INTRODUCTION
Congenital cleft hand (CCH) was originally classified as typical versus atypical cleft
hand1. With the advancement of genetics and molecular biology, the atypical cleft hand
was reclassified as a teratological sequence of symbrachydactyly2.
This anomaly is characterized by the “V” shape, which may be associated with the absence
of one or more digits, and may be unilateral or bilateral, with or without the involvement
of the feet3,4. Generally, it is an autosomal dominant inheritance, with variable penetrance and
expressiveness4.
Resulting from a longitudinal deficiency of the central rays of the hand, CCH can
range from a simple skin cleft of soft tissues to the suppression of all rays except
the smallest digit5. Based on the three axes of hand and upper limb development, CCH is currently classified
as hand plate malformations - abnormal axis differentiation (patterning/late limb
differentiation)6.
Manske & Halikis and Sharma and Sharma stand out among the most used classifications.
The first is based on the involvement of the first commissure7. The second provides a complete hand description and assigns a numerical value to
each element, with the subsequent recommendation of the indicated surgical procedure8.
Indications for surgical treatment range from space deficiency in the first commissure,
absence of the thumb, progressive deformity to severe flexion contractures of one
or more fingers4,9. However, this topic remains controversial and challenging, especially due to the
patient’s adaptation to the deformity and acceptable functionality of the limb10.
OBJECTIVE
This literature review aims to present the classifications, the most relevant surgical
techniques reported in the literature and the results obtained from the studies included.
METHODS
Databases and research
The bibliographic search was carried out between April and October 2020 in journals
indexed in the Web of Science, PubMed, Scopus, Cochrane and Embase databases. The
search terms used were a combination of “Typical Cleft Hand,” “Cleft Hand,” “Ectrodactyly,”
“Central hand,” “Central ray deficiency,” “EEC syndrome,” “Cleft hand,” “Cleft-hand”
, “Cleft- Hand Malformation”, “Lobster claw”, “Fingers/ abnormalities”, “Muscle, Skeletal/abnormalities”,
“Hand Deformities, Congenital/pathology”, “Trigger Finger Disorder/congenital”, “SHFM”,
“Collateral Ligaments/surgery”, “Hand Deformities, Congenital/ surgery”, “Metacarpophalangeal
Joint/surgery”, “Tendon Transfer”, “Surgical Flaps”, “Suture Techniques”, “Syndactyly/surgery”,
“Reconstructive Surgical Procedures”, “Hand deformities, Congenital/ surgery”, “Fingers/surgery”,
“Congenital/surgery”, “Treatment”, and other related terms. All records returned by
the search were imported into Mendeley’s bibliographic management software, and duplicate
publications were removed. We also identified relevant articles through bibliographic
linking with eligible articles.
Selection of studies
The included studies were related to CCH and may have the following approaches: classification
of the anomaly, classification of severity of expression, surgical techniques, intervention
or cohort of patients undergoing surgical treatment. The search did not limit language
or study design. For intervention analysis, in order to observe current practical
trends, the search was restricted to studies published between 2000 and 2020.
Studies that analyzed patients with cleft hands resulting from trauma sequelae or
syndromic association, review articles or secondary analyses and publications that
were incomplete or did not provide sufficient data for one of the outcomes of interest
were excluded. Patient cohort data from studies approaching surgical techniques were
not used for interventional analyses.
Data extraction
Two independent investigators reviewed the search results to select eligible studies
using pre-established inclusion and exclusion criteria. Disagreeing decisions were
discussed with a third reviewer. Data were extracted using a form according to predefined
variables for each analysis. In order to summarize the findings in the literature,
we chose to include a topic unifying surgical techniques and classification of severity
of expression according to suggestive reports observed in the literature.
RESULTS
Selection of studies
Five hundred twenty-seven studies were identified by searching the descriptors in
the databases. Three hundred sixty-nine articles were excluded due to duplicity or
by title, abstract and/or keywords. After applying the determined inclusion and exclusion
criteria, 36 were considered potentially eligible; four were discarded based on clinical
outcome, lack of data or inadequate study design, resulting in 32 studies for analysis
of results. In Figure 1, the flowchart of the search for studies in the chosen databases is represented.
Figure 1 - Prism flowchart for literature review.
Figure 1 - Prism flowchart for literature review.
Analyses of included studies
The included studies were divided into four groups: (1) congenital anomaly classification
(n=8); (2) expression severity rating (n=10); (3) surgical techniques (n=12); and
(4) intervention analysis (n=4). Of these, only two studies met the inclusion criteria
in two concomitant groups; therefore, they were counted as unique inclusions11,12. Therefore, the main features of the 32 included studies stratified into groups are
presented in subsequent topics.
CCH functional classification
The classification system for congenital limb malformations was developed by Swanson
et al.13, based on the grouping of anomalies according to the part affected during development.
This system, accepted by the American Society for Surgery of the Hand (ASSH), the
International Federation of Societies for Surgery of the Hand (IFSSH) and the International
Society for Prosthetics and Orthotics (ISPO), is called the IFSSH classification14. Subsequently, the Japanese Society for Surgery of the Hand (JSSH) suggested modifications
to include two groups: “Abnormal lightning induction” and “Unclassifiable cases “15,16.
A new classification for congenital anomalies of the upper extremity, known as the
OMT classification and considered an alternative to the Swanson, Barsky and Entin
classification17,18, was presented by Oberg et al.19 in 2010. Since its publication, the OMT classification has been vigorously evaluated
by several authors for its usefulness and reliability, and recently, in 2020, an update
was published6.
Therefore, according to the current classification, CCH is classified as IB1IV: I-
Malformation; B- Hand plate: abnormal axis differentiation (late limb standardization/
differentiation); 1- Proximaldistal Axis; IV: Cleft hand (cleft foot/hand malformation)6.
Congenital differences can also be classified according to their severity of expression,
which can help in the functional determination and treatment orientation. Due to the
unpredictability and peculiarity of the phenotypic presentations of this anomaly,
a large number of classification systems have been proposed, which may be based on
the number of defective rays11,20,21, teratological mechanism of aplasia and synostosis22, first commissure contracture7,12, the complexity of associated anomalies23 and radiological morphology and cleft position - medioulnar and ulnar24.
More current classifications tend to present a greater complexity of information.
Valenti et al.25 proposed a classification based on six groups, each with a therapeutic strategy based
on describing all clinical and radiographic abnormalities observed. In line with this,
Sharma & Sharma8 described a new comprehensive functional classification considering all morphological
determinants of the anomaly, such as absent digits, associated anomalies, cleft location
and thumb functional status, calling it DAST8.
Among these, the most widely used clinical classification is that of Manske & Halikis7, which is based on the condition of the first commissure, with type I being normal
(normal web); type II (narrow web) with moderate (IIA) or severe (IIB) narrowing;
type III (syndactylized web) fused first commissure, syndactyly between thumb and
forefinger; type IV (merged web) the first commissure included in the cleft, there
is no index finger and the thumb is unstable; and type V (absent web) with the absence
of the commissure due to the absence of the thumb and forefinger.
The most recent functional classification with multivariate analysis, advocated by
Sharma & Sharma8, can be presented as follows:
Type I: described as having a normal first commissure, characterized by having the
first commissure not reduced, with a slight cleft and no abnormal bone;
Type IIA: described as having a slightly narrowed first commissure, characterized
by having a slightly reduced first commissure and abnormal bone;
Type IIB: described as having a severely narrow first commissure, characterized by
a severely reduced first commissure, abnormal bony syndactyly;
Type III: described as having thumb/ index syndactyly without first commissure, characterized
by fused first commissure, syndactyly between thumb and second finger and abnormal
bone;
Type IV: described as having the first commissure included in the cleft, characterized
by the suppression of the second finger and syndactyly of the ulnar digit;
Type V: described as having an absent first commissure, characterized by the absence
of the thumb.
As a general rule, a sum of scores greater than 4 or individual scores in any morphological
determinant of the anomaly greater than 2 indicates a potentially more complex deformity
with less possibility of satisfactory functional and aesthetic results8.
Surgical technique
Recommended surgical procedures for treating central deficiency include cleft closure,
reduction of the intermetacarpal space, the release of syndactyly, and excision of
polydactyl or transverse bone elements when present. Different techniques can reduce
intermetacarpal space and interventions secondary to cleft closure, and the most frequently
reported in the literature, along with indications, advantages and disadvantages,
as reported by the study when available, are listed in Figure 2.
Figure 2 - Recommended treatment according to classification.
Figure 2 - Recommended treatment according to classification.
Surgical technique according to expression severity classification
Grading systems are essential to facilitate communication and guide surgical reconstruction.
Therefore, we present the behaviors most commonly reported by the included studies,
stratified according to the classification by Manske & Halikis7.
In order to expose the advances in knowledge of this anomaly, we present in Figure 3 the Manske & Halikis7 classification and the recommendations for procedures suggested by some authors of
the new classification system for hands with typical clefts using the DAST8 classification.
Figure 3 - DAST classification for cleft hand.
Figure 3 - DAST classification for cleft hand.
Intervention analysis
Surgical information from cohort studies of patients undergoing procedures for the
treatment of cleft hands is presented in Chart 1.
Chart 1 - Surgical techniques, advantages and disadvantages.
| Author, Year |
Technique |
Advantages disadvantages |
| Barsky, 19641 |
Uses a retail place, pedicle foursquare, in diamond shape to recreate the commissure
after the cleft is narrowed.
Indication: Cleft hand.
|
Benefits: Aesthetic improvement.
Disadvantages: Insufficient functional concern; without reconstruction of the first commissure.
|
| Snow and Littler, 196726 |
The cleft is elevated like a palmar flap, with a small radial flap preserved by recreating
the commissure; the first commissure space is freed, which may require splitting the
dorsal interosseous and surrounding fascia; the second metacarpal is transposed and
attached to the remainder of the third metacarpal base; Fixation is obtained with
axial and transverse wires, and the palmar flap is transferred, recreating the new
commissure between the second and fourth ray.
Indication: Third metacarpal segment present.
|
Benefits: Functional; Gain cosmetic.
Disadvantages: Risk of distal flap necrosis due to its high length-to-base ratio; Traction of the
adductor and dorsal interosseous muscles may cause some radial angulation in the local
translocation, incompletely correcting the central cleft.
|
| Miura and Komada, 197927 |
Index transposition in a central position and palmar and dorsal redesign as separate
flaps to create the first commissure. Cleft incised from side to side. The index finger
is raised in its neurovascular bundles and transposed by osteosynthesis with the third
metacarpal or by angulation osteotomy.
Indication: Cleft hand with an adducted thumb.
|
Advantage: Small flaps of random transposition of the dorsal and palmar skin.
Disadvantage: Incidence in necrosis distal and contracture secondary
|
| Ueba, 198128 |
Use of transverse flaps from any edge of the cleft and transposition of the index
digit; Reconstruction of the intermetacarpal ligament by a free tendon.
Indication: Total absence of the third metacarpal.
|
Advantage: Improved aesthetics without changing the function of the hand.
Disadvantage aesthetic of transferring the palmar to dorsal skin and dorsal to palmar skin.
|
| Buck-Gramcko, 198529 |
Cleft narrowing, syndactyly separation, crossbones removal, correction of joint flexion
contractures, rotation or wedge osteotomies for axial deviations and ulnar translocation
of the index digit.
Indication: Deep intermetacarpalpal ligament reconstruction.
|
Advantages: Cosmetically acceptable without translocation.
Disadvantages: Inadequate correction of the central space.
|
| Ogino, 199011 |
The index and ring fingers reconstruct the deep metacarpaltransverse ligament using
flexor sheaths (part of the A1 or A2 pulleys).
Indication: Total absence of the third metacarpal.
|
Advantages: Possibility of spontaneous correction of the deformity in flexion of the ring finger.
Disadvantages: -
|
| Upton, 200412 |
Wide incision that extends from the ulnar side of the cleft around the malpositioned
index digit to the thumb; may include index transposition, metacarpal and/or phalangeal
osteotomies, joint releases, phalangeal osteotomies, adductor pollicis muscle preservation,
first dorsal interosseous muscle release, syndactyly separation(s), and thumb duplication
correction.
Indication: Typical cleft hand.
|
Advantage: Provides clear identification of all anatomical structures of the palm.
Disadvantage: Grasp and precision maneuvers remain poor despite considerable functionality.
|
| Foucher, Loréa, Hovius, Pivato, Medina, 200630 |
Translocation in the radial direction of the ulnar finger(s) by intracarpal osteotomy;
When necessary, a synostosis metacarpal can be performed in the same procedure.
Indication: Type IIA of the Manske & Halikis classification7.
|
Advantage: No functional loss; Good alignment of the second metacarpal with the radius.
Disadvantage: Mobility between hamate and capitate is physiologically limited in all biomechanical
studies.
|
| Oberlin, Korchi, Belkheyar, Touam, MacQuillan, 200931 |
Reverse policing: The incision wraps around the second digit in the middle, extends
over the dorsal edge of the cleft, and ends on the radial side of the third digit,
where the second commissure space should be created. The index metacarpal is released
(extraperiosteally) and translocated into the space of the absent third ray. After
internal bone fixation, the flap, with its volar cutaneous pedicle, is transposed
to reconstruct the first space of the mesh.
Indication: Type II of the Manske & Halikis classification7
|
Advantages: Preservation of the dorsal venous network; no need for grafting; It does not harm
the normal musculature of the thumb.
Disadvantages: Possibility of ectopic bone deformation; Index finger misalignment; Divergence of
the metacarpals if reconstruction of the transverse metacarpal ligament is insufficient.
|
| Upton, Taghinia, 20109 |
Simple circumferential incision around the index radius; straightline incisions inside
the cleft; and thumb extensions; Elevation of the dorsal and palmar flaps to provide
exposure of the metacarpal portion of the hand; Reconstruction of the intermetacarpal
ligament employing simple sutures, circumferential sutures around adjacent metacarpals,
or by joining adjacent A1 pulleys. The new space of the first commissure is closed
by a small flap based on the radial side of the radius of the annulus with a dorsal-palmar
slope of 45°.
Indication: Type II and Type III of the Manske & Halikis7 classification.
|
Advantages: Preservation of the adductor pollicis muscle for a functional pinch.
Disadvantages: Instability of the thumb in the joint metacarpophalangeal; Progressive camptodactyly
of fingers adjacent to central cleft; Narrowing of the first commissure; Excessive
length and radial deviation of the transposed index radius; Risk of recurrence of
commissure narrowing due to scarring along the edge.
|
| Christen, Dautel, 201310 |
Dorsal skin incision; Autologous tendon graft (preferably the palmar longus, otherwise
the plantaris or a toe extensor); Positioning the tendon in a figure of eight around
the base of the proximal phalanx and corresponding metacarpal neck.
Indication: Hyperlaxity of the joint(s) metacarpophalangeal adjacent to the cleft.
|
Advantages: Prevents the occurrence of excessive progressive narrowing;
Disadvantages: Over-tightening of the graft will result in a limited range of motion in flexion
and extension, whereas under-tensioning will lead to persistent laxity.
|
| Yasin, Amin, Mahmoud, Abdel-Ghani, 202032 |
Use cleft skin as a bipedicled flap for reconstruction and widening of the first commissure’s
narrow space of the first commissure. The skin of the cleft maintains its fixations
on the dorsal and palmar surfaces of the hand, and the index digit is passed (tunneled)
under it so that the skin of the cleft will occupy the space of the U commissure.
Indication: Type IIB and Type III of the Manske & Halikis classification7.
|
Advantages: Ensures the maintenance of a good blood supply to the skin of the cleft; a rounded
edge of the U commissure without scars.
Disadvantages: In a deep fissure, the skin retains its shape after reconstructing the first commissure.
|
Chart 1 - Surgical techniques, advantages and disadvantages.
DISCUSSION
Many cases of CCH do not require surgical treatment10,33. Therefore, the follow-up to be adopted demands delicate management when outlining
the indications for intervention and the surgical plan must specifically address the
unique abnormality of the patient34. Since several authors report the psychological consequences of CCH caused by social
stigmas, members of an interdisciplinary team may be requested to advise families
with this condition35.
The main indication for correcting the deformity is restoring function as close to
normal and as early as possible, with the esthetic correction being a natural result
of the intervention8. As described in the literature, surgeons must assess the spacing of the first commissure,
the presence or absence of metacarpals in the central rays, transverse bones, syndactyly
that may be present and, especially, the vascular supply to the hand4. If the cleft hand is due to vascular insufficiency, as in cases of associated syndromes,
or if there are other concomitant deformities, a stepwise surgical correction is suggested34.
It has been observed that understanding of the etiology of congenital limb anomalies
is restricted and continues to progress, and we confirm this in the recent modification
of cleft hand subcategorization in the OMT classification update published in 20206.
A large number of classifications according to the severity of CCH expression were
found, with Manske & Halikis7 being the most frequently used, being cited in 100% of the studies included for analysis
of surgical intervention32,36,37,38,39. However, the most complete and current classification with recommendations for surgical
procedures and prognosis is that of Sharma & Sharma8.
It is important to highlight that some studies were redundant, describing modifications
of the skin incision or refining techniques already consolidated, with the main objective
of correcting the first commissure. If the first commissure is normal, so-called ‘simple
cleft closure’ is suggested, with the reconstruction of soft tissue, ligaments, tendons
and bones. The incision can be closed using different methods; the skin is usually
removed from the lateral side of the adjacent digits to form a longitudinal scar.
Often, a flap from the ulnar half of the cleft is used to increase the depth of the
first commissure, creating a wider, more functional space between the index finger
and thumb. Authors emphasize that detecting the neurovascular pedicle and careful
division of the cleft are important steps in identifying additional tendons.
According to studies included in the intervention analyses, if the second and fourth
digits remain divergent, an osteotomy of the base of one of the metacarpal bones can
be considered. In cases of transverse bones, total or partial removal is suggested,
leaving parts of it in continuity with the metacarpal joints to avoid damage to collateral
ligaments, instability or stiffness40. Reconstruction of the deep, transverse metacarpal ligament can be performed using
flexor sheaths (part of the A1 or A2 pulleys) of the index and annular digits, dorsal
base rotation graft11, tendon/ fascia grafts36, or even absorbable stitches and K-wires.
For complex cases with syndactyly of the first commissure, options are presented according
to the severity of the narrowing. If the syndactyly is proximal and loose, it can
be released with an appropriate flap (z-plasty, combined z-plasty, or rotation-transposition
flap) in association with cleft closure, as described.
For severe narrowing, attention should be paid to the distal bifurcation of the neurovascular
bundle, not only between the thumb and index but also between the ring and little
digit. If microvascular procedures cannot solve this problem, authors warn of the
possibility of necrosis41,42. Therefore, the transposition of the second ray to the third metacarpal bone (if
any) may be indicated. In advanced cases of partial or complete aplasia, transfer
of toes or bone graft covered with periosteum is suggested42. Conservative treatment is reserved for the most severe cases, in which there is
usually a great functional adaptation and in patients with severe neuropsychomotor
developmental deficit, making adequate rehabilitation impossible43,44.
It was observed that practical application studies and long-term follow-up are limited.
Only four studies that presented details of surgical intervention for a cohort of
patients undergoing cleft hand treatment were identified, as shown in Chart 2. Of these, the most frequent distribution of cleft hands was type II. No type IV
or V patients were described, a similar observation described by Manske & Halikis7, who reported that they are the rarest subtypes and are difficult to manage, a factor
that would justify the absence of studies containing these subtypes.
Chart 2 - Intervention analysis of articles published in indexed journals included in the systematic
review.
| Study (Author, year) |
Patients/hands (n) |
Mansk type and (n) |
Correction of central deficiency |
Additional procedures (n) |
Component and transverse bone (n) |
Observation |
| Rider, Grindel, Tonkin, Wood, 200045 |
12/12 |
Type IIB:
(-) Type III:
(-)
|
Snow & Littler26 |
Osteotomy: 3 Osteotomy for delta phalanx: 2 Revision of the first commissure: 3 Revision
of the syndactyly scar: 1 Religation of the cleft: 1 None: 2
|
Dorsal base rotation graft11, bone suture or tendon graft: 8
|
There were no cases of graft necrosis, although two grafts showed ischemia at the
edge; Four (36%) secondary revisions of the first commissure were performed.
|
| Goldfarb, Chia, Manske, 200836 |
12/16 |
Type I: 5
Type IIA: 7
Type IIB: 1
Type III: 3
|
Cleft reconstruction using soft tissue and/or bone procedures.* |
Osteotomy: 3 Revision of the first commissure: 2 Proximal interphalangeal joint extensions:
3 Extensor mechanism imbrication: 2 Extensor indicis proprius transfer: 1.
|
Dorsal base rotation graft11 or tendon/ fascia grafts37 wrapped around the adjacent metacarpal neck: 8.
|
Flexion contracture of the proximal interphalangeal joint of the ring finger with
a mean of 31° was the most notable clinical finding. The metacarpal divergence angle
significantly improved from 33° to 12°, and the phalangeal divergence angle improved.
significantly from 38° to 12°.
|
| Aleem, Wall, Manske, Calhoun, Goldfarb, 201438 |
18/23 |
Type I: 5
Type IIA: 9
Type IIB: 5
Type III: 4
|
Standard cleft closure; Soft tissue reconstruction alone or combined with bone transposition
of the index ray; and deepening of the first commissure space.*
|
Corrective osteotomies, tendon realignment, and soft tissue capsular tightening: 11 |
Excised with intrinsic muscles inserted: 11 |
The presence of transverse bone in the cleft hand was not associated with worse outcomes
after cleft reconstruction. The use of the forceps cleft was more dependent on the
status of the index digit and preoperative thumbindex space than on the presence of
a transverse bone.
|
| Beck, Chang, Jones, 201539 |
1/1 |
Type IIB |
Miura & Komada27 |
It was not necessary. |
Transverse osteotomy at the base of the index metacarpal; Ulnar translocation and
fixation at the base of the metacarpal of the middle finger with K wire: 1
|
All incisions and flaps healed mostly without any evidence of skin necrosis. Bone
union was present 6 weeks after surgery.
|
Chart 2 - Intervention analysis of articles published in indexed journals included in the systematic
review.
In recent studies, the Snow procedure is often related to its limitations, such as
low viability of the palmar flap, technical difficulties, and records of complications
resulting from necrosis. However, Rider et al.45, when studying the technique, observed a low flap necrosis rate, but the revision
was necessary for one third of the patients. Despite this finding, there is a relevant
use of the technique by Miura & Komada27, which is justified in the simpler design and less risk of flap necrosis while producing
similar functional and cosmetic results39. A study with longterm results of the technique by Miura & Komada27 demonstrated excellent patient satisfaction in function and esthetics39. The same occurrence was reported in the literature for interventions performed using
the Upton technique12.
In general, patients with a cleft hand due to vascular deformity are at high risk
of skin loss and poor perfusion of the surgical site after surgery, especially if
the procedure is not staged properly. In addition, finger stiffness remains the most
common postoperative complaint, despite improving functional results34. Because of this, we emphasize that the median cleft of the hand is a complex but
rare malformation that requires individualized management based on the severity of
expression.
CONCLUSION
Studies on FCM are directly affected by discoveries in embryological, genetic and
molecular biology. During the last few years, advances in these fields have led to
restructuring the classification system and understanding different presentations.
Regarding treatments, pioneering techniques include cleft closure and reconstruction
of the first commissure. The main complications described were problems with necrosis
of the distal flap and stiffness. Several studies on updating these techniques were
found. In addition to better quality research, standardization in the description
of techniques and results could elucidate existing treatment options gaps.
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1. Universidade Estadual de Campinas, Departamento de Ortopedia e Traumatologia, Campinas,
SP, Brazil.
Corresponding author: Marcela dos Santos Martins Cidade Universitária Zeferino Vaz - Barão Geraldo, Campinas, SP, Brazil Zip Code:
13083-970 E-mail: marcela.m.1509@gmail.com
Article received: May 16, 2021.
Article accepted: December 13, 2021.
Conflicts of interest: none.
Institution: Universidade Estadual de Campinas, Hospital de Clínicas, Campinas, SP,
Brazil.
