The initial search generated in total 428 studies. After the removal of duplicates, two reviewers independently screened titles and abstracts of 423 studies, of which 371 studies were excluded due to predefined inclusion and exclusion criteria. Of the remaining 52 full texts reviewed, 29 studies were excluded: of which 18 were reviews or surgical technique descriptions without information on the outcome (1, 2, 14, 19, 23–36), 6 thereof were case reports(37–42), two studies were written in another language than English, German or French(43, 44), two studies reported on findings from the same study population (45, 46), and 1 study was excluded as it involved animals(47).
Eventually, 23 studies published between 1988 and 2021 were included in our systematic review (Table 1), including patients from Germany(48–54) (30%), Japan(13, 55–57) (17%), United Kingdom(58–60) (13%), France(11, 61–63) (17%), China(64) (4%), USA(15) (4%), Australia(65) (4%), Sweden(66) (4%), and India(67) (4%). All studies were retrospective case series. The Modified Coleman Methodology Score of these studies ranged from 25 to 47 (Additional file 1).
Table 1
Characteristics of included studies
Author (year)
|
Country
|
Number of patients (digit elongations)
|
Study design
|
Average follow up time [months]
|
Treatment method
|
Buck-Gramko (1990)
|
Germany
|
40 (69)
|
retrospective
|
36
|
non-vascularized toe to hand transfer
|
Cavallo (2003)
|
United Kingdom
|
22 (64)
|
retrospective
|
59
|
non-vascularized toe to hand transfer
|
Deutinger (1989)
|
Germany
|
29 (62)
|
retrospective
|
-
|
web reconstruction
|
Foucher (2001)-V
|
France
|
58 (65)
|
retrospective
|
62
|
vascularized toe to hand transfer
|
Foucher (2001)-D
|
France and Italy
|
41 (41)
|
retrospective
|
8
|
Distraction osteogenesis
|
Garagnani (2012)
|
United Kingdom
|
40 (126)
|
retrospective
|
122
|
non-vascularized toe to hand transfer
|
Gohla (2005)
|
Germany
|
48 (113)
|
retrospective
|
72
|
non-vascularized toe to hand transfer
|
Hierner (1998)
|
Germany
|
5 (9)
|
retrospective
|
12
|
distraction osteogenesis
|
Iba (2012)
|
Japan
|
2 (3)
|
retrospective
|
49.5
|
web reconstruction
|
Hulsemann (2002)
|
Germany
|
11 (22)
|
retrospective
|
64
|
vascularized toe to hand transfer
|
Kawabata (2018)
|
Japan
|
29 (54)
|
retrospective
|
89
|
non-vascularized toe to hand transfer
|
Leca (2008)
|
France
|
3 (7)
|
retrospective
|
84
|
non-vascularized and vascularized toe to hand transfer
|
Li (2013)
|
China
|
34 (120)
|
retrospective
|
12
|
web reconstruction
|
Lister (1988)
|
USA
|
12 (12)
|
retrospective
|
48
|
vascularized toe to hand transfer
|
Mann (2016)
|
Germany
|
60 (71)
|
retrospective
|
-
|
distraction osteogenesis
|
Matsuno (2004)
|
Japan
|
15 (23)
|
|
59
|
distraction osteogenesis
|
Miyawaki (2002)
|
Japan
|
4 (7)
|
unclear, retrospective?
|
-
|
distraction osteogenesis
|
Nikkhah (2016)
|
United Kingdom
|
12 (19)
|
retrospective
|
78
|
Vascularized toe to hand transfer
|
Richardson (2003)
|
Australia
|
13 (18)
|
retrospective
|
-
|
vascularized toe to hand transfer
|
Sabapathy (2021)
|
India
|
19 (40)
|
unclear
|
53
|
non-vascularized toe to hand transfer
|
Schenker (2007)
|
Sweden
|
8
|
retrospective
|
57
|
Vascularized toe to hand transfer
|
Unglaub (2006)
|
Germany
|
20 (56)
|
retrospective
|
42
|
non-vascularized toe to hand transfer
|
Van Holder (1999)
|
France
|
14 (28)
|
retrospective
|
-
|
vascularized toe to hand transfer
|
A total of 539 patients comprising 1037 digital corrections were included. Patient inclusion period was 50 years in total and ranged from 1969 to 2019. The mean age of patients was 46 months at the time of operation, and 36% thereof were female. 8 studies(13, 50–52, 55, 64–66) described outcomes on symbrachydactyly only. In the remaining studies, symbrachydactyly cases were included alongside other congenital upper limb anomaly conditions such as constriction band syndrome(15, 48, 53, 54, 56, 58, 59, 61) (n = 8), aphalangia, syndactyly, thumb hypoplasia, and transverse arrest. One study described outcomes on different congenital diseases, however, the outcomes for symbrachydactyly were analysed separately (Table 2). Eight studies in total described non-vascularized toe to hand transfers(48, 50, 54, 55, 58, 59, 63, 67), 8 studied vascularized toe to hand transfers(11, 15, 52, 60, 61, 63, 65, 66), five looked at distraction osteogenesis(13, 51, 53, 56, 62), and 3 examined web release(49, 57, 64). Only one study combined both lengthening techniques on a single patient(63). No other study compared different surgical treatments. Follow up time ranged from 12 to 122 months postoperatively, with a median follow-up time of 4.9 years. Short term follow-up (≤ 3 years) was reported in 4 studies(48, 51, 62, 64), middle term follow up (3–5 years) in 7 studies(15, 54, 56–58, 66, 67), and long term follow up (> 5 years) in 7 studies(11, 50, 52, 55, 59, 60, 63). Five authors did not provide information on follow-up time(13, 49, 53, 61, 65). Information on post-operative treatment was very limited, and a precise description of postoperative rehabilitation was provided by one study only (52).
Table 2
Author (year) | Years of patient inclusion | Female (%) | Age in months [mean] | Included patient diagnosis |
Buck-Gramko (1990) | 1976–1990 | not reported | 7 months to 17 years (range) | Symbrachydactyly and constriction ring syndrome. Finger devoid of a bony skeleton at the level of the proximal phalanges, or intermediate large bone defects in digits (mainly thumb). |
Cavallo (2003) | 1988–1997 | 41 | 15 | Symbrachydactyly, constriction band syndrome, aphalangia. |
Deutinger (1989) | 1969–1986 | 34 | 144 | Synbrachydactyly and syndactyly. |
Foucher (2001)-V | 1976–1998 | not reported | 16 | Symbrachydactyly (n = 45), transverse deficiency (n = 2), thumb hypoplasia (n = 5), miscellaneous (n = 6). |
Foucher (2001)-D | unclear | not reported | 109 | Symbrachydacty (n = 21), Clinodactyly (n = 5), Apert (n = 4), Brachydactyly (n = 7) others (n = 4). |
Garagnani (2012) | 1991–2007 | 58 | 32 | Digital hypoplasia resulting from symbrachydactyly (n = 33), constriction ring syndrome (n = 3), thumb hypoplasia (n = 3), perinatal subclavian venous thrombosis (n = 1). |
Gohla (2005) | 1975–2003 | 44 | 43 | Symbrachydactyly. |
Iba (2012) | 2005–2009 | Not reported | 14.6 | Symbrachydactyly (n = 2) |
Hierner (1998) | 1990–1993 | 20 | | Symbrachydactyly of the monodactylous type oder peromelic type, adactyly with transversal defect and akrosyndactyly. |
Hulsemann (2002) | 1989–2001 | not reported | 48 | Symbrachydactyly of the peromeliic type. |
Kawabata (2018) | 1993–2010 | not reported | 18 | Symbrachydactyly. |
Leca (2008) | 2007 | 0 | 14 | Symbrachydactyly |
Li (2013) | 2000–2007 | 38 | 31 | Symbrachydactyly of the short finger type. |
Lister (1988) | Not reported. | Not reported. | 36 | Congenitally deficient thumbs: Symbrachydactyly (n = 3), constriction ring syndrome (n = 3), transverse arrest (n = 6). |
Mann (2016) | 1994–2014 | Not reported. | 104 | Symbrachydactyly (n = 32) and amniotic band syndrome (n = 10). |
Matsuno (2004) | Not reported. | Not reported. | Not reported. | Symbrachydactyly, amniotic band syndrome, hypoplastic thumb, hypoplasia of the small finger, cleft hand, metacarpal synostosis and brachymetacarpia. |
Miyawaki (2002) | 1988–2001 | 25 | 88 | Symbrachydactyly (Müller Type D). |
Nikkhah (2016) | 1998–2012 | Not reported. | 28 | Symbrachydactyly (n = 12), Trauma (n = 3) |
Richardson (2003) | 1995–2001 | Not reported. | 31 | Symbrachydactyly (n = 7 monodactylic form, n = 6 adactylic). |
Sabapathy (2021) | 2005–2019 | 37 | Not reported. | Symbrachydactyly (n = 18), bilateral transverse deficiency (n = 1). |
Schenker (2007) | unclear | Not reported. | 34,5 | Symbrachydactyly (n = 8) |
Unglaub (2006) | 1975–1995 | Not reported. | 58 | Symbrachydactyly (congenital short finger (n = 4), oligodactylic type (n = 6), monodactylic type (n = 6), peromelic type (n = 3)) and ring constriction syndrome (n = 1). |
Van Holder (1999) | 1985–1996 | 29 | 44 | Symbrachydactyly (n = 2), constriction ring syndrome (n = 3), transverse absence (n = 9). |
Functional outcomes of the hand
For non-vascularized transfers, the range of motion (ROM) was the mainly reported outcome (Table 3). The analysis of the postoperative ROM showed modest results(50). Yet, a relevant improvement of the functional performance was confirmed in the majority of patients, when asked a general question about overall satisfaction with the postoperative function(54, 55, 67). Buck-Gramko and colleagues carried out an age-specific subanalysis of the ROM in the new joint, with better average results in the group up to 18 months(48).
For vascularized-transfers, more neurovascular functions were investigated including sensation(11, 52, 61), pincer strength(52), and sweating(15). The overall results were satisfactory and up to 77% of the parents were happy with the function of the hand(65).
Distraction osteogenesis showed limitations in improving circumference or joint motion(11), but was able to successfully affect pinch power(13). Power was improved wherever studies reported on pinch strength. Yet, a pinch grip was not achievable in all patients(51).
Table 3
Function, aesthetic and lengthening outcome measurements in non-vascularized transfers
Author (year) | Functional results hand | Aesthetic results hand | Aesthetic or functional results foot | Lengthening |
Buck-Gramko (1990) | Mean ROM in new joint: Age to 18 months: 35° (range 0°-90°) 19 months to 4 years: 10° (range 0° to 50°) Over 4 years: 15° (range 0° to 70°). | Not reported. | Mobility of the donor toes was mostly limited in active flexion, but not in extension. Function feet was mostly not impaired. Toe shortening: - no shortening in 12 (17%) − 3mm or less in 21 (30%) − 4-7mm in 18 (26%) − 8-12mm in 12 (17%) − 9 toes in need of surgical treatment due to shortening (13%) | Age to 18 months: in 70% average 3mm growth. 30% no growth. 19 months to 4 years: Average 3,2mm growth. 15% radiologically open growth plates. 15% no growth. Over 4 years: Average 1mm growth. |
Cavallo (2003) | Mean ROM it the new joint was 60°. | Not reported. | Toe shortening: − 3 mm or less in 46 (72%) − 4-7mm in 15 (23%) - >8mm in 3 (5%) - Minor shortening in 93% middle phalangeal transfers. - Minor shortening in 54% proximal phalangeal transfers. Toe ROM: - normal flexion in 60 patients (94%) - active extension in 49 patients (77%). | Age to 18 months: Average resorption of 0.4mm 18 months to 4 years: 0.1mm resorption Proximal phalanges transfer: average 0.7mm resorption Middle phalangeal transfer: 0.8mm growth. |
Garagnani (2012) | Not reported. | Not reported. | Emotional problems related to foot appearance were common. 100% tendency to hide feet. 7% hygiene issues. 7% cold intolerance, 3% balance problems. Instability of donor toes universal. Oxford Ankle Foot Questionnaire: − 89% parents dissatisfaction − 93% patient dissatisfaction | Not reported. |
Gohla (2005) | Instability (n = 6). Range of motion was modest. The improvement of hand function was remarkable in the most cases, yet not measurable. | Not reported. | Not reported. | Age to 18 months: 87% phalangeal growth, 4% resorption 18 months to 4 years: 49% phalangeal growth, 14% resorption Over 4 years: 24% growth, 45% resorption |
Kawabata (2018) | Active motion: good (n = 24), fair (n = 7), and poor (n = 16). Stability: good (n = 37), fair (n = 8), poor (n = 2) Alignment: good (n = 33), fair (n=)5, poor (n = 9). | Not reported. | Not reported. | Growth arrest at 5 years in 23%, and at 10 years, 78%. Gain in length of the transferred toe phalanx was 4.1 mm at 5-year follow-up and 5.2 mm at 10-year follow-up. |
Leca (2008) | A) 1 phalanx resorbs other gets pinch MCPJ D2 15-0-19° ; B) MCPJ D2 0-30-60° C) 10-0-30° and 20-0-40° MCPJ movement after 2 FPT | Not reported. | Not reported. | 1/6 non-vascularized phalangeal transfers resorption. No other growth parameters reported. |
Sabapathy (2021) | Mean ROM at the MCP joint was − 4° to 65° flexion. PIP-joint complete stiffness (n = 2), PIP-joint poor flexion (< 30° flexion) (n = 2). Mean key pinch strength 1.3 kg (2.6 kg on the normal side). Children evaluated with Pediatric Outcomes Data Collection Instrument had high mean scores in all domains. The average PODCI scores were low for the upper-extremity domain. The mean PROMIS scores were within normal limits. | On the aesthetic component of the Michigan Hand Questionnaire, children gave higher scores than parents. Children gave a high score of 78.1/100, whereas parents gave a score of 63.3/100. | Donor toes were short in all children. No gait disturbance or toe instability. No difficulties in footwear, walking or running. | An open epiphysis was found in 24 of 31 grafts. The mean growth was 3.4mm. Length was 71.8% of the contralateral phalanx. |
Unglaub (2006) | 75% of the parents confirmed significant improvements of the functional performance and confirmed improvements in manual skillfulness of their child. | 50% of parents confirmed improvements of self-confidence; 50% could not confirm improvements. 66% assessed the benefit of the operation and the postoperative procedure as justifiable. | 15% minor problems. 10% putting on socks complicated. No functional disability. | Growth arrest: Age to 18 months: 27% 18 months to 4 years: 10% Over 4 years: 29% |
Table 4
Function, aesthetic and lengthening outcome measurements in vascularized transfers
Author (year) | Functional results hand | Aesthetic results hand | Aesthetic or functional results foot | Lengthening results |
Foucher (2001) | All patients < 1 year of age had good integration, except for one “spur” with poor wrist mobility. Passive mobility in the 47 toes averaged 62° (range 32° to 72°). Active ROM mean of 38° and mean extension lag of 25°. The mean 2PD was 5 mm in 22 toes of 19 children over 7 years of age. | Not reported. | No morbidity in the donor feet. All were able to run, no neuromas. No cold intolerance. | 4/65 premature growth plate closure. Growth follow up in 12 cases average similar to contralateral toe (-12% to + 17%). |
Hulsemann (2002) | In 4/8 children without necrosis, a pincer grip with Maximal active span was in average 3,3cm. Pincer strength postoperative was in average 560g. In 6/8 children the 2-ponit-discrimination between 4-8mm. | Not reported. | No child had cosmetic issues concerning the foot modification. Normal footwear. One child had foot problems after several hour walks on asphalt. | Not reported. |
Lister (1988) | Active motion at the two interphalangeal joints has been achieved in 3 of 11 patients reviewed over the long term, totaling 35°, 40° and 40°, respectively. All showed sweating and good tactile adhesion by 6 months postoperatively. | Not reported. | The donor feet were of good appearance according to the surgeon. No difficulties. | Not reported. |
Nikkhah (2016) | Satisfactory large grip function in all by ability to hold large play brick. 91% could perform fine, small and twist grip. No instability. Active ROM was generally poor. The passive ROM was average 54° more than the active ROM. Light-touch sensation in all present. Mean S2PD of 5mm (4–6mm). Eleven out of 12 parents strongly agreed that their child’s toe-to-hand transfer had improved function. One parent disagreed (Patient H), as they felt their child bypassed the transferred toe. Eleven out of 12 parents reported feeling very satisfied with the results of surgery and also reported a willingness to recommend similar surgery to other families. Eleven strongly agreed that their toe transfer had improved hand function. Seven reported that their hands worked ‘normally’, four reported minor functional difficulties, and one child reported major functional problems. Cold intolerance was assessed using the Campbell & Kay classification system; seven children had no symptoms, and four reported having had some symptoms but they were not causing problems. One child experienced troublesome symptoms of cold intolerance. | Out of 12 parents, four were neutral, three agreed that it had improved the appearance of the child’s hand, and five strongly agreed that appearance had been improved. | No significant long-term, donor-site problems. Mild toe clinodactyly was noted in five out of 12 patients in the review clinic, but this did not cause any functional or aesthetic concern; there were no problems with gait, balance, or neuroma formation, nor were there any problems with cold intolerance. Two children complained of minor scar hypersensitivity. No concerns of donor site appearance. Mild toe clinodactyly was noted in five out of 12 patients in the review clinic, but this did not cause any functional or aesthetic concern; there were no problems with gait, balance, or neuroma formation, nor were there any problems with cold intolerance. Two children complained of minor scar hypersensitivity. Ten out of 12 children reported never having any issues with donor-site appearance, including that of the scar. Two children had sensitivity, particularly when they wore shoes, however, none of the children were self-conscious about the donor site | On radiographic analysis, the majority of physes in the transferred toes remained open and there was near normal longitudinal growth. The length of the transplanted digit varied in control digit from 83–98% compared with the unoperated donor site |
Richardson (2003) | 2 tinger stiff. 77% parents happy with function of the hand. | 85% parents happy with appearance of hand. On the aesthetic component of the Michigan Hand Questionnaire, children gave higher scores than parents. 3/13 parents reported that their child hid their hand occasionally. | 100% parents happy with function and appearance of foot donor site. | Not reported. |
Schenker (2007) | Average pinch strength ratio 50.9%. All patients had normal tactile sensibility in the trans- ferred digits except two who underwent toe trans- fer as adolescents. The postoperative motor function after toe transfer was more vari- able and was poor in one patients with exception- ally weak pinch strength and limited active range of movement. Two patients who had a toe transferred onto the first ray with- out a competent basal joint had a very weak pinch grip (< 10 N). With one exception, all patients could perform precision grip tasts with their operated hand. | Not reported. | Not reported. | Not reported. |
Van Holder (1999) | Mean extension deficit of 20° and active compound flexion of the PIP and DIP joints of 80°. In hands without fingers in which the two toes were transferred to create opposing pinch, a useful pinch was obtained in all. Sensation was reported by the patients as “normal” and similar to adjacent digits in those toes where both digital nerves were repaired. Even when nerve repairs were not possible the patients reported protective sensation. | In all cases nail growth was normal and without deformity. The children’s assessment of the aesthetic appearance of the transfers was satisfactory to good. In no cases was a prosthesis worn or requested by the patient or the parents. | In most cases the scar on the foot was regarded as hypertrophic by the surgeon, yet no foot problems were reported by the patient. | In all cases growth of the transferred toes was comparable with growth of the remaining toes and the other transferred toes. No growth arrest was recorded. No correlation of growth with ischaemia or nerve repair was seen. |
Table 5
Function, aesthetic and lengthening outcome measurements in distraction osteogenesis and web syndactyly release
Author (year) | Functional results | Aesthetic results | Lengthening results |
Distraction osteogenesis |
Foucher (2001) | Author comment that slender digits may experience stiffness. | might get stiff and slender digits, not necessarily good outcome | 41 cases with 2.3cm length gain |
Hierner (1998) | Lengthening does not provide normal circumference or interphalangeal joint motion. | Fibrosis of soft tissues: None (n = 3), little (n = 3), moderate (n = 3) | Average finger extension 20.4mm. |
Mann (2016) | Not reported. | Not reported. | With respect to preoperative length: Metacarpal average 18.4mm (9-40mm) = 73%. Phalange distraction average 14.0mm (7-17mm) = 77%. In 89% the distraction aim could be achieved. |
Matsuno (2004) | No pinch at the follow-up evaluation. The range of motion was preserved to within 10° in all patients. | 2 cases of symbrachydactyly, however, the bone growth was different not only for the lengthened metacarpals but also for the other metacarpals in the hand, thus causing the hand shape to change undesirably. | 48% increase in lengthening with average 8.1mm distraction length. |
Miyawaki (2002) | Pinch power in the affected hand was improved in all patients. The mean pinch power between the thumb and the elongated fingertip was 1.73 kg (range, 0.4 to 2.2 kg) on the involved side, compared with 1.70 kg (range, 0.3 to 2.2 kg) on the contralateral side. Careful examination demonstrated intact sensation throughout the lengthened rays. | Not reported. | No growth disturbance compared with the corresponding bones in the contralateral, normal hand at a mean of 3.9 years (range, 2–12 years) postoperatively. The bones were lengthened by a mean of 22.3 ± 4.2 mm (range, 20 to 28 mm), with a mean increase in bone length of 81.6% ± 21.5% (range, 50–115%). |
Web reconstruction |
Deutinger (1989) | Full extend of flexion and extension was achieved in 20/22 hands. full extent of flexion in 13/19 hands, in 6 hands the range of flexion was incomplete | Not reported. | Not reported. |
Iba (2012) | Tape measure test83 0.5 preoperative to 4.5 postoperative. Parents’ assessment function markedly improved. | Not reported. | Not reported. |
Li (2013) | Parents of 94.1% of the patients were satisfied with the overall function of the hand. | Parents of 76.5% were satisfied with the cosmetic appearance of the hand. | Not reported. |
Table 6
Comparison recipient site (hand) related complications in vascularized vs. non-vascularized transfers
Vascularized transfer | Non-vascularized transfer |
• 6% complications: Skin necrosis (n = 2; 1 partial, 1 full), instability (n = 2)11. • 100% complications: Skin necrosis (n = 3; 1 partial, 2 full) with bad sensibilty. All 22 patients were tenolysed 5 to 24 months postoperatively. Second tenolyse required (n = 2), correction osteotomy (n = 2), tendon transposition (n = 2), CMCJ arthrodesis of the radial toe (n = 1)52. • 8% complications: tenolysis required (n = 1)15. • 56% complications: wound breakdown (n = 1), skin graft loss (n = 1), K-wire infection (n = 1), in the long term, tenolysis was required (n = 6), development of a hammer toe (n = 1)65. • 100% complications: Secondary operations were required in 100% (14 patients). These included tenolysis, repair of tendon rupture, secondary tendon grafting or transfer, opponensplasty, web space deepening, metacarpal osteotomy, and ligamentplasty for joint instability61. • 42% complications: tenolysis (n = 5), wound infection (n = 3)60 Average of 54% hand related complications. | • 43% complications: Limited postoperative growth, so secondary lengthening was performed (32%), Wound issues (n = 5), subluxation (n = 2), resorption (n = 1)48. • 3% complications: Wound necrosis (n = 1), infection (n = 1), 58. • 6% complications: Resorption (n = 7)59. • 25% complications: Resorption (n = 22), skin necrosis (n = 4), infection (n = 2)50. • 9% complications: Partial necrosis (n = 5)55. • 5% complications: Skin necrosis (n = 1), resorption (n = 1)67. • 5% complications: Wound issues with partial skin necrosis (n = 2), wound infection (n = 1) with total resorption54. • 33% complications: phalanx resorption (n = 1)63 Average of 16% hand related complications. |
Table 7
Comparison donor site (foot) related complications in vascularized vs. non-vascularized transfers
Vascularized transfer | Non-vascularized transfer |
• 0% complications: No morbidity in the donor feet was noted. All patients were able to run, and no neuromas were noted11. • 9% complications: One child had little problems after walking several hours on asphalt52. • 0% complications: No difficulty was encountered at the donor feet15. • 0% complications: 100% parents happy with function and appearance of foot donor site65. • 0% complications: no foot problems were reported61. • 8% complications: minor wound dehiscence (n = 1)60 2% foot related complications. | • 13% complications: 9 relevant toe shortenings in need of reoperation. 48. • 8% complications: unacceptable deformity of > 8 mm (n = 3), accidental transection flexor tendon (n = 2)58. • 100% complications: 100% toe instability 59. • 6% complications: hypertrophic scarring (n = 1), instability (n = 1), axis deviation (n = 1)50. • 0% complications: No complications reported. 67. • 5% complications: hypertrophic scar (n = 1) 54. Average 22% foot related complications. |
Table 8
Hand complications in distraction osteogenesis and web release
Author (year) | Complications hand |
Distraction osteogenesis |
Foucher (2001) | 31.7% complications: delayed unions, infection, dislocation, pain, too short, joint stiffness, clynodactyly, fracture |
Hierner (1998) | 44% complications: infection (n = 1), early consolidation (n = 1), excessive pain (n = 1), refractures (n = 3), axis rotations (n = 2). |
Mann (2016) | 30% complications. Early consolidation (n = 10), deviation (n = 2), joint dislocation (n = 1), pin infection (n = 4), tendon dislocation (n = 1), late consolidation (n = 2) 16 reoperations were needed. |
Matsuno (2004) | 67% complications: Painful bony prominence of the fingertips (n = 2). |
Miyawaki (2002) | 14% complications: Fracture of the fifth metacarpal (n = 1). |
Web reconstruction |
Deutinger (1989) | 17% complications: Recurrence of syndactyly (n = 5). |
Iba (2012) | No complications reported. One patient required secondary additional first metacarpal rotation osteotomy to reconstruct an opposing position |
Li (2013) | 18% complications: scar tissue contracture due to partial necrosis of the local skin flaps (n = 6). |
Two studies reported on web reconstruction only. Deutinger described good results in improving the range of motion without further detail (49). Li reported a 94% parents’ satisfaction rate with the postoperative function of the hand(64).
Aesthetic outcomes of the hand
The report on aesthetics of the hand in non-vascularized transfers was limited to the two of eight studies by Sabapathy and Unglaub(54, 67), as most of these studies focused on the donor feet. Sabapathy evaluated the aesthetic component with the Michigan Hand Questionnaire. Children gave higher scores (78.1/100) than parents (63.3/100)(67). This was the only study assessing the aesthetic component with a validated outcome questionnaire(67). In contrast Unglaub, reported only a 50% improvement in patients’ reported self-confidence of the child(54).
Only two of the five studies investigating vascularized transfers reported on aesthetic results of the hand, whereas all of them reported on the aesthetic results of the foot. Richardson and Van Holder reported high satisfactory levels with the appearance of the hand(61, 65).
In distraction lengthening, no study reported on subjective aesthetic outcome. One study described results for aesthetic appearance as reported by the surgeon, which was undesirable(56).
Li described that in web reconstruction, 76% (n/N) of parents were satisfied with the cosmetic appearance, being the only reference for web reconstruction in symbrachydactyly(64).
Lengthening results
In non-vascular phalangeal transfer, digital growth was documented as radiographic closure of the growth plate or millimetre growth. Studies agreed that younger age is related to higher average growth, and growth rates are highest in an age under 18 months(48, 50, 58). One study reported finger length compared to the contralateral side, reporting a finger length of 71.8% compared to the contralateral toe phalanx (67).
In vascularized transfers, growth was described as similar to the contralateral toe side, with premature growth plate closure in only 4/72 children(11, 61).
In distraction osteogenesis the lengthening results reported were 20.4 mm(51), 18 mm(53), 22 mm(13) or 48%(56).
Comparison donor site (foot) related results and complications
On average, there was a 22% foot donor site complication rate in non-vascularized transfers, compared to a 2% foot related complication rate in vascularized transfers.
In non-vascularized transfers, toe shortening was reported in all studies, describing outcomes of the feet. Functional impairment and aesthetic issues were described in some cases, reaching up to 100% of the patients(50) and with up to 93% patient dissatisfaction in another study(59). The main donor site complications are toe shortening(48, 59, 67), instability of the toe remainders (50), and axis deviation(50). Cavallo et al. showed that the middle phalanx of the toe seems to be more robust than the proximal phalanx in terms of resorption. Garagnani reported emotional disorders to foot appearance(59), and Hulsen described that no child had cosmetic issues concerning the donor site(52). Buck-Gramcko explained that surgery-related severe shortening (> 8–12 mm) only was seen when flexor-extensor interposition had not been performed. No functional gait disturbance was noted.
In vascularized transfer, only one study described an aesthetic issue, a hypertrophic scar of the foot(61). Only one child had some difficulty walking - on asphalt for several hours (52). All others stated very good results at the donor feet with no morbidity or cosmetic issues, and no reported complications occurred. Richardson outlined that 100% of the parents of his 13 patients in his study were happy with the appearance of the donor site foot (65).
Complications hand
Hand related complications of non-vascularized phalangeal transfers were at 16% for all studies reporting on this technique. The most commonly reported, but not in all studies mentioned, complication was bone resorption of the transplanted phalanx, reported in 33/170 patients(48, 50, 54, 59, 63, 67). Bone resorption of the transplanted phalanx especially occurred in trimmed or partially explanted phalanges(50). Digital complications were at the highest when the skin and soft tissue envelopes were scarred and limited.(58) Other less reported complications included wound issues (8%)(48, 50, 54, 55, 58, 67), dislocation (1%)(48, 58), and infection (1%)(50, 54).
Overall, the hand related complication rate of 54% was much higher in the vascularized group than in the non-vascularized transfer. Van Holder and colleagues reported a 100% complication rate(61), including tenolysis, tendon rupture, secondary tendon grafting or transfer, opponensplasty, webspace deepening, metacarpal osteotomy, and ligamentoplasty for joint instability. The authors did not describe the frequency of these complications. The remaining studies(11, 15, 52, 65) reported vascular problems that required reoperation. In addition, 13% required a secondary tenolysis(15, 52, 65), 4% axis malformation were noted, 2% with instability and in 1% infections.
Of the five studies performing distraction osteogenesis, complications included infection (5/113–4%), early consolidation (11/113 − 10%), late consolidation (2/113 -2%), 4/113 (4%) axis deviation, 4/113 (4%) re-fracture, 3/113 (3%) excessive pain, and 1/113 (1%) joint dislocation and 1/113 (1%) tendon dislocation. The average complication rate was therefore 38%.
For web reconstruction, reported complication rates were high, with 18% recurrence of syndactyly(49) and partial skin necrosis(64). Syndactyly recurrence occurred in 9 divided pairs of fingers, in 7 cases, a split thickness skin graft was used. The use of split thickness skin grafts resulted in a 60% recurrence rate, whereas the use of full thickness skin graft merely led to 7.5% recurrence rate.(49)
Surgical timing
Buck-Gramcko(48), Cavallo(58) and Gohla(50) divided their patients treated with non-vascularized toe transfers into three groups according to age at surgery. Patients receiving transfers between 18 and 48 months according to the authors reported the best functional outcomes without detailing measurements. Surgery at a younger age results in less bone resorption(58), and the transplanted toe phalanx physis is more likely to remain open in younger patients(10, 68). Yet, all ages show disappointing phalangeal growth after transfer(42, 58, 69).