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PubMed1975

[Preliminary report on the follow-up examination of 101 surgical cases of syndactylia].

A review of 48 patients in whom 101 syndactyly separations were performed between 1963 and 1972 is reported with the peaks of frequency of the inital operation (being in early infancy, and just before entering school). Different surgical techniques were used for restoration of the web space, with the two-flap-technique of ZELLER most often used. About 60% of the fingers were separated by longitudinal incisions with the remaining 40% with zig-zag incisions. Of the skin grafts, about two-thirds showed excellent cosmetic appearance with the remaining third showing evidence of hyperpigmentation. Digital abduction was measured and approximated the spread seen in normal children.

Adult↗
PubMed1991

[Apert's syndrome with polymetatarsia].

Three cases of acrocephalosyndactyly with duplication of the first toe and presence of six well-individualized metatarsals are reported. This type of polysyndactyly should suggest the diagnosis of Carpenter syndrome, which is inherited on a recessive autosomal basis. However, in the three patients reported here, the facial dysmorphism was distinct from that seen in Carpenter syndrome and the syndactyly was more marked. The correct diagnosis therefore seems to be Apert acrocephalosyndactyly, a disease with dominant transmission. A mutation seems very likely and consequently the risk of recurrence in siblings is probably minimal.

Acrocephalosyndactylia↗
PubMed1977

Amniogenic bands associated with facial dysplasia and paresis.

A case of the amniogenic band syndrome is described with typical digital constriction bands amputations and syndactyly. The amniotic bands were demonstrated macroscopically and histologically, thus establishing the diagnosis. Facial dysplasia and unilateral facial paresis were attributed to undefined mechanical effects following amniotic rupture. In addition, there was evidence of partial umbilical cord compression which did not appear to have affected fetal well-being.

Abnormalities, Multiple↗
PubMed1978

[Results of various operations for syndactylia (follow-up studies and statistical evaluation of 101 primary operations)].

101 cases of syndactyly in 34 male and 14 female infants who were aged three months to seven years at the time of their initial surgery were reviewed one to 9.5 years thereafter. The web space was formed by a palmar and a dorsal flap; in 50% of all commissures the result was found satisfactory in the follow-up series. Straight incision - performed in two thirds of our cases - caused scar contractures and deformities of several fingers. In contrary, zig-zag or wavy incisions showed good results. Using almost exclusively full thickness skin grafts from the arm, the donor site looked poor because of non-cosmetic scares and keloids. After surgery two thirds of all patients used a special splint for finger abduction for an average of about one year; neither an amelioration of insufficient primary surgery was achieved nor reappearance of syndactylism could be avoided by this device. The mobility of joints was partially decreased regardless of the pre-operative condition. Combined functions in grasping, however, were not restricted to a considerable degree. There was no difference between cutaneous and osseous syndactylism with respect to the abduction ability. Even slight impairment of wound healing caused reappearance of syndactylism; reseparation of fingers was necessary 32 times during a ten year period of follow-up. Scar contractures - especially after necrosis of skin grafts and deep infections - required early correction, while slowly developing secondary webs had to be separated some years later. Disturbance of wound healing must, therefore, be treated adequately, or better should be avoided by precise surgery and careful management of postoperative dressing.

Bandages↗
PubMed1978

[An unusual case of polydactylia].

A woman of 85 had a symetrical malformation of the hands manifested as a combination of syndactyly of the soft parts of the 3rd and 4th fingers and an incomplete bifurcation of the bones of the 4th finger.

Female↗
PubMed1978

Abnormalities resulting from intra-adnexal injection of glucose in the rabbit embryo--an experimental model of "amniotic disease".

Intra-adnexal injections of glucose into the rabbit embryo have induced amputations of digits or segments of limbs, congenital grooves, amniotic bands, club feet, syndactyly, hare lip, anencephaly, and ulcerations of the scalp. We have thus reproduced all the anomalies which are encountered in the clinical syndrome of "amniotic disease." These anomalies result from destruction of the cutaneous epithelium and the subjacent mesenchymatous cells, and extravasation of blood with hematoma formation around the superficial vessels. This general mechanism explains most of these anomalies. Intra-adnexal injections of glucose thus constitute an external trauma for the embryo and is good experimental model of amniotic disease.

Abnormalities, Drug-Induced↗
PubMed1990

[The acrocallosal syndrome. Report of an additional case].

The acrocallosal syndrome is a rare congenital malformation syndrome with the main findings: agenesis of the corpus callosum, craniofacial dysmorphisms, mental retardation, polydactyly of the hands and/or feet. We report a patient with partial agenesis of the corpus callosum, dolichocephalus, broad and short nose, low-set and posteriorly rotated ears, pre- and postaxial polydactyly of feet, duplication with syndactyly of big toes, and postaxial hexadactyly of the left hand. Differential diagnosis and the genetic counseling are discussed.

Abnormalities, Multiple↗
PubMedDate not supplied

[Synpolydactyly: unusual features and variable expression in 5 generations of an Italian family].

The Authors report a family with synpolydactyly (or syndactyly type II) present in eleven out of thirty four members in five generations. Affected members do not show any other anomalies so that chromosomal or other genetic syndromes may be excluded. The possible mode of inheritance, the variability in expression, the penetrance in five generations are discussed. The peculiar bone deformities of hands and feet already reported by Cross et Al. are confirmed. The Authors presume that the present report is the first observation of synpolydactyly in an Italian family.

Adult↗
PubMed1979

[Complex bone abnormalities with fatal outcome: a new familial syndrome].

A new lethal disorder affecting two brothers is described. The features were curvature of the long bones, multiple fractures, dysmorphic facies, syndactyly and absence of ossification in the cranial vault. The condition could be inherited as an autosomal recessive or sex linked condition.

Abnormalities, Multiple↗
PubMed1979

Sclerosteosis in South Africa.

Sclerosteosis is a potentially lethal inherited disorder of skeletal overgrowth which has a minimum prevalence of 1 in 60,000 in the Afrikaner community of South Africa. Early presenting features are syndactyly and facial palsy, and the diagnosis should be considered in any infant of Afrikaner stock with these abnormalities. No medical treatment is available, but cosmetic surgery, cranial nerve decompression and prophylactic craniectomy be be of value.

Adolescent↗
PubMed1979

Endocrine function in sclerosteosis.

Sclerosteosis is a rare autosomal recessive condition which is characterized by excessive skeletal overgrowth, distortion of the facies, cranial nerve abnormalities and raised intracranial pressure. Syndactyly and digital malformation are associated features. Radiological examination reveals thickened sclerotic bone maximally involving the skull, including the pituitary fossa. Sclerosis and hyperostosis are present throughout the skeleton. Biochemical and endocrine tests were carred out on 3 patients with sclerosteosis in an attempt to detect any dysfunction of calcium regulation of the pituitary. Results revealed no abnormality of basal parathyroid or calcitonin secretion. Histological examination revealed quantitatively increased bone resorption in comparison with normal subjects, although the pattern resembled osteosclerosis. Regulation of growth hormone, adrenocorticotrophin, gonadotrophin and thyrotrophin function were intact. We conclude that pituitary function and calcium 'homeostasis' are normal in this disorder.

Adult↗
PubMed1986

Lyme disease during pregnancy.

Lyme disease is an increasingly recognized tick-borne illness caused by a spirochete, Borrelia burgdorferi. Because the etiologic agent of Lyme disease is a spirochete, there has been concern about the effect of maternal Lyme disease on pregnancy outcome. We reviewed cases of Lyme disease in pregnant women who were identified before knowledge of the pregnancy outcomes. Nineteen cases were identified with onset between 1976 and 1984. Eight of the women were affected during the first trimester, seven during the second trimester, and two during the third trimester; in two, the trimester of onset was unknown. Thirteen received appropriate antibiotic therapy for Lyme disease. Of the 19 pregnancies, five had adverse outcomes, including syndactyly, cortical blindness, intrauterine fetal death, prematurity, and rash in the newborn. Adverse outcomes occurred in cases with infection during each of the trimesters. Although B burgdorferi could not be implicated directly in any of the adverse outcomes, the frequency of such outcomes warrants further surveillance and studies of pregnant women with Lyme disease.

Adult↗
PubMed1989

[Knee pterygium syndrome in a newborn infant].

We report on a newborn boy with popliteal pterygium syndrome. Congenital anomalies included popliteal pterygia, symblepharon, cleft lip and palate with syngnathia, severe hypoplasia of both thumbs, a characteristic nail anomaly of both first toes, multiple syndactylies, and a hypoplastic scrotum with cryptorchidism. Mental development can be expected to be normal. Overall prognosis of the disorder after detailed operative correction is apparently favorable. Differential diagnosis is discussed in detail. The syndrome is autosomal dominantly inherited, our patient represents an isolated case.

Abnormalities, Multiple↗
PubMed1989

Oto-palato-digital syndrome in an Iranian infant.

A male infant is presented with wide fontanels, micrognathia, mid-face hypoplasia, hypertelorism, broad nasal root, down-slanting palpebral fissures, small thorax, funnel chest, short wide toes, camptodactyly and cutaneous syndactyly of fingers and toes, dysplastic bones with thin wavy ribs and bowed femore, cryptorchidism, and hypospadias grade I. The mother of this infant showed some signs of the same condition, including hypertelorism, micrognathia, small nose with depressed bridge, flat mid-face, impacted teeth and small chest. This case shows many similarities to oto-palatal-digital syndrome types I and II.

Abnormalities, Multiple↗
PubMedDate not supplied

[Syndactylia associated with multiple malformation syndromes. Observation of a new symptomatologic complex in 3 brothers].

Hand and feet malformations are often part of complex malformation associations. The present paper reports on a family whose three sibs (two males and one female) are affected with symmetric soft tissue syndactyly involving both fingers and toes, fifth finger clinodactyly, a pattern of dysmorphism including down slanting palpebral fissures, long flat nasal saddle, out turned nostril openings ("Greek warrior helmet"-like profile), dysplastic teeth, and, in addition, severe growth retardation, microcephaly, severe mental deficiency with immaturity of cerebral activity of EEG, hypergonadotropic hypogonadism and some skeletal anomalies. All cases show large secondary constriction in one of the chromosome 1 pair (1qh+).

Abnormalities, Multiple↗
PubMedDate not supplied

Desyndactyly without skin graft: case presentation and literature review.

Many articles have been written on the correction of syndactyly, most of which refer to the fingers. The use of skin grafts appears necessary to obtain satisfactory correction in much of the literature. The authors present a case of desyndactyly without using a skin graft.

Adolescent↗
PubMed1985

Management of thumb duplication.

Radial polydactyly, a common congenital problem, most often occurs as thumb duplication. It appears in pentadactylous, polydactylous, and hypoplastic hands and is simulated by complex syndactyly. Classically, there are three categories for radial polydactyly: severe hypoplasia, partial duplication, and total duplication (easily confused with pseudoduplication). The common second category is subcategorized by Wassel's seven types. Management techniques vary with these classifications. Until a decade ago, the standard recommendation was simple excision of one of the duplicates. Problems of deformity, instability, and weakness became apparent, and reconstructive options were developed. Review of a series of 54 duplicate thumbs, all surgically treated, reflects the changing pattern of reconstruction. With current techniques, good results are usual, although secondary reconstructive procedures may be required. Simple excision is seldom indicated. Combination procedures, involving core tissues of bone, joint, and tendon or peripheral tissues--neurovascular, subcutaneous, and skin--or sometimes all of these, are preferred for reconstruction. The major current problem is to achieve the maximal good result with a minimal number of surgical operations.

Arthrodesis↗
PubMed1985

[Blood circulation in the fingers and electric activity of the muscles after the surgical treatment of children with ectrodactylia].

The rheographic examination of hands in patients aged from 2 to 15 with ectrosyndactyly has revealed a less volume of blood flow, worse elastic properties of the vessels of diseased fingers and lower level of the electric activity of interosseous muscles of the hand. Elimination of syndactyly with the help of different variants of operations did not cause complications in the postoperative period associated with the disturbed blood flow in the fingers.

Adolescent↗