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PubMed1981

Familial dermatoglyphic analysis in syndactyly type I.

A familial dermatoglyphic study of 25 syndactyly type I patients and 53 unaffected first-degree relatives showed a significant increase of complex fingertip patterns in the patients and in their relatives as well. An embryonic link between the formation of complex fingertip patterns and syndactyly type I is postulated. The study contributes new data on the hereditary transmission of syndactyly type I which cannot be used for individual genetic counseling. Calculation of the recurrence risk is difficult in view of the reduced penetrance of the gene.

Belgium↗
PubMed2003

Open treatment of syndactyly of the foot.

The aim of reconstruction of the webs for syndactyly of the foot is cosmetic improvement, so skin grafting should be avoided. We present our long-term results of 19 feet with simple cutaneous syndactyly in 15 patients who were treated by an open technique. The bottom of the web was covered with a dorsal rectangular flap and the remaining skin defect was left open to epithelialise spontaneously. This took about 4 weeks. After a mean follow-up of 5.7 years (range 3-9), no hypertrophic scar or pigmentation of the skin had developed. Creeping of the web was seen only during the first postoperative year. The final depth of the web satisfied the families of all patients. As the aim of web reconstruction for syndactyly of the foot is purely cosmetic, we conclude that this simple method is reasonable treatment.

Female↗
PubMed1994

Release of acquired syndactylies in Kindler syndrome.

Kindler syndrome is a rare, blistering skin disease characterized by acral bullae, poikiloderma, and diffuse cutaneous atrophy. Kindler syndrome has been established as a separate entity from epidermolysis bullosa; however, controversy still remains as to whether Kindler syndrome can be differentiated from Weary's hereditary acrokeratotic poikiloderma. Fusion of the digits secondary to blistering and scarring, "pseudosyndactyly," has been reported in several patients with Kindler syndrome; however, surgical correction of the syndactylies in these patients has not been described. In this report, a patient with Kindler syndrome underwent surgical treatment of acquired syndactylies. Treatment included a tailored approach to preparation of the patient for surgery, surgical separation of fused tissues, selection of donor site for skin-graft harvest, postoperative dressings, splinting, and therapy. Results in our patient 2 years after correction demonstrate that syndactyly release in Kindler syndrome can be accomplished effectively, with improvement in both function and appearance.

Adolescent↗
PubMed2004

Usefulness of a palmar crease template for the treatment of complicated syndactyly.

The treatment of complicated syndactyly has been a difficult problem because it involves not only cutaneous syndactyly but also abnormal arrangement of the finger rays. This means that this anomaly is characterized by abnormal patterns of the palmar creases. The authors prepared a template for tracing the proximal, middle, and distal palmar creases of the nonaffected hand on a clear film and used it as a mirror image to create the preoperative design, to measure the distance between the fingers, and to estimate the location and size of the skin graft intraoperatively. The authors think that the recreation of the normal palmar crease pattern in the affected hand can lead to normal arrangement of the fingers and it is very useful for the reconstruction of multiple finger webbing as part of the treatment of complicated syndactyly.

Female↗
PubMed1996

Identity-by-descent mapping of recessive traits in livestock: application to map the bovine syndactyly locus to chromosome 15.

Twelve animals affected with syndactyly or mulefoot were sampled in the Dutch black-and-white cattle population. Analysis of the pedigree data reveal that all of these individuals traced back to a single acknowledged carrier founder individual. Between seven and nine generations separated the founder from its 12 affected descendents. The 12 affected offspring were genotyped for a battery of 213 microsatellites spanning the 29 bovine autosomes. The resulting genotypes were analyzed using a maximum likelihood approach searching for shared homozygous haplotypes among affected individuals. Three candidate regions for the syndactyly locus emerged from this initial screening. syndactyly was shown to map to one of these candidate regions on chromosome 15 by genotyping 29 additional individuals linking founder and affected offspring and performing a conventional linkage analysis with the LINKAGE programs. This study illustrates the potential of identity-by-descent mapping in livestock populations.

Animals↗
PubMed1993

Familial syndactyly type III--report of a large pedigree.

We present a pedigree containing 14 cases of familial syndactyly of the ring and little finger (Type III syndactyly). Three of these were treated surgically in this unit, and a fourth had previously been treated elsewhere by the senior author of this paper. Analysis of the pedigree confirms the autosomal dominant nature of the genetic defect, while clinical study reveals variable phenotypical expression. This is one of the largest pedigrees et reported for this variety of syndactyly.

Adolescent↗
PubMed1991

Syndactyly correction of the hand in Apert syndrome.

Surgical correction of syndactyly of the Apert hand should begin by 6 months and be completed by 3 years of age. As much surgery as possible is carried out at each sitting. Digit separation should be in order of functional importance. The first web space is deepened with a four-flap Z-plasty or a dorsal skin flap from the web and index finger. Syndactyly release using a dorsal flap and zig-zag technique is used to create the second and fourth web spaces. The complex long-ring syndactyly often requires a pedicled groin flap for reconstruction and preservation of growth potential. A five-digit hand can be achieved with adequate grasp and stable, sensate, well-aligned digits. These children can attain some degree of independent finger motion and aesthetically acceptable hands with this approach.

Acrocephalosyndactylia↗
PubMed1969

Chromosome abnormalities in patients with syndactyly.

Chromosome studies on 105 patients with syndactyly included two trisomy-21 mongols, a chromatin-positive boy with 47, XXY, a chromatin-negative short girl with 45,X0 and a boy with a familial D/D translocation. Chromosome patterns were normal in the other cases which included three patients with acrocephalosyndactyly and one patient with oro-facial-digital syndrome.The incidence of chromosome abnormalies was greater than expected since syndactyly of the fingers is uncommon in the chromosome disorders.THIS INCIDENCE MAY BE RELATED TO THE INCREASED MATERNAL AGE (MEAN: 29.4 years) of the syndactyly group compared to maternal age (mean: 26.64 years) of the control group although, paradoxically, four mothers of the five patients with chromosome abnormalities were young.

Child↗
PubMed1994

[Anesthetic management of a patient with a cryptophthalmos syndactyly syndrome and subglottic stenosis].

We experienced five episodes of anesthesia for a girl with dryptophthalmos syndactyly syndrome and congenital subglottic stenosis from the age of 1.3 year to 4 years. A girl was born at 34 weeks of gestation. The birth weight was 1360 g. The Apgar score was 8 at one minute and there was a hoarseness. She had right cryptophthalmos, syndactyly of hands and left foot, left polydactyly, anomalies of ear and nose, and agenesis of right kidney. The operation was scheduled for syndactyly under general anesthesia when she was 17 days and 5 months. As intubation was unsuccessful in both occasions, the operation was cancelled and subglottic stenosis was pointed out. We decided to postpone the operation until she could cry fully without cyanosis. We evaluated her respiratory ability from the time she became able to cry fully without cyanosis. As a result, we could manage her without any complications such as hypoxia or hypercapnea except mild wheezing.

Abnormalities, Multiple↗
PubMed1996

Tissue expanders in syndactyly: a brief review.

Tissue expansion has been advocated as a method of obtaining enough skin to correct syndactyly without using skin grafts. However, results have been disappointing, indications are unclear, and complication rates are still relatively high. We review the published cases of syndactyly treated with tissue expansion, acknowledging the lack of data and guidelines for its use in such condition. After more than 10 years since its introduction in the treatment of syndactyly, its use is still not as widespread as one would have been led to expect.

Humans↗
PubMed2003

Clinical experience using the dorsal reverse metacarpal flap for the treatment of congenital syndactyly: report of four cases.

Skin grafts and local flaps are conventional methods of repair for simple complete syndactyly, but the results obtained are not always as functional and aesthetic as desired and frequently leave postoperative scars and residual syndactyly. In this article, we describe a new surgical technique for the correction of syndactyly that utilizes a metacarpal dorsal reverse flap to provide skin coverage to the interdigital space and also to 1 side of the finger, therefore avoiding the use of skin grafts and resulting postoperative scar contracture. We report 4 cases using this procedure, with a 12-month follow up. The technique is safe, easily performed, and reproducible and requires a single surgical procedure. It uses donor tissue identical in color, texture, and thickness, which renders acceptable cosmesis and good hand function.

Journal Article↗
PubMed1998

Thanatophoric dysplasia type I with syndactyly.

We report on a case of thanatophoric dysplasia type 1 (TD1) due to a Tyr373Cys mutation in the fibroblast growth factor receptor 3 (FGFR3) gene with soft tissue syndactyly of the fingers and toes. Syndactyly has not been previously described in TD or other conditions with FGFR3 mutations, but occurs in several craniosynostosis syndromes due to mutations in FGFR2. We conclude that mutations in FGFR3 may also be associated with developmental abnormalities due to interference with programmed cell death.

Cysteine↗
PubMed2001

Confirmation of genetic homogeneity of syndactyly type 1 in an Iranian family.

Syndactyly type 1 (SD1) is the most common type of syndactyly, inherited in an autosomal dominant fashion and characterized by complete or partial webbings between the third and fourth fingers and/or between the second and third toes. We recently encountered an Iranian family in which 33 members in six generations were affected with SD1. As a locus of SD1 in a German family has recently been assigned to chromosome 2q34-q36, we performed a linkage analysis of the Iranian SD1 in order to know whether the disorder is genetically homogeneous. With the analysis on 15 affected and 16 unaffected persons in the Iranian family, using dinucleotide repeat polymorphisms as markers, we mapped the SD1 locus to 2q34-q36 with a maximum LOD score of 6.92 at a recombination fraction straight theta = 0.00 (penetrance = 1.00) for the D2S2179 locus. The result not only confirmed the gene assignment, but also suggests genetic homogeneity of the disease.

Alleles↗
PubMed1995

A new operation for syndactyly and polysyndactyly of the foot without skin grafts.

A new surgical technique is described for the treatment of polysyndactyly and syndactyly of the toes. The lateral aspects of the toes are covered by only the interdigital skin without any skin grafts. A subcutaneous pedicled pentagon flap designed on the plantar skin is used for the repair of the base of the interdigital space. 12 interdigital spaces involved in polysyndactyly of the 4th, 5th and 6th toes and 6 interdigital spaces involved in incomplete syndactyly of the toes have been treated. Good aesthetic and functional results were achieved.

Adult↗
PubMed1989

Syndactyly: a review of the factors which influence surgical treatment.

A review was undertaken of 100 patients with 218 surgically-treated syndactylies operated upon over a ten year period. The pre-operative factors which influenced the result of surgical treatment were the complexity of the syndactyly and the presence of other congenital abnormalities in the hand. The operative factor which most influenced outcome was the type of skin-graft used. 42 patients required at least one secondary operation to obtain an acceptable result, comprising 22% who developed web creep and 26% a significant flexion contracture; five patients developed both a contracture and web creep. In contrast, the type of flap used to reconstruct the web floor had little influence on the result.

Adolescent↗
PubMed1990

Syndactyly: can web creep be avoided?

Syndactyly is one of the most common congenital hand deformities but there still remains a high incidence of contractures and web creep after attempts at surgical correction using many assorted techniques. Little attention has been paid to the potential junctional scar in the aetiology of web creep. To remedy this, a technique is described which involves a dorsal flap and two palmar laterally-based flaps. This method not only breaks up the palmar junctional scar but also completely reconstructs the web, not just the floor. The procedure can be used in all varieties of syndactyly and has reduced the incidence of creep in a series of 49 webs.

Adolescent↗