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The Bilhaut-Cloquet Procedure for Wassel Types III, IV and VII Thumb DuplicationFrom the Department of Hand Surgery and Peripheral Nerve Surgery, University of Sydney, Royal North Shore Hospital, Sydney, Australia Correspondence: Prof Michael Tonkin, Department of Hand Surgery and Peripheral Nerve Surgery, University of Sydney, Royal North Shore Hospital, St Leonards, NSW 2065, Australia. Tel.: +61 2 9926 7778; fax: +61 2 9926 7774. E-mail:mtonkin{at}surgery.usyd.edu.au
Five cases of Wassel types III, IV and VII thumb duplication underwent a Bilhaut-Cloquet procedure. A stable and mobile metacarpophalangeal joint was achieved in all cases. Interphalangeal joint motion was limited but this joint was stable in all cases. The nail ridge in these thumbs was minimal. A strong, stable thumb of normal size and good appearance can result from the Bilhaut-Cloquet procedure. When one nail is 70% of normal width, a modified procedure using the whole of one nail will avoid the nail ridge, but the nail will still differ from normal.
Key Words: Bilhaut-cloquet • thumb duplication • Wassel classification The ideal reconstruction of thumb duplication aims to produce a thumb that has full motion, normal width and length, normal growth, stable interphalangeal and metacarpophalangeal joints, minimal scarring and minimal nail deformity. Hypoplasia and joint instability compromise these aims. The surgical options fall into two groups: ablation of one of the duplicated thumbs, with or without reconstruction of the other part, and the Bilhaut-Cloquet procedure, or a modification of it, in which parts of both thumbs are combined. The use of the Bilhaut-Cloquet procedure was first described by Bilhaut (1889) when he used two equal parts of a Wassel type I duplication to reconstruct the thumb. Subsequently, this procedure has been used for more proximal Wassel types (Hartrampf et al., 1974; Naasan and Page, 1994; Tada et al., 1983). It has its advocates and its critics. The advantages are that the reconstructed thumb will be of good size with stable joints. The disadvantages are those of a ridged nail and joint stiffness. Restricting the procedure to Wassel types I and II deformities retains good metacarpophalangeal joint motion. This may be compromised if the procedure is used for Wassel types III and IV deformities. However, reconstruction of the retained thumb, which may be unstable and hypoplastic after ablation of the other, may result in too small and weak a thumb, with significant joint instability. Although many reports describe the use of the Bilhaut-Cloquet procedure as part of a general review of thumb duplication surgery, few have documented precise results for the proximal Wassel duplications. This paper presents the indications, techniques and results in five such thumbs.
Inclusion criteria were patients with a thumb duplication of proximal Wassel types (III–VII) in whom each thumb was inadequate alone and the two thumbs were of similar size and shape. Patients with one thumb significantly larger than the other were treated by ablation of the smaller thumb. There were five patients, of average age at surgery 18 (range 9–24) months, with a mean follow-up of 21 (range 13–44) months (Table 1). Clinical examination and plain X-rays were used to assess the patients, pre-operatively and postoperatively.
Operative procedure A zig-zag incision matching volar and dorsal surfaces is made (Fig 1). If the nail of one of the thumbs is more than 70% of the contralateral normal, or predicted, thumb nail width, it is retained in its entirety with excision of the other nail (Dobyns et al., 1985; Evans, 1993). When the nail width is less then 70% of the contralateral normal, or predicted size, the two nail beds are divided and combined to achieve a nail with the equivalent width of the opposite thumb (Fig 1). The extensor mechanism is assessed with regard to abnormal insertion, ensuring that continuity with both distal phalanges is maintained after reconstruction. The conjoint base of a Wassel type III bifid proximal phalanx in case 3 was separated using a beaver blade. The required width of bone is then excised from the proximal and distal phalanges, using a beaver blade or fine osteotome and bone nibblers, to complete the longitudinal osteotomies (Fig 2). The flexor tendon insertions are maintained. The metacarpal is broad and may prevent even apposition of the proximal phalanges along their length. A triangle of bone and cartilage is removed from each side of the metacarpal head, retaining collateral ligament–periosteal continuity, as described previously for the proximal phalanx in Wassel types I and II (Tonkin and Rumball, 1997) (Fig 3). A K-wire (0.7 mm) is used to drill holes in the aligned phalanges, ensuring that the growth plates of each phalanx are matched. Physeal matching is necessary to diminish the possibility of growth deformity. If the joint surface is incongruous, this may be sculpted to a satisfactory shape. Transverse osteotomies may be necessary for triphalangeal thumbs, or when the comparative phalangeal lengths differ, be this in the proximal or the distal phalanges. An attempt is made to retain the periosteum on one side of the transverse osteotomy to improve stability. Either a 30 gauge interosseous wire or 4.0 PDS suture is used for osteosynthesis (Fig 4). The extensor tendons are combined using a 5.0 Vicryl suture. It is rarely necessary to reconstruct the flexor tendons. The nail bed is repaired using an 8.0 Vicryl suture. The skin is closed with 6.0 Vicryl Rapide sutures. One nail is replaced between the nail fold repair and the nail bed repair.
For case 3, the larger nail was retained, being 70% of the width of the opposite thumb nail. Case 5 presented bilateral thumb duplications. One was treated by ablation and reconstruction, the other by a Bilhaut-Cloquet procedure. In cases 1 and 5, both thumbs were triphalangeal (Fig 9). In case 3, one thumb was triphalangeal. Each required transverse osteotomies to match the proximal and distal phalangeal growth plates.
Outcome measures included size of nail and thumb (nail width and pulp circumference), range of motion, joint stability of the interphalangeal and metacarpophalangeal joints and an assessment of appearance, including thumb shape and nail ridging. The grading system described by Tada et al. (1983), which considers joint range of motion, joint stability and alignment to assess the postoperative outcome, was used (Table 2).
Parents were also asked to rate both appearance and function as poor, fair, good or excellent. Radiographic assessment of joint congruity and growth plates was also performed.
In three patients, thumb nail width and pulp circumference were equivalent to the unoperated opposite thumbs (Table 3). In the bilateral case, the Bilhaut-Cloquet thumb pulp and its nail were the larger. Regrettably, it was not possible to obtain these measurements for Case 1.
Full metacarpophalangeal and interphalangeal joint extension was present in all thumbs. Interphalangeal joint flexion was decreased significantly to an average of 13°. For the contralateral thumb, the average range of flexion was 68°. Metacarpophalangeal joint flexion averaged 55°. For the contralateral thumb, the average range of flexion was 75°. The metacarpophalangeal range of flexion was greater in case 2 (type IV) and case 4 (type III) than in the triphalangeal thumbs. Table 4 details individual joint motions for each thumb.
All thumbs were stable at both joints at the time of this review. However, one secondary procedure had already been performed for case 3 (on whom one nail only was used, in combination with unequal portions of the distal phalanges). Instability of the interphalangeal joint ulnar collateral ligament, with a radial deviation of 20°, was corrected by secondary reconstruction of an ulnar collateral ligament. A small retained nail remnant from the ulnar thumb was also excised at the same time. In case 5, with bilateral duplications, the opposite thumb was reconstructed after ablation of the smaller radial thumb. Interphalangeal joint motion was similar to that present in the Bilhaut-Cloquet patients and metacarpophalangeal joint motion differed minimally. However, both joints were unstable and weak in the thumb which did not have the Bilhaut-Cloquet procedure. The nail ridge was judged to be minimal in three of the four combined nails and minor in one. The nail of case 3 was smaller and differed in shape to that of the opposite side. Appearance and function were both rated as good by the four parents available for interview (cases 2–5) (Figs 5–8).
Radiologically, all of the proximal phalanges and distal phalanges had united but with the distal phalanges retaining a bifid appearance. In case 3, the smaller remnant of distal phalanx appeared to bridge the growth plate of the larger distal phalanx (Fig 10). It was not possible to comment on the growth plates of case 2. In the other three thumbs, the alignment was symmetrical.
Wassel (1969) described his classification and, retrospectively, reviewed 70 cases of radial polydactyly, nine of which were bilateral. All of these patients were treated by ablation. However, he commented that the Bilhaut-Cloquet procedure may be a better option, especially where there was divergence of the duplication from the longitudinal axis. The Bilhaut-Cloquet procedure has been used for the correction of proximal Wassel thumb duplications but, despite a number of large retrospective studies (Andrew and Sykes, 1988; Naasan and Page, 1994; Tada et al., 1983), details of the operative technique and the results have not been described formally. The Bilhaut-Cloquet procedure was performed by Hartrampf et al. (1974) for Wassel type III and IV thumbs. Their paper includes clinical photographs but no data, or discussion, about joint movement or nail ridging. The authors comment that there were no changes in growth at three year follow up because of the removal of the median portion of the phalanges, including the epiphysis. An exhaustive retrospective study was performed by Tada et al. (1983), who reviewed 130 of 193 operated hands. Ninety-four of the hands reviewed were assessed following primary surgical reconstruction. Thirty-six hands were assessed following secondary surgery for residual deformity. Of 125 primarily operated cases, the majority were treated by resection and joint reconstruction, but seven out of 73 Wassel types III and IV thumbs were treated by a Bilhaut-Cloquet procedure. The authors stopped using this method because of reduced ranges of motion of the interphalangeal and metacarpophalangeal joints. However, they did report that the resulting thumb was of a more normal size. We employed the same grading system used by Tada and his colleagues to grade their series, resulting in four cases being classified as good and one as fair. We believe that good metacarpophalangeal joint motion was achieved by careful alignment of the epiphysis and fashioning the joints to ensure joint congruity, both of the metacarpal and phalanges. Even case 3, with the lowest score, had good motion at the metacarpophalangeal joint and good function (Table 4). The disadvantage of diminished interphalangeal joint motion is a lesser problem than joint instability. All but one joint of one thumb in this series remained stable after the primary surgery. Case 3 required revision for interphalangeal joint radial collateral instability. The other criticism of the Bilhaut-Cloquet procedure is the ridging of the nail after coaptation of the two halves (Fig 7). This must be balanced against producing a nail which is too small, if using the alternative surgical technique. We agree that, if one of the thumbs has a nail of greater than 70% of normal, it should be used in its entirety without combining it with the other side, as suggested by others (Dobyns et al., 1985; Evans, 1993). However, the decision to do this does create some difficulties of technique. If a small and unequal portion of distal phalanx is attached to a larger distal phalanx, retaining the collateral ligament, physeal and joint matching is compromised. If only pulp is used, interphalangeal joint collateral ligament stability may be compromised. In neither instance is the nail ‘normal’. If the nails are too small we believe that combination is necessary. The nail ridge obtained in the cases in which this was done in this small series was minimal, as has been our experience following reconstruction of type I and II cases (Tonkin and Rumball, 1997).
The postoperative phalanges in cases 4 and 5 resulted in radiologically bifid distal, although united phalanges. The possible reasons for this are inadequate bone excision, inadequate apposition or inadequate strength of the osteosynthesis. We advise the use of interosseous wire for osteosynthesis and advise careful attention to ensure adequate bone resection and apposition. Excision of bone from each side of the metacarpal head aids in obtaining satisfactory apposition. Despite the radiographic findings, both patients had good functional and cosmetic results. Treatment of thumb duplication has a high incidence of secondary surgery, irrespective of the method of correction. Of the 43 thumbs reviewed by Naasan and Page (1994), 49% had secondary deformities and, of the four Wassel types III and IV that were corrected with Bilhaut-Cloquet procedures, three underwent further procedures. These authors concluded that the incidence of secondary surgery was related to the complexity of the initial deformity. Of the 12 Wassel type III and IV thumbs reviewed by Andrew and Sykes (1988), none of which were treated by a Bilhaut-Cloquet procedure, 10 had second operations. The retrospective review of Tada et al. (1983) detailed 36 cases undergoing secondary surgery for deformity. Overall, the high incidence of secondary surgery would appear to be associated with the problems of hypoplasia and instability which occur in thumb duplication. The results of these series, and the contralateral thumb of our Case 5, would suggest that these may be more likely to remain as a source of problems after ablation and reconstruction as in case 5 (Fig 8c). One secondary procedure was performed in our five cases. This followed a modified Bilhaut-Cloquet (case 3), to correct instability and deviation at the interphalangeal joint. As the follow-up of these five patients is short, it is not possible to rule out the need for future surgery. However, the joint stability and lack of deformity after a mean follow-up of 21 (range 13–44) months of growth since surgery is encouraging.
Manuscript received November 28, 2005. Accepted for publication May 31, 2007.
Andrew JG, Sykes PJ (1988). Duplicate thumbs: a survey of results in twenty patients. Journal of Hand Surgery, 13B: 50–53.[CrossRef][Medline] [Order article via Infotrieve]Bilhaut M (1889). Guerison d’un pouce bifide par un nouveau procede operatoire. Congrés Francais de Chirurgie, 4: 576–580 [Translated into English by Tonkin HE, Tonkin MA (1997). Hand Surgery, 2: 75–77.].Dobyns JH, Lipscomb PR, Cooney WP (1985). Management of thumb duplication. Clinical Orthopaedics, 195: 26–44.Evans D (1993). Polydactyly of the thumb. Journal of Hand Surgery, 18B: 3–4 [Editorial].Hartrampf CR, Vasconez LO, Mathes S (1974). Construction of one good thumb from both parts of a congenitally bifid thumb. Plastic and Reconstructive Surgery, 54: 148–152.[Web of Science][Medline] [Order article via Infotrieve]Naasan A, Page RE (1994). Duplication of the thumb. A 20-year retrospective review. Journal of Hand Surgery, 19B: 355–360.Tada K, Yonenobu K, Tsuyuguchi Y, Kawai H, Egawa T (1983). Duplication of the thumb. A retrospective review of two hundred and thirty-seven cases. Journal of Bone and Joint Surgery, 65A: 584–598.Tonkin MR, Rumball KM (1997). The Bilhaut-Cloquet procedure revisited. Hand Surgery, 2: 67–74.[CrossRef]Wassel HD (1969). The results of surgery for polydactyly of the thumb. Clinical Orthopaedics, 64: 175–193.
Journal of Hand Surgery (European Volume), Vol. 32, No. 6,
684-693 (2007)
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Abstract
PATIENTS AND METHODS
