzed the type of cleft, location of fistulae and type of surgeries performed in all the patients.

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We studied distribution of fistula in various types of cleft lip and/or palate as shown in [Table 2]. In our institute, we had comparatively higher fistula occurrence in submucous cleft (6% of total 37 patients). In retrospection, it was noticed that sutures were put in thinner hypoplastic mucoperiosteum, which should have been excised.

The distribution of site of fistula in all 194 palatal fistulae showed most common site is hard palate and in postalveolar region contributing to 67% of all fistulae. This was followed by exclusive postalveolar region (9%) and junctional area [Table 3]. Table 3: Site of fistulae, procedures for fistula repair and adjuvant procedures

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As the majority of the fistula were repaired by different procedure depending on the size of fistula, the availability of local tissue and necessity of adjuvant procedures. The procedures like local flap, Von Lagenback repair, two flap, AEP, palate repair and tongue flap were done to close the fistula. In 65 patients fistula repair was combined with other procedure like VPI correction, ABG and lip revision [Table 3].

Out of total 194 patients had fistula repair, 48% had followed up and 10 patients had residual fistula. All these patients had very poor local condition and oral hygiene. Two of these were operated by AEP flap closure for postalveolar fistula and eight had tongue flap. Of two with AEP flap, one had successful closure with revision surgery while one had prosthetic rehabilitation. Eight out of 54 patients with tongue flap had postoperative problems. Five patients had flap detachment in postoperatively period. Four out of these five patients were resutured in the first 10 days, and two had complete closure while two had persistent fistula. One patient has revision tongue flap after duration of 8 months successfully. Remaining three patients had residual fistula at the posterior border of tongue flap observed during follow-up.

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Although palatal fistula is a common morbidity after cleft palate repair, minimal literature is available regarding the guidelines of management. Palatal fistula is a complication of cleft palate repair observed in different studies ranging from 0 to 34%. [2] We have analysed the descriptive data of type of cleft, site of fistula and management of fistula in our institute. From this data, we learned that smaller fistula are tend to be managed in the same hospital, while the larger fistula are more likely to go to specialized center or tertiary center due to difficulty in management and needing multidisciplinary care. The larger fistulae in difficult site like perialveolar region tend to be neglected and need interdisciplinary care in tertiary centers.

Management of cleft palate fistula is multidisciplinary approach and proper evaluation of speech and dental arch in addition to the local tissues are very essential. The functional aspect of the fistula should be given appropriate attention before a decision is reached on its structural surgical repair. After proper evaluation and decision regarding surgical repair, one needs to pay attention to the local tissue condition. The previous surgical techniques, scarring and shortage of tissue, inflammation of tissue and oral hygiene, availability of local tissue and concomitant planned procedures will decide the type of procedure for the fistula repair [Figure 3]. This approach will help to decide the proper timing and appropriate technique for surgical repair. Tongue flap has been a work horse for difficult palatal fistula with shortage of tissue. Similarly, buccal flaps also known as facial mayo-mucosal flap, is the most appropriate for junction fistulae if there if shortage of tissue. We have used the buccal flap for VPI correction but not for fistula repair. However, if utilized in junctional fistula, this will also help to lengthen the palate for VPI correction at the same time. [10],[11] Other flaps which has been described and utilized very rarely in specific indication are, temporalis muscle flap and free microvascular free flaps. [12],[13] However, the later options are more commonly utilized in closing non-cleft palatal reconstruction.

In certain situation like failure of multiple attempts of fistula repair, refusal for surgery by patients and associated demands for tooth prosthesis, the prosthetic cover for fistula can be applied. However, prosthesis can never provide nature barrier like tissue repair and has implication on oral hygiene and dental health.

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Veau V. Division Palatine. Paris, Masson et Cie, Paris: 1931.  Back to cited text no. 1
Cohen SR, Kalinowski J, LaRossa D, Randall P. Cleft palate fistulas: A multivariate statistical analysis of prevalence, etiology, and surgical management. Plast Reconstr Surg 1991;87:1041-7.  Back to cited text no. 2
[PUBMED]    Smith DM, Vecchione L, Jang S, Ford M, Deleyiannis FW, Haralam MA, et al. The Pittsburgh fistula classification system: A standardized scheme for the e description of palatal fistulas. Cleft Palate Craniofac J 2007;44:590-4.  Back to cited text no. 3
Nanda V, Sharma R, Mehrotra S, Makkar SS, Munjal S. The "chewing gum test" for cleft palate speech. Plast Reconstr Surg 2005;116:18-22.  Back to cited text no. 4
Murthy J. Salvaging procedure for mutilated cleft palate by simultaneous tongue and pharyngeal flap surgery. Plast Reconstr Surg 2008;122:29e-30e.  Back to cited text no. 5
[PUBMED]  [FULLTEXT]  Muzaffar AR, Byrd HS, Rohrich RJ, Johns DF, LeBlanc D, Beran SJ, et al. Papaioannoua: incidence of cleft palate fistula: An institutional experience with two-stage palatal repair. Plast Reconstr Surg 2001;10:1515-8.  Back to cited text no. 6
Denny AD, Amm CA. Surgical technique for the correction of postpalatoplasty fistulae of the hard palate. Plast Reconstr Surg 2005;115:383-7.  Back to cited text no. 7
[PUBMED]  [FULLTEXT]  Carsten MH. Sequential cleft management with the sliding sulcus technique and alveolar extension palatoplasty. J Craniofac Surg 1999;10:503-18.   Back to cited text no. 8
Jackson IT. Use of tongue flap to resurface lip defect and close palatal fistulae in children. Plast Reconstr Surg 1972;49:537-41.   Back to cited text no. 9
[PUBMED]    Abdel-Aziz M. The use of buccal flap in the closure of posterior post-palatoplasty fistula. Int J Pediatr Otorhinolaryngol 2008;72:1657-61.   Back to cited text no. 10
[PUBMED]  [FULLTEXT]  Ashtiani AK, Emami SA, Rasti M. Closure of complicated palatal fistula with facial artery musculomucosal flap. Plast Reconstr Surg 2005;116:381-6; discussion 387-8.  Back to cited text no. 11
[PUBMED]  [FULLTEXT]  Krimmel M, Hoffmann J, Reinert S. Cleft palate fistula closure with a mucosal prelaminated lateral upper arm flap. Plast Reconstr Surg 2005;116:1870-2.  Back to cited text no. 12
[PUBMED]  [FULLTEXT]  Schwabegger AH, Hubli E, Rieger M, Gassner R, Schmidt A, Ninkovic M. Role of free-tissue transfer in the treatment of recalcitrant palatal fistulae among patients with cleft palates. Plast Reconstr Surg 2004;113:1131-9.  Back to cited text no. 13
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[Figure 1], [Figure 2], [Figure 3]

[Table 1], [Table 2], [Table 3] Top

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