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Constriction Band

Syndrome
Kenji Kawamura, MD, PhDa, Kevin C. Chung, MDb,*

KEYWORDS
 Constriction band syndrome  Malformation
 Congenital anomaly  Hand  Treatment

Constriction band syndrome is a relatively rare con- have some sort of abnormal gestation history.9 Pre-
dition in which fetal parts become entangled in the natal risk factors associated with constriction band
amniotic membrane, leading to deformation, mal- syndrome include: prematurity (<37 weeks); low
formation, and amputation. Many terms have birth weight (<2,500 g); maternal drug exposure;
been used for this complex anomaly, including and maternal illness or trauma during pregnancy. At-
constriction band syndrome,1 amniotic band syn- tempted abortion in the first trimester is also a highly
drome,2 congenital annular constrictions,3 con- associated risk factor. No autosomal inheritance
genital ring constrictions,4 and intrauterine pattern has been identified, and maternal prenatal in-
amputation.5 The overabundance of synonyms for fection does not appear to be an associated feature.
constriction band syndrome is caused by confu- Although the etiology of constriction band syn-
sion regarding its etiology. Recent reports revealed drome remains controversial, there are two main the-
that constriction band syndrome or amniotic band ories that may explain the development of this
syndrome might be the most frequently used term syndrome. The first is the intrinsic theory, proposed
to describe this complex congenital anomaly.6–11 by Streeter in 1930,5 which suggests that constric-
Although capricious manifestations do occur in tion band syndrome represents an inherent develop-
constriction band syndrome, several characteristic ment defect in embryogenesis. In this theory, the
features are relatively consistent findings. Distal bands arise from an endogenous defect in germ
ring constrictions, intrauterine amputations, and plasm differentiation that causes the limb to become
acrosyndactyly are the most common findings in necrotic and form fibrous bands. Some authors who
this syndrome and are typically seen in the distal support Streeter’s intrinsic theory have expanded on
aspect of extremities.8,9 Multiple extremity involve- it by suggesting teratogenic insult, viral infection, or
ment is usually expressed, with an average of three vascular disruption as the cause of the malforma-
affected extremity parts.9 Deformations affecting tions in constriction band syndrome. In 1961, Patter-
the upper extremities are disabling and pose son15 explained the etiology of the syndrome as
a treatment challenge for hand surgeons because a primary failure of the development of the subcuta-
of the unique presentations in each individual.12 neous tissue during the morphogenetic period. Van
Allen and coworkers16 suggested that amputations
and constriction rings may be due to vascular distur-
EPIDEMIOLOGY AND ETIOLOGY
bances. Lockwood and coworkers17 reviewed 14
The reported incidence of constriction band syn- cases of twin gestations associated with constriction
drome varies from 1/1,200 to 1/15,000 live band syndrome and reported that the disorder was
births.4,9,13,14 No distinct sex predilection has been more common in monozygotic twinning, thereby
determined. Nearly 60% of the documented cases supporting a teratogenic cause.

Supported in part by a Midcareer Investigator Award in Patient-Oriented Research (K24 AR053120) from the
National Institute of Arthritis and Musculoskeletal and Skin Diseases (to Kevin C. Chung).
a
Department of Orthopaedic Surgery, Nara Medical University, 840 Shijyo-cho, Kashihara, Nara 634-8522,
hand.theclinics.com

Japan
b
Section of Plastic Surgery, Department of Surgery, University of Michigan Medical School, 2130 Taubman
Center, 1500 E. Medical Center Drive, Ann Arbor, MI 48109-0340, USA
* Corresponding author.
E-mail address: kecchung@umich.edu (K.C. Chung).

Hand Clin 25 (2009) 257–264


doi:10.1016/j.hcl.2008.10.007
0749-0712/08/$ – see front matter ª 2009 Elsevier Inc. All rights reserved.
258 Kawamura & Chung

An alternative view for the etiology of constriction CLINICAL PRESENTATION


band syndrome was described by Torpin in 1965.18
He proposed an extrinsic theory, in which early am- The clinical manifestations are predominantly dis-
nion rupture forms adherent bands that constrict, tal deformities such as constriction of limbs and
entangle, and amputate limbs. As the amnion rup- fingers, syndactyly, acrosyndactyly (fenestrated
tures, it slips off the chorion to form the detaching syndactyly), phalangeal hypoplasia, and amputa-
mesoblastic fibrous strands. These strands subse- tion of limbs and fingers.8,9 Multiple malformations
quently become entangled around the digits or such as clubfoot (30% of patients), leg length dis-
limbs. The denuded chorion then absorbs the crepancies (24%), other bone anomalies (12%),
amniotic fluid and causes a temporary oligohy- special craniofacial defects such as cleft lip and
dramnios or compressive environment. Torpin’s palate (8%), visceral and body wall defects, and
extrinsic theory is supported by the findings of anencephaly (5%) have been detected in 70% of
many authors. Kino1 successfully produced acro- infants with the disorder.8,9,21,22 Owing to the
syndactyly in animal experiments involving amni- fact that no two cases with this syndrome are
otic sac puncture, and concluded that this exactly alike, only some of the above-mentioned
anomaly was due to hemorrhage from the marginal anomalies are present in each individual case.
blood sinuses of the digital rays in already devel- The clinical presentation in the hand varies from
oped or developing limb buds. Higginbottom and slight indentations on the affected part to distal at-
coworkers19 suggested that early amniotic rupture rophy, lymphedema, acrosyndactyly, and amputa-
leads to severely affected infants who are tion (Fig. 1). From the hand surgeon’s perspective,
frequently absorbed or stillborn, whereas later am- the most important aspect is that the structures of
niotic rupture primarily results in limb involvement. the hand proximal to the constriction band are nor-
Foulkes and Reinker9 conjectured that amniotic mal. If the ring constriction is severe, the veins,
disruption before 7 weeks of gestation is more likely arteries, lymphatics, and nerves may be compro-
to produce limb reduction, polysyndactyly, and mised.21 Neurologic impairment is usually attrib-
syndactyly secondary to interference with segmen- uted to axonotmesis or neurotmesis. This may be
tation, whereas later amniotic rupture yields me- caused by direct pressure from the constriction
chanical deformation of amniotic bands, clubfoot, band or attributable to compartment syndrome
distal hypoplasia, lymphedema, and intrauterine distal to the band in infants with rapidly progres-
amputations. The occurrence of asymmetric defor- sive swelling. Distal digits are typically malformed
mities with nonembryologic distributions also sup- owing to phalangeal hypoplasia or terminal ampu-
ports Torpin’s extrinsic theory. tation. Acrosyndactyly is frequently associated
Although there has been much debate over the with distal amputation.23 This type of syndactyly
cause of constriction band syndrome, the majority involves binding of adjacent digits in a lassoed ap-
of contemporary authors have described Torpin’s pearance. If acrosyndactyly is present, it is invari-
extrinsic theory as the most appropriate explana- ably associated with a proximal interdigital sinus or
tion for the entire clinical spectrum of constriction cleft that communicates from the dorsal side to the
band syndrome.1,9,10,13,19,20 volar side (Fig. 2). Cutaneous syndactyly does not
usually involve underlying bony fusion.

Fig.1. A 4-year-old boy with constriction band syndrome. (A) Right hand showing amputation of the thumb and
ring finger and acrosyndactyly of the index and long fingers, with left hand showing amputation of the long and
ring fingers, angulated index and little fingers, and lymphedema of the little finger. (B) Radiograph of the same
patient showing acrosyndactyly of right index and long fingers associated with distal amputations.
Constriction Band Syndrome 259

characteristic features and restriction of fetal


movements.25 Radiography may reveal severe
skeletal defects such as absent cranial ossifica-
tion, severe limb deformities, or spinal defor-
mities.13 As the more serious manifestations of
the condition are outside the limbs, in the head,
neck, and trunk, neonatal diagnosis is frequently
difficult and is accurately accomplished in only
29%–50% of cases in the absence of specialized
genetic consultation.2,19 Only 13% of constriction
band syndrome cases with severe craniofacial de-
formities have been reported to be correctly diag-
nosed.13 The craniofacial deformities present in
constriction band syndrome are typically bizarre
Fig. 2. Acrosyndactyly with a probe in the sinus tract.
and frequently nonembryologic in location. As
many as one in 20 anencephalic babies may
There is a predilection for the hand as the site of have constriction band syndrome.19 The incorrect
involvement.7,9 The central digits are often in- diagnosis is usually that of the most prominent
volved, whereas the frequency and severity of anomaly observed. The presence of fibrous bands
thumb involvement are minimal, even when all at constriction points is helpful in the diagnosis.
the fingers are severely affected. Foulkes and For isolated extremities, the important differen-
Reinker9 reported that, among 93 affected hands, tial diagnoses of constriction band syndrome
the ring finger was most frequently involved include symbrachydactyly and transverse
(89.2%) followed by the long (86%), index (71%), deficiency. Symbrachydactyly usually affects the
and little (51.6%) fingers. The incidence of thumb entire hand, and the patient typically has a small
involvement was only 21.5%. Several reasons hand with simple syndactyly. Bilateral cases of
have been proposed for the lack of thumb involve- symbrachydactyly are rare, ranging from 1.6%–
ment.1,7,11 First, with fetal hand positioning, with 10%,12 whereas multiple areas in more than one
either outstretched fingers or a clenched thumb extremity are usually involved in constriction
in the palm, the thumb is protected by the other band syndrome.9 The defect of symbrachydactyly
fingers. Second, the developmental difference of is believed to be mesodermal, which explains the
each digital ray may have correlation with the pre- presence of distal finger ectodermal structures
dilection. Thumb development precedes that of such as pulp, nail fold, and nail. Symbrachydactyly
the central digits, and it is assumed that constric- may occur with Poland syndrome. Ogino and
tion band syndrome occurs at the later stage of Saitou26 performed a detailed comparison of
limb development because the involved parts are constriction band syndrome and transverse
relatively well developed—and delivery of a well- deficiency. Transverse deficiency is more often
developed amputated extremity has been unilateral, whereas constriction band syndrome
reported. Third, anatomic differences should be is bilateral. Transverse deficiency tends to be
considered. The thumb and little finger have inde- more proximal, and rudimentary fingernails are
pendent blood supplies from separate digital ves- common. Bone hypoplasia may be found in neigh-
sels. If the etiology of constriction band syndrome boring fingers or proximally in the limb in trans-
is due to hemorrhage within the limb bud, as sug- verse deficiency. Amputations in constriction
gested by Kino,1 this anatomic difference may play band syndrome tend to have tapering bone
an important role in the predilection for site stumps, whereas transverse deficiencies more
involvement. often take the form of disarticulations.

DIAGNOSIS CLASSIFICATION
Ultrasonographic analysis allows prenatal detec- Several classification systems of the limb lesions
tion of constriction band syndrome by visualization in constriction band syndrome have been devised,
of amniotic bands attached to the fetus.24 In the but they add little to the clinical understanding be-
first trimester, it is extremely difficult to detect cause constriction band syndrome is a complex
the syndrome, especially if the amniotic bands collection of asymmetric congenital anomalies, in
are limited to the extremities. In the second and which no two cases are exactly alike. The most
third trimesters, it is relatively easy to detect the widely used classification system was proposed
major anomalies of the syndrome by their by Patterson,15 and is based on the severity of
260 Kawamura & Chung

the syndrome (Box 1). There are four categories in Timing of Surgery
Patterson’s classification, namely: simple con-
The timing of surgery is determined by the dis-
striction rings, constriction rings associated with
ease severity and predicted skeletal growth.
deformity of the distal part with or without lymphe-
Constriction bands with severe distal lymphede-
dema, constriction rings associated with acrosyn-
ma, cyanosis, and circulatory problems may
dactyly, and intrauterine amputation. In addition,
progress quickly to irreversible ischemia and
Patterson further divided the constriction rings as-
subsequent ulceration or infection. In such pa-
sociated with acrosyndactyly into three types:
tients, urgent release of the bands should be
type I, conjoined fingertips with well-formed
performed within a few days after birth. In other
webs of the proper depth; type II, the tips of the
cases, removal of the constriction bands is ac-
digits are joined, but web formation is not com-
complished by single or two-stage release, usu-
plete; and type III, joined tips, sinus tracts between
ally beginning at 3 months of age. Some
digits, and absent webs.
authors advocate a two-stage procedure to
avoid vascular interruption to the distal seg-
ment.21 Only 50% of the band is released at
TREATMENT
a time, and once the cutaneous circulation has
Treatment of constriction band syndrome must be been reestablished across the scar, the remain-
individualized, and ranges from cosmetic repair to ing 50% of the band can be released safely.
emergency limb-sparing band release. Shallow An interval of 6–12 weeks between the proce-
bands may require no operative treatment unless dures is advised.11 Most experts recommend
they interfere with circulation or lymphatic drain- single release for superficial bands and two-
age. Cosmetic repair of shallow bands without stage release for deep bands.28,29 In patients
lymphedema may be done electively. Deep bands with acrosyndactyly, surgery is recommended
require release of the constriction bands by cir- between the ages of 6 months and 1 year to al-
cumferential Z-plasty or W-plasty. In cases with low for proper longitudinal bone growth.
severe ischemia, which may lead to osteomyelitis,
amputation of the distal part may be considered. Release of Constriction Bands
On-top plasty (partial digital transfer), toe-to- Regardless of the technique used for release of
hand transfer, bone lengthening procedures, and the constriction band, all authors agree that the
pollicization procedures have been performed to constriction band should be excised and dis-
restore function in cases with digital hypoplasia carded, and not used as part of the reconstruc-
and amputation. In patients with acrosyndactyly, tive flap (Fig. 3). The contracted portions of the
separation of digits and web reconstruction are re- band remain deformed during transposition and
quired. Current improvements in prenatal diagno- can add to the residual defect. Other surgical
sis and fetoscopic surgical techniques may considerations include the preservation of at
eventually allow in utero treatment of constriction least one or two large subcutaneous veins along
band syndrome.25,27 with the neurovascular bundle to prevent postop-
erative distal venous congestion. In cases with
deep dorsal bands, there is often a paucity of
dorsal veins, and two-stage release should be
Box 1 considered.
Patterson’s classification of constriction band Traditionally, release of constriction bands has
syndrome been performed by serial Z-plasties of skin follow-
1. Simple constriction rings ing excision of the fibrotic constriction band. This
2. Constriction rings associated with the defor- traditional technique is not effective for eliminating
mity of the distal part, with or without the contour deformity in severe cases. The sand-
lymphedema glass deformity, which results from subcutaneous
3. Constriction rings associated with acrosyn- tissue deficiency under the constriction band, per-
dactyly sists after using the traditional technique. In 1991,
Type I: conjoined fingertips with well- Upton and Tan30 described a new technique for
formed webs of the proper depth
constriction band release to prevent recurrent
Type II: the tips of the digits are joined, but
contour deformities. After excision of the constric-
web formation is not complete
Type III: joined tips, sinus tracts between tion band and debulking of excess adipose tissue,
digits, and absent webs the mobilized subcutaneous adipose flap is ad-
4. Intrauterine amputation vanced into the defect as a separate layer, with
thin Z-plasties transposed separately (Fig. 4).
Constriction Band Syndrome 261

Fig. 3. (A) Ring constriction of right distal forearm. (B) The design of Z-plasties is shown. (C) Appearance after
excision of the groove and repair with Z-plasties.

Z-plasties are positioned along the side of the digit important as their spacing, length, bulk, stability,
with a straight-line closure dorsally to minimize vis- and control.11 Standard syndactyly techniques
ible scarring. Many authors have reported suc- are used as much as possible. In general, the fin-
cessful results using Upton’s technique for the gers are separated with carefully planned zigzag
treatment of constriction bands.11,12,31 However, incisions, and a broad commissural space is cre-
in cases with a broad constriction band, a cross- ated with a dorsal skin flap. Surgery should only
finger flap may be used to replace the deficient be performed on one side of a finger at a time.
area. If multiple digits are involved, a large flap Most patients with acrosyndactyly associated
such as a groin flap may be considered. As with with constriction band syndrome have type III de-
every case of constriction bands, the treatment fects (joined tips, sinus tracts between digits and
should be tailored to the individual. absent web) according to Patterson’s classifica-
tion. If the sinus tract is inadequate to function
as a web space because of its distal location
Surgery for Acrosyndactyly
and narrow space, it can be excised and may
Acrosyndactyly is a condition in which two or be used as a skin graft. Occassionally, the sinus
more fingers are fused at their terminal portions tract may contain adequate skin at its base; this
with proximal epithelial lined clefts or sinuses be- skin can be retained to serve as the web space
tween the fingers.23 The goals of surgery for acro- skin. Finger separation is easiest when performed
syndactyly are to separate the fingers and create in the proximal to distal direction. However, stan-
a web space to provide the best functional results dard syndactyly separation techniques occasion-
(Fig. 5). Surgical planning should be guided by ally cannot be used distally because the fingers
the dictum that the number of fingers is not as distal to the point of fusion may not be clearly
262 Kawamura & Chung

Fig. 4. Schematic drawings for releasing of the constriction band with Upton’s technique. (A) Excision of all skin in
the side walls. (B) Debulking of excess adipose tissue. (C) Subcutaneous adipose flaps are mobilized as needed to
correct the contour deformity. (D) Skin and subcutaneous closures are preferably staggered.

defined as belonging to a specific finger. As the the child is larger. Full-thickness skin grafts
dissection proceeds distally, a decision should are used to cover bare areas. Postoperative care
be made regarding which fingertip goes to which is the same as for other syndactyly surgical
finger. An allocation should be then made by con- procedures.
sidering the survivability of the distal part as well as
the resulting length and stability. Preservation of
Reconstruction for Digital Hypoplasia
the distal tips is preferred over amputation be-
and Amputation
cause the tips may contain phalangeal buds that
can be associated with articular spaces. Osteoto- Many procedures have been described for the
mies can be performed to straighten severely an- treatment of digital hypoplasia and amputation
gulated fingers. Every effort is made to preserve associated with constriction band syndrome, in-
digital length, which can be reconstructed when cluding on-top plasty, toe-to-hand transfer, web

Fig. 5. (A) Acrosyndactyly of the index and long fingers. (B) Appearance after separation of the fingers.
Constriction Band Syndrome 263

space deepening, pollicization procedures, and SUMMARY


bone lengthening procedures.32,33 Management
is aimed primarily at restoring basic hand func- Constriction band syndrome is an uncommon con-
tion, specifically power grasp and precision pinch, genital abnormality with multiple disfiguring and
and secondarily at improving the cosmetic disabling manifestations. Early amnion rupture
appearance, which is inevitably going to be with subsequent entanglement of fetal parts by am-
impaired. If the function of the hand is acceptable, niotic strands is the current primary theory for the
no treatment may be a reasonable alternative.11 development of this syndrome. Management of
As described previously, the thumb is preserved constriction band syndrome is focused on improv-
in most patients with constriction band syndrome, ing function and development while providing
and thus treatment is often directed toward im- a more acceptable esthetic appearance. The treat-
proving the functions of the remaining fingers. ment should be tailored to the individual. Timing of
The structures proximal to the level of amputation repair and surgical planning are important to pro-
are normal, making toe-to-hand transfer an at- vide the best functional results for affected hands.
tractive consideration. Toe-to-hand transfer is pri-
marily performed at the ulnar digits to provide REFERENCES
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