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Biterminal tenotomy for the treatment of congenital muscular torticollis.

Long-term results
CJ Wirth, FW Hagena, N Wuelker and WE Siebert
J Bone Joint Surg Am. 1992;74:427-434.

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 Copyright 1992 by The JournalofBont. andioini Surgery, Incorporated

Biterminal Tenotomy for the Treatment of

Congenital Muscular Torticollis
LONG-TERM RESULTS*

BY C. J. wIRTII. M.D.t. F-V.’. HAGENA. M.D4. N. WUELKER. M.D.t. AND W. E. SIEBERT. M.D.t. EIANNOVER. GERMANY

In vestigation performed at I/ic Ort/iopaedic Departmemiis of Hannover Medical School. Hannover, amid Ludwig-Maxirnilians- Universit: Manic/i

ABSTRACT: Fifty-five patients who, in a twenty- of the cervical spine, tontico!lis due to acute rheumatoid
eight-year period, had been managed with a biterminal arthritis on other inflammation around the neck, torticol-
open release because of congenital muscular torticollis, lis due to vestibular dysfunction or disease of the eye’7,
were re-examined at an average of fifteen years and a and from spasmodic torticollis.
minimum of five years after the operation. Forty-eight The etiology of congenital muscular torticollis is still
patients reported no functional or cosmetic impairment. unclear. Trauma at birth with subsequent hematoma and
Palpable soft-tissue strands remained in twenty-nine myositis of the stennocleidomastoid muscle or with vas-
patients, but bending toward the untreated side was cular occlusion has been descnibed35, but a direct nela-
limited more than 10 degrees in only one patient. The tionship to the histological changes of congenital
rate of recurrence was 2 per cent. Facial asymmetry muscular tonticollis has not been demonstrated. The fre-
improved or resolved in more than one-half of the pa- quent occurrence of congenital muscular tonticollis after
tients. We recommend that biterminal release be per- a complicated pregnancy or deliveny” has been linked
formed at the age of three to five years in all patients to abnormal intrauterine position and to trauma at
who do not respond to non-operative treatment. bimth’27’. Heredity appears to play a certain role in the
etiology of congenital muscular tonticollis””47; how-
Congenital muscular tonticollis is the third most com- ever, no precise path of hereditary transmission has been
mon congenital deformity3’,with only dysplasia ofthe hip identified.
and pes equinovarus being more common. The cosmetic Several associations of congenital muscular tonticol-
deformity causes most parents to seek medical cane for lis with congenital disorders of the hip’, with facial
their child, but late sequelae such as facial asymmetry. asymmetry’5’,and with congenital club foot4’52 have been
scoliosis, and disturbed vision can occur. reported.
Several authors have reported a preponderance of The lowest rate of recurrence of congenital muscu-
involvement of the right side compared with the left side lan tonticollis has been reported after biterminal open
of approximately 15 per cent in patients who have con- melease’’5”24”4249, which is the technique that we prefer.
genital muscular tonticollis’2’>’4. Neither sex has been The purpose of this study was to evaluate the effective-
reported to be affected more often than the other. A ness of bitenminal open release in patients who had
soft-tissue swelling over the sternocleidomastoid muscle long-term follow-up. The frequency of recurrence and
is noticed during the first weeks of life in approximately complications, the optimum timing of the operation, and
20 per cent of all patients who have congenital muscular the prognosis for facial asymmetry were of particular
tonticollis6. The swelling then may progress gradually into interest.
a contracture of the stennocleidomastoid muscle. How-
Materials and Methods
ever, the rate of spontaneous remission before the age of
one year has been reported to be as high as 90 per Fifty-five (85 per cent) of sixty-five patients in whom
cent”24. Histologically, the muscle is replaced by increas- congenital muscular torticollis had been treated with
ingly dense and cell-depleted fibrous tissue’2749. This is biterminal tenotomy were ne-examined five to twenty-
also referred to as fibromatosis colli49. seven years (average, fifteen years) postoperatively. One
Congenital muscular tonticollis has to be diffenenti- of the original sixty-five patients had died and nine had
ated from torticollis due to congenital osseous deformity moved to other locations and were unavailable for fol-
low-up. The ages of the fifty-five patients at the time of
*No benefits in any form have been received or will be received
the operation ranged from seven months to twenty-nine
from a commercial party related directly or indirectly to the subject of
this article. No funds were received in support of this study.
years and averaged approximately eight years. Most pa-
tOrthopaedic Department. Hannover Medical School. 3000 Han- tients were operated on between the ages of six and ten
nover 61 . Germany. Please address requests for reprints to Dr. Wirth, years. Three patients (6 per cent) were adults at the time
P.O. Box 610 172.
of the operation.
lOrthopaedic Department. Ludwig-Maximilians-University.
Marchioninistrasse IS, 80(X) Munich 70. Germany. The right sternocleidomastoid muscle was affected in

Vol.. 74-A, NO. 3. MAR(H 1992 427

428 C. J. WIRTH. F.-w. HAGENA, N. WUELKER. AND w. E.

FIG. 1-A FK;. 1-B FIG. I-C

Figs. 1-A. 1-B, and 1-C: Grading of facial asymmetry.
Fig. 1-A: Severe asymmetry.
Fig. 1-B: Slight asymmetry.
Fig. 1-C: Absent asymmetry.

thirty-five patients and the left, in twenty. Thirty-three done by three independent examiners, and agreement
patients were male and twenty-two, female. Two patients was reached by discussion. There was no profound dis-
had a positive family history for muscular torticollis: the agreement. Slight facial asymmetry was found in thirty-
father of one and the cousin of the other were affected. two patients and severe facial asymmetry, in twenty-two
Twenty-eight patients (51 pen cent) were first-born chil- patients. Only one child had no asymmetry. Facial asym-
dren. Twenty-five patients (46 per cent) had been born in metry was equally distributed in all of the age-groups. A
a breech position. Forceps had been used in the delivery complete examination of the eyes was carried out preop-
of eight patients (15 per cent). Combinations of risk eratively for all children. There were no markedly abnor-
factors during pregnancy and delivery were common: mal findings.
fifteen patients were both first-born babies and had been Preoperative radiographs of the cervical spine, which
born in a breech position; seven were first-born babies had been made for all of the patients, were available for
who had been delivered with the use of forceps; four had thirty-one patients at the time of the latest follow-up.
been born in a breech position and had been delivered Fourteen of the thirty-one patients had an s-shaped cem-
with forceps;and another four were first-born babies, had vicothonacic scoliosis, three had a c-shaped scoliosis, and
been born in a breech position, and had been delivered fourteen patients had no scoliosis. The c-shaped scolioses
with forceps. were confined to children who were one to four years old
Four patients (7 pen cent) had another congenital ( average, two years old), whereas s-shaped scolioses ap-
abnormality: one had a congenital dislocation of the hip; peared in all age-groups. Radiographs of the cervical
two, dysplasia of the hip; and one, metatarsus adductus. spine in two planes and anteropostenion radiographs of
Congenital muscular torticollis was diagnosed imme- the thoracic spine were included in the follow-up exam-
diately after birth in almost half of the patients. For the ination.
others, the time of the diagnosis was distributed equally
Operative Techniquefor Biterminal Tenotomy
among the first year of life, the ages of one to six years,
and the time of the examination for entrance into school. With the patient supine, the sternocleidomastoid
Four of the fifty-five patients had been operated on muscle is put under tension by hyperextension of the
unsuccessfully elsewhere: two patients had had a subcu- neck over a stack of towels and notation of the head
taneous distal tenotomy; one, an open distal tenotomy; toward the shoulder of the unaffected side. The entire
and one, a subcutaneous distal tenotomy followed by a area of the sternocleidomastoid muscle is shaved and
proximal, cepha!ad tenotomy. prepared stenilely, with the ear taped anteriorly.
Complete preoperative photographic documenta-
Proximal (Cephalad) Tenotomy (Figs. 2-A and 2-B)
tion was available for all patients. For the grading of facial
asymmetry (Figs. 1-A, 1-B, and 1-C), one line was drawn The incision is placed longitudinally over the cranial
in the orientation of the eyes and another, in the onien- portion of the stemnocleidomastoid muscle. It is not lo-
tation of the mouth. Asymmetry was classified as absent cated near the anterior margin of the muscular insertion,
if the two lines were parallel, slight if the angle formed so that injury to the facial nerve is avoided. After tran-
by two lines was less than 3 degrees, and severe if the section ofthe platysma and the cervical fascia,the cranial
angle was 3 degrees or more. The measurements were insertion of the sternocleidomastoid is dissected, mostly

THE JOURNAL OF BONE AND JOINT SURGERY

 BITERMINAL TENOTOMY FOR THE TREATMENT OF CONGENITAL MUSCULAR TORTICOI.LIS 429

L
FIG. 2-A Fio. 2-B
Figs. 2-A and 2-B: Cephalad mastoidal tenotomy. I = sternocleidomastoid muscle and 2 = auricular artery.
Fig. 2-A: The cephalad insertion of the sternocleidomastoid is divided close to the bone. Transection should he carried out from dorsal to
ventral in order to avoid damage to the facial nerve, and the facial muscles must he closely observed.
Fig. 2-B: After transection of the muscle and deep cervical fascia, the auricular artery becomes visible.

by blunt dissection with a sponge. The surgeon carefully tomical course. The muscle is undermined with a Kocher
avoids the facial nerve, which is located directly anterior sound. Electrical stimulation before transection is some-
to the cranial insertion, and the accessory nerve, which is times needed to ensure that no neural structures remain
located farther caudally, by staying away from their ana- attached to the muscle; it was used in 20 per cent of

H”

,‘

FIG. 3-A FI;. 3-B

Figs. 3-A and 3-B: Caudad sternal and clavicular tenotomy. I = sternal insertion of the sternocleidomastoid. 2 = clavicular insertion of the
sternocleidomastoid, 3 = internal jugular vein. 4 = subclavian vein. S = external jugular vein. 6 = transverse vein of the neck. and 7 = phrenic
nerve.
Fig. 3-A: With a Kocher sound placed underneath it, the sternal insertion is transected. The clavicular insertion of the sternocleidomastoid
and the deep cervical fascia have already been divided.
Fig. 3-B: After division of the deep cervical fascia, the anatomical proximity of neurovascular structures becomes obvious.

VOL. 74-A, NO. 3. MARCH 1992

430 C. J. WIRTH. F.-W. HAGENA. N. WUELKER. AND W. E. SIEBERT

FI;. 4-A Fi. 4-B

Fig. 4-A: Wry neck with severe facial asymmetry at the age of five years.
Fig. 4-B: Complete resolution twenty-two years postoperatively.

the patients. Electrical stimulation is particularly helpful position of the head, such as the sternocleidomastoid
in revision operations in the patients who have marked muscle fascia or the deep cervical fascia, are identified
scarring. The surgeon divides the muscle completely by and carefully divided.
cutting onto the Kocher sound, preferably in a ventral
Distal (Caudad) Tenotomy (Figs. 3-A and 3-B)
direction and with close observation ofthe facial muscles.
Additional structures preventing full correction of the The skin is incised horizontally, just cranial to the

Fi;. S-A Fii;. S-B

Fig. S-A: Wry neck with severe facial asymmetry at the age of two years.
Fig. S-B: Incomplete resolution twenty-three years postoperatively.

THE JOURNAL OF BONE AND JOINT SURGERY

 BITERMINAL TENOTOMY FOR THE TREATMENT OF CoNGENITAL MUSCULAR TORTICOLLIS 431

clavicle, with the incision centered between the two in- FABLE 1

sentions of the sternocleidomastoid muscle. The subcu- FAcIAI ASYMMETRY AilIIE FoLI.ow.tlI’ ExAsIINAlloN RtL1vI’IvI ii)

taneous tissues and the platysma are divided sharply. ‘II IL A;t Al II IF TF1NIE UI II II Ort-tvIIoN

Dissection is then carried out, bluntly with a sponge.

Facial Age at the Time of the Operation (No. of patie,il.s)
around the insertions of the muscle, and a Kocher sound Asymmetry SYrs. 6-lOYrs >lOYrs. Total
is placed. An assistant places the lateral structures of the
Unchanged 8 12 6 26
neck under tension, and additional strands of contracted Improved 14 II 4 29
tissue, such as the deep cervical fascia lateral to the Total 22 23 10 55

sternocleidomastoid muscle, are identified and divided.

Only the subcutaneous tissue and skin are closed. in two
layers, for both the cephalad and caudad incisions. one and 20 degrees in the other. The patients felt more
handicapped by limitation of notation than by limitation
Postoperative Management
of bending.
All but three patients were placed in a Minerva cast Palpable soft-tissue strands remained in twenty-nine
immediately after the operation, while they were still patients (52 pen cent). From their consistency on palpa-
under anesthesia. The head was rotated toward the op- tion, these stnands were thought to be muscle tissue in
eratively treated side and was bent laterally toward the nineteen patients and scar tissue in ten. Most ofthe bands
opposite side. Two weeks postoperatively, the cast was were localized toward the clavicle. Three of the patients
changed. with the patient standing. The total duration of who had strands had been operated on for recurrent
immobilization in the described position was six weeks. congenital muscular torticollis. Lateral bands caused lim-
One eight-year-old child, one sixteen-year-old adoles- itation of bending of 10 degrees or less toward the un-
cent, and one adult had no postoperative immobilization, treated side in five patients and of more than 10 degrees
because they were highly motivated and it was thought in one patient.
that they would cooperate well with physiotherapy. Sev- An unsightly scar at the site of the clavicular incision
enteen of the patients who had been immobilized were was noted in two patients, who had been operated on at
neadmitted to the hospital for two weeks for removal of the ages of six and nineteen years.
the cast and physiotherapy. The remaining patients were One recurrence. with re-formation of a soft-tissue
referred back to their home physicians. After removal of strand and a tilt of the head, was noted at the time of the
the cast, the patients were managed mostly with straight- latest follow-up examination, so the rate of recurrence
ening and muscle-strengthening exercises, under the su- was 2 per cent. This patient had been managed with two
pervision of a physiotherapist, for at least three months. operations for congenital muscular torticollis before
Additional training was performed in front of a mirror, being referred to us at the age of five years. She was
by the patient alone or by the parents. The duration of ne-examined fourteen years postoperatively.
postoperative physiothe rapy ave raged six months.

Complications TABLE II

Intraoperatively, there was one injury to the external FACIAL AsYsIsIviRY AT1IIF FoII.ow.t1P ExA,IINArIoN

jugular vein. There were no nerve injuries. Postopera- RELATIVE ‘10 FIlE PosIoI’IRArIvl INI1RVAE.

tively, functional paralysis of the deltoid muscle was

Facial Posopeavc1ntervaI(No.ofPat,nts)
noted two days after a revision operation in a sixteen- Asymmetry S-9Yrs. IO-I4Yrs. IS-I9Yrs. >l9Yrs. Total
year-old girl. This was related to passive overcornection
Unchanged 8 2 26
in the cast, and it resolved after immediate removal Improved 2 I1 8 8 29
of the cast. No evidence of damage to the axillary nerve Total 9 19 17 10 55
was found on electromyographic examination. A par-
tial paralysis of the cervical plexus developed four days
postoperatively in a fourteen-year-old girl; it resolved Preoperatively, the asymmetry of the face had been
completely two weeks after immediate removal of the graded as severe in twenty-two patients, slight in thirty-
cast. two, and absent in one patient. Facial asymmetry at the
time of follow-up was graded as severe in seven patients,
Results slight in twenty-seven, and absent in twenty-one. Asym-
All of the patients had full flexion and extension of metry had thus improved by one on two grades in more
the cervical spine at the follow-up examination. Lateral than one-halfofthe patients (Figs.4-A through 5-B).The
bending toward the untreated side was limited in eleven asymmetry improved more often in younger patients
patients (20 pen cent): the limitation was 10 degrees or (Table I): it improved in fourteen of twenty-two patients
less in ten of these patients and 20 degrees in one patient. who had been operated on when they were five years old
Two patients (4 pen cent) had limitation of notation of the or younger, in eleven of twenty-three who had been
head toward the treated side:it was limited 10 degrees in operated on between the ages of six to ten years. and in

VOL. 74.A, NO. 3. MARCII 1992

432 C. J. WIRTH. F.-W. HAGENA. N. WUEI.KER. ANt) W. E. SIEBERF

only four of ten who had been operated on after the age Non-operative treatment ofcongenital muscular ton-
of ten. Severe asymmetry resolved completely only when ticollis, therefore, has its place during the first year of
the operation had been done when the patient was five life’4’3. Simple measures. such as positioning of the bed
years old or younger. relative to the window or placement of toys above the
The longer the postoperative interval, the more the infant so that he or she will turn the head toward the
asymmetry was noted to have improved (Table II). It had affected side, are the first steps of treatment’3”7. Gentle
improved in only two of nine patients who had been stretching exercises should be performed by the physio-
followed for five to nine years, in eleven of nineteen therapist or parents several times daily. A number of
patients who had been followed for ten to fourteen years. appliances have been designed for correction of wry-
in eight of seventeen patients who had been followed for neck deformity’’’. Congenital muscular torticollis that
fifteen to nineteen years. and in eight of ten patients who is present primarily at birth is less responsive to non-
had been followed for more than nineteen years. This operative treatment. The over-all rate of failure of non-
relationship
tive interval
between
was most
improvement
obvious in the
and the
patients who had
postopera- operative
percent
, treatment
cc
has been reported to average 5.3

been operated on before the age of five years. Severe Subcutaneous tenotomy was introduced during the
asymmetry had resolved completely only in the patients nineteenth century and it is still being advocated5”3”45.
who had been followed for eleven to twenty years post- However, injuries to the external jugular and subclavian
operatively. veins’9’7’ and to the brachial plexus5, and even lethal air
Eight patients had a c-shaped spinal scoliosis at the embolism5, have occurred with this technique. Also, the
follow-up radiographic examination. The scoliosis af- procedure does not allow release of additional soft-tissue
fected ten to sixteen segments (average, twelve seg- contractures such as those of the deep cervical fascia.
ments), and the Cobb angle ranged from 6 to 12 degrees Caudad tenotomy of the sternal and clavicular insertions
(average. 8 degrees). Two patients had an s-shaped scoli- (Figs. 3-A and 3-B) became increasingly popular during
osis: in one. the primary curve involved six segments and the end of the nineteenth century5. The rate of injury to
measured 8 degrees and, in the other. the primary curve the external jugular vein has been reported to be 3.3 pen
involved seven segments and measured 12 degrees. The cent’5”9, and the over-all rate of recurrence has been
secondary curve involved seven segments in one of these almost 7 per cent”5’9””3””’25’79”’42’. Cephalad mastoidal
patients and ten segments in the other. Both secondary tenotomy (Figs. 2-A and 2-B) was advocated in the be-
curves measured 10 degrees. Compared with the findings ginning of the twentieth century>’49. Its main advantage
on preoperative radiographs that were available for is that it corrects the position of the head even when there
thirty-one patients, these findings suggested complete are adhesions between the stennocleidomastoid muscle
resolution of the scoliosis in two-thirds of the patients. and the surrounding fascia. Injury to the facial nerve has
With regard to the jen patients who had scoliosis at the been reported in 3.4 per cent of patients3’”> and injury to
time of follow-up, a primarily c-shaped scoliosis had the accessory nerve, in 2.4 per cent3. The over-all nate of
developed into an s-shaped scoliosis in one of them and recurrence has been I 1 .5 per cent””42. Total resection of
an s-shaped scoliosis had developed in a patient who had the contracted sternocleidomastoid muscle2’7”’37 should
had no curve preoperatively. The age at the time of the be limited to patients who have severe deformity and to
operation did not influence the outcome with regard to revision procedures. The carotid artery.jugulan vein, and
scoliosis. Complete resolution of s-shaped scoliosis. how- vagus. accessory, phrenic, and facial nerves are at risk,
ever, was seen mostly in infants, and a higher prevalence with a prevalence of injury of as high as 13.6 per cent3”7.
of persistent secondary curves was observed in the olden With biterminal tenotomy2, the sternal and clavic-
patients.The duration ofthe postoperative follow-up did ular insertions of the sternocleidomastoid are divided
not influence the result with regard to scoliosis. However, through an open approach. The rate of complications
scoliosis improved more rapidly in young children than with this technique has been reported to be no higher
in older patients. than that with open, caudad tenotomy of the sterno-
cleidomastoid or with cephalad mastoidal tenotomy
Discussion
alone”’9. Due to its flexibility and the reportedly low
Operative treatment of congenital muscular torticol- (2.9 per cent) rate of recurrenceSSS2S42, we use this
lis is generally considered to be indicated if the sterno- technique most often.
mastoid pseudotumon persists beyond the age of six Postoperatively. a Minerva cast was used for six
months”. if wry neck persists beyond the age of twelve weeks9”9 to maintain reduction. Only a few authors7”7
months”2, or if the electromyographic activity of the have advocated early range-of-motion exercises without
affected sternocleidomastoid muscle is markedly me- external fixation. However, in recent years. we have had
duced4. Release of the stemnocleidomastoid is not mdi- good results with exercises that were begun during the
cated in patients in whom wry neck is due to other causes, first postoperative days by fully cooperative patients.
such as osseous deformity. and it is not indicated for the Now, if there is no reason to believe preoperatively that
treatment of ocular or spastic tonticollis. the patient will not cooperate after the operation, no cast

TIlE J()I,’RNAI. OF BONE AND JOINT SURGERY

 BITERMINAL TENOTOMY FOR THE TREATMENT OF CONGENITAL MUSCULAR TORTICOLLIS 433

is applied. Physiotherapy is begun on the first postoper- secondary changes, such as facial asymmetry, to correct
ative day, and our therapists see their patients several postoperatively. Recommendations about the age at
times daily. Since 1985, we have not had to apply a cast which the procedure should be performed have ranged
in the postoperative period due to lack of cooperation by from six months3’5 to ten years and older39. If secondary
the patient. changes progress or do not resolve completely, as some
Both postoperative complications in this series were authors have suggested’543’’, the patient should be op-
related to passive ovencommection in the cast, as has been enated on during the first twelve months oflife. However,
described previously5. If immobilization in a cast is con- if these changes do connect, as other authors have sug-
sidered necessary, meticulous attention must be be paid gested2”24334’, and in view ofthe high rate of spontaneous
to proper technique. remission that has been reported during the first year of
Rates of recurrence of 0.8 to 20 per cent have been life”24, the operation should be delayed. In addition, it is
reported after treatment of congenital muscular tomticol- preferable that the child be old enough to cooperate with
lis with biterminal tenotomySS224A42. The 1.8 per cent postoperative rehabilitation so that recurrence can be
rate in the present study is comparable with the rates avoided.
reported by other authors, especially when the relatively The results of the present study indicate that even
olden age of our patients at the time of the operation is severe facial asymmetry may resolve completely within
taken into account. The results of the revisions for treat- a few years if the patient is operated on before the age
ment of recurrent congenital muscular torticollis natu- of five years. Less and slower improvement can be ex-
rally were inferior to those of the primary operations. pected afterward. Our experience suggests that the op-
Formation of scam tissue made precise localization in the emation should be performed between the ages of three
tissue difficult and impeded radical resection of the tis- and five years. Patients of this age can be reasonably
sue. In addition, patients who had a recurrence may have cooperative during postoperative physiotherapy, and a
had a more severe form of the disease in the first place. low mate ofrecummence can be expected. Facial asymmetry
The best timing of the operation is still being debated, will correct or improve in most patients, and the patient
mostly because opinions differ about the capability of does not miss school.

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