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Congenital Muscular Torticollis: Etiology and Pathology

RALPH T. LIDGE, ROBERT C. BECHTOL and CLAUDE N. LAMBERT J Bone Joint Surg Am. 1957;39:1165-1182.

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Congenital
ETIOLOGY BY
AND

Muscular
AND M.D.,
LAMBERT,

Torticollis

PATHOLOGY ROBERT
M.D.,

RALPH
CLAUDE

T.

LIDGE,
N.

C.

BECHTOL, ILLINOIS

M.D.,

CHICAGO,

This collis. causes teristics is derived Torticollis

in Websters position

article This review of congenital

is a review was made torticollis.

muscular

of

the as

important the
is

literature part
distinct

on an

the

subject

of congenital investigation into charactorticollis

tortithe

first
a

of

experimental primary

Congenital

torticollis

entity, the

pathological

of

which are limited to the sternocleidomastoideus from the Latin wordstortus, meaning twisted, may be head. the result Synonyms of irregular
dictionary as

muscle 15 The term and collum, meaning of the muscles and with stiff-neck, combined

neck. been unnatural obsticommon factors, traudescribed

contraction
twisting

has an caput

unabridged of the

the ofneck

sometimes both from are

used

include wry-neck,

crooked-neck, and twisted-neck. There are many types of torticollis, congenital types include those resulting while
matic,

pum,

congenital muscular, resulting muscular

and acquired. osseous, or from type

The most neurogenic

the

commoner
or

acquired
paralytic

types here.
HISTORICAL

hysterical,

factors. Only

those the

infectious, of torticollis,

neoplastic, congenital

in

origin,

will

be

considered

BACKGROUND

Dutch

surgeon, be

Isacius the first Fabrizio to

torticollis.

Minnius, surgeon to dAquapendente the first

performed obtain operative (1537-1619) author to

tenotomy had correction devised the case

for

torticollis

in

1685 Prefor of the

He is believed to viously, Girolama

correction Taylor congenital of torticollis.

of this condition. an apparatus symptoms

is believed muscular

be

describe the autopsy muscle

pathological

had

died that
muscle an present

at
as

six

weeks

of age of

In 1875, he presented and upon whom an the sterno-mastoid was and symptom displaced the

of a white male infant who had been done. He described tumour. He

deformity

an induration

or sterno-mastoid of fibrous tissue between the fibers and had its usual thickness. case report by a fibrous tissue between

said
of such were

the
extent

fibers.

main pathological This growth that, in of

throughout

had

growth destroyed

the bundles continued to The drawing changes of the defeel man to give a

some the the

areas, muscle. affected that He should

muscle was He illustrated muscle not showing until the

double this the

microscopic fibers. In scribed that 1893,

section Anderson

muscle

stated

eighteenth

century torticollis was

in surgical textbooks. congenital wry-neck to

said that be treated treatment

Samuel Sharp, writing in 1740, did not by surgery and that the first of this condition was
the great

serious consideration Dupuytren.

EVOLUTIONARY

surgical

French

surgeon

DEVELOPMENT

OF

THE

STERNOCLEIDOMASTOIDEUS

Straus lutionary the

origin.
.

and Howell, development known fact following have the

NO.

in a monumental paper of the sternocleidomastoideus that the information long innervation is based been puzzled

written 1936, in carefully muscle, basing their

trace the evodiscussion on of the its two primitive authors.

well The

of a muscle is a good indication entirely upon the work of these by the fact that in mammals

Anatomists

musculature

comprising
VOL. 39-A.

trapezius
5. OCTOBER

and
1957

sternocleidomastoideus

is innervated

both

by

the

spinal
1165

part

1166
of

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

the

accessory

nerve

and of

by

branches

of

the

anterior

rami

of

the of its nerve

cervical

nerves.

Since since it is deriva-

the source of innervation almost universally believed tion, it has been conjectured

a muscle that the that

is an accurate indication spinal part of the accessory the trapezius-sternocleidomastoideus musculature these muscles in origin but According to in of and the the most a

origin and is of visceral is itself

complex

of visceral origin. The double innervation of this the basis of this anatomical feature, it is felt that visceral somatic origin or (2) are primarily visceral later shifted to follow a somatic (spinal) pathway. origin of the trapezius-sternocleidomastoideus the region of the branchial arches. In the dogfish (Squalus), a trapezius the vagus lature (the tissue arising not only in upper portion lateral trapezius musculature is simply nerve which supplies sternocleidomastoideus musculature typical the largest genus

in mammals is unique. On either (1) are of compound contain components which Edgeworth, the primordial of vertebrates to branchial the rami of is

variety

Elasmobranchii, the posterior of

nerve the

segments of is undifferentiated

gill musculature. in this genus)

The consists

trapezius muscuof a single back. It inserts cartilage of the with the small indicate that which sternoand cannot musthe fish, spevagus the are

from the skin and superficial fascia over the muscles of the the shoulder musculature and in the girdle but also in the of the last branchial arch. It is thus perfectly in sequence muscles is a of the other gill posterior arches. division This of seems the to interarcualia specialized,

interarcuate musculature

branchiogenic. cleidomastoideus, more powerful be moved des can visceral especially cialization musculature branches The
being tissue

Thus, it appears that the trapezius musculature, which includes the is probably of visceral origin. In sharks, this musculature is larger and serves to move the girdle. In bony fish the membranous girdle

because of its firm attachments, as a result of which the attached move only part of the gill apparatus. Because of this anatomical muscles are much weaker in bony fish than they are in the dogfish. the Teleostei, this muscle mass is not even present as a result during evolutionary is innervated, which supply the trapezius mass
from from the

visceral difference In some of extreme of not is a broad the

so far posterior main

change. In all fish, the trapezius-sternocleidomastoideus as is known, solely by the posterior twig part of the gill musculature. in In most amphibians, Caudata, the the sternocleidomastoideus trapezius mass mass.

is also the

undivided

differentiated arising

is an that most
of the

exception it more authorities, vagus; of

in closely

superficial fascia of the back. The musculature of the common that it is very narrow and tends to arise exclusively from the resembles sole source (1913) has and second involved undivided innervated a sternocleidomastoideus of innervation however, nerves are sensory mass branch which of of than this a trapezius. According muscle in amphibians the small is extremely so far accessory In some the of

toad head to

so

the Norris the first

is a branch dorsal likely as is field of species that known, musthis

branches the
culature

reported, spinal carry only muscle by a

that Siren in lacertina also sources. It fibers. the In summary, the nerve. represents vagus

spinal

branches have
and which

amphibians

an is

musculature may The accessory dorsal side of the sertion length insertion is not trapezius
1900, development,

also be partly innervated field of musculature in shoulder girdle and from

by spinal nerves. reptiles arises from the posterior portion scapula above In species of both the

aponeurosis the cranium.

on the Its inentire the which of the in of in

is located on the anterior border of the of the clavicle, and on the interclavicle. was probably located along the length found mass
demonstrated from

the clavicle, along the of reptiles now extinct, cleithrum (a structure reptiles the the girdle. function Furbinger, all stages the stage the lowest

in

extant was to

species) advance extant in scale

and and,

the at

clavicle, and in the same time, an

these elevate

which animals

the in

that in the stage sternocleidomastoideus evolutionary

species of Lacertilia which the muscle is element is completely in which the

there may be found undivided mass to distinct. These are

the

trapezius-sternocleidomastoideus
THE JOURNAL OF BONE AND

musculaJOINT SURGERY

CONGENITAL

MUSCULAR

TORTICOLLIS

1167 present the in accessory unit, indicates. is regularly vertebrates field rather The of below musculathan from reptile is by the spinal and Howell follow Furtherin may upon be all be the mdithe the

ture reptiles ture two also

is divided. in the has developed (dorsal and the lowest nerves afferent that
of the

Since evolutionary

division a single sources, in the by

of scale,

this it

muscle seems

mass conclusive

is not that

from ventral) animal as these

source, as the in which the visceral the basis which

the dorsal condition the nerves. of their in the so that pathway.

branchiomeric in mammals accessory Apparently, experimental lower vertebrates they follow field

scale On fibers,

innervated

the spinal nerves are concluded

pathway

well as in nature. afferent

in most classes work, Straus presumably a spinal route.

more, The mammals in some separate mastoid vidually the forms

cervical
nerve

some

vagus, have, in reptiles, motor fibers also follow field of is a distinct this portion of origin from and occipital example, portion, life, and

shifted this new

accessory there species slips process

musculature is present in all mammals. Furthermore, portion which represents the sternocleidomastoideus, although may be partially covered by the trapezius. There the sternum bone. These the the and clavicle and various sections portion, the portion. all In separate of the sterno-occipital but a few of insertions muscle may portion, the higher

named-for of animal

sternomastoid cleido-occipital

cleidomastoid

muscles
nerve

innervation is obtained by both the spinal accessory nerve and the nerves. In mammals a larger proportion of the motor fibers follows the spinalcourse, and the source of innervation of the trapezius and sternocleidomastoideus is more frequently double. The shifting of the motor fibers to follow the spinalcourse is imilar s to but more extensive than that found in reptiles. the evidence musculature forms, in the nucleus this main, of the is mass by the vagus it seems visceral. and to when spinal nerve indicate that This muscle it shifted, by the its vagus the mass origin of apparently nerve, but the trapezius-sternooriginally develshifted higher with forms the

In summary, cleidomastoideus
oped it. it part in the In the is supplied,

branchial-arch lower

region

visceral-nerve the nucleus by some to

supply in

is supplied

related

to

the

accessory nerve, and is thought in higher muscle

of which is closely have originally been to supply the of the nerve

of the vagus sternocleidomastoideus

nucleus. Thus, muscle and muscles

OF THE

appears that the trapezius tract.

forms the nerve may be related

supply

of

the

of

the

gastro-intestinal
AND

EMBRYOLOGY

TRAPEZIUS

STERNOCLEIDOMASTOIDEUS

IN

THE

HUMAN

Lewis, on

writing

in

theManual

of Human

Embryology,

gives

the from a

following the

information mesoderm. They later into

the embryology The trapezius in muscle The

of the trapezius and and sternocleidomastoideus the At mass of the seven occipital a very extends

sternocleidomastoideus. are derived region as early period, caudally, the

arise migrate this when occipital of the

the lateral portion of to the shoulder girdle. mass, and as the rudiment is about anterior

common muscle mass and the accessory nerve extends nerve is carried first with it. appears to the two

common the embryo and two branchial-arch its

trapezius millimeters

and sternocleidomastoideus long. It lies anterior position of the is determined myotomes,

caudal location

cervical series than the origin and nerve In this early

myotomes. Its by the location of the supply stage,

more by however. direct No

observations but from branchial-arch

tinguishable

concerning position series. the except

anterior

cells composing it seems that it consists of

this rudiment have been made, it is the caudal member of the closely packed cells and is indisexcept which region for its runs for in which caudalward greater some the toward condistance accesthe about some

from

densation
within
sory

and
it.
nerve

The
leaves

surrounding mass for the presence endf the o muscle vagus. From this

of mesenchymal of the accessory mass lies close point it gradually

cells nerve to the extends

the

arm the
VOL.

bud level
39-A,

of the of the
NO. 5,

pre-muscle tissue, which fourth cervical myotome.

1957

in a nine-millimeter At this stage its

embryo caudal end

is located at already shows

OCTOBER

1 168 signs trapezius In of splitting

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

into

the

two

divisions the about to trapezius from two the the the

from caudal

which divisions

the are

sternocleidomastoideus quite widely while which

and separated.

develop. an eleven-millimeter portion portion the

embryo, extends extends to nearly

The trapezius mastoideus far

sixth cervical nerve, rudimentary clavicle, attached region

the sternocleidoat this stage girdle and

lies but is close

anterior to a thick columnar

first mass

rib. The extending extends of fascia. masses. embryo, and

is not yet occipital

to the caudalward, processes separates girdle

shoulder parallel

to the vagus nerve. It with them by a layer dorsal In caudally. entire of the
spinous extend

only slightly The thick the entire to

toward the deep cervical arm are the as spine far as and now of the

spinous fascia shoulder separate

and is connected it from the more have adjoining dorsally migrated their portion toward the does not milli-

myotomic a The length. clavicle. processes

as yet tothe

muscle trapezius The It

sixteen-millimeter

sternocleidomastoideus

throughout and and of is

trapezius is attached has extended caudally and the occipital the ligamentum cartilage. trapezius be found also the shows

the scapula sixth rib end embryo The

nuchae. The anterior Not until after the obtain in the marked process and its adult final as form.

the muscle over twenty of the ; it is not

meters

in

length may embryo. extends

does

splitting process

trapezius present

into divisions in the early The

embryo

sometimes

a secondary

sternocleidomastoideus and begun The from to split accessory is muscle.

OF THE
AND

development occipital

in a fourteen-millimeter region to the clavicle. and trapezius for It has cleidoand any

mastoid

already mastoid.

into two divisions motor nerve supply secondary and

corresponding from the not

to the sternomastoid cervical region to the a myotomic origin

sternocleidomastoideus portion of the

does

indicate

EMBRYOLOGY

NERVE
TRAPEZIUS

SUPPLY
IN

TO
THE

THE
HUMAN

STERNOCLEIDOMASTOIDEUS

Streeter,

also

writing

in

the Manual

of

Human

Embryology,

presents nerves, are

the

following the adapted forms bundle group man it vagus and to supply the more which is of muscle functions

facts. The accessory vagus complex nerves. In man, is composed the motor are of several branchial fibers of this complex including specially the higher a distinct to the and in

the group of muscles derived caudal rootlets of the vagus known as the spinal accessory cells
to

from the branchial arches. In predominantly motor and form nerve. This bundle is distributed the more caudal branchial arches,

which
innervate

are

derived

from

As these increased creased by down other

oblongata

the muscles muscles grow, importance development down into

of the arm girdle-the the nerve is carried of this musculature of the the accessory roots of nerve the

sternocleidomastoideus down across the neck. in the higher vertebrate ; additional spinal cord. rootlets These origin nucleus cord,
IN

and trapezius. Coincidental with the forms, there is inof origin may are extend obtained as far

extension as the border and

ANATOMY

rootlets of

fourth cervical segment. rootlets of the vagus a portion of the lateral

OF THE

The nucleus of is composed of the region of the spinal

AND

the spinal accessory and ambiguus of the medulla the two being continuous.
THE HUMAN

STERNOCLEIDOMASTOIDEUS

TRAPEZIUS

In tinuous
attachment position downward,

1948, Grant attachment

is of the aponeurotic

stated which

that the trapezius and sternocleidomastoideus extends from the inion to the tip of the and therefore produces a ridge, the superior one by the attached
TRE

have a mastoid nuchal

conprocess. line.

This The of the the

forward,

Below,

one

mastoid process which constitutes and medial pressure exerted head of the sternocleidomastoideus is

end of this line is the result pull of the sternocleidomastoideus. to the superior border of
OF BONE AND JOINT SURGERY

JOURNAL

CONGENFAL

MUSCULAR

TORTICOLL1S

1169

medial
articulation large

third and Chandler lateral to layer of insertion. consistently

of

the

clavicle, by

while fibrous

the tissue

other to

head the

crosses sternum just

in below.

front

of

the

sternoclavicular a by composite found a

is join&l

and Altenberg have written obliquemucle which eftends from the mastoid of the egion extrnal It is usually in dissections superficial Human the clavicle. The and of cervicl t temporal fascia. It

that the sternocleidomastoid the anteriorsuperior portion bone has two more of the skull. of origin heads

muscle is of the pectoral It is and enveloped one

girdle double head most

made up of five or we1e (1) superficial (4) is stated front origins deep that of head

distinct muscle bellies. Those sternomastoid, (2) superficial sternoand or the (5) sternal a double portion deep of

occipital, (3) cleidomastoid. In Morris sternocleidomastoideus notch and that third cervical mastoid
bone. course

cleido-occlital, Anatomy arises lateral Between muscle it

sternomastoid, the medial

the

fromthe or clavipular these

the manubrium arises from there

just upper and

below the clavicular border of the medial area covered by external the outer line of surface of the occipital a nearly parallel

of

the

is a triangular border nuchal bundles

fascia. process The

from

()
are
to

into insertion.

inserts (1) into the lateral half short

the anterior of the superior and the fiber

tendons
origin

comparatively

follow

:
Middleton,
three located major at the

BLOOD

SUPPLY

in
portions.

1930, The
artery

wrote upper end of whichis course. trunks

that portion th&muscle derived

the

sternocleidomastoideus shorter is supplied the occipital than

muscle by

is

divided believed. portion artery

into It of is short appears and join portion branch the the the the is the

mastoid

is much and from

is generally the superior artery. This

sternocleidomastoid

and
to

follows
travel

a transverse through short veins sternal

from muscle muscle.

T1e venous return which penetrate muscle a receives branch

a short

from this portion the deep aspect arterial supply

of the muscle of the muscle the artery. middle This

the
of

large The the

beneath. head
its

of the artery,
parent

its of

through thyroid

sternocleidomastoid and

the

superior

separates hyoideus omohyoideus

trunk

distance

runs backward On reaching

and downward the sternal head it, reappearing downward (there to follow by supply branch of deep the

the above upper border of parallel to the upper of the muscle, it does in along no the junction the the artery of

the omoborder of not enter it and of

head
clavicular sternal

at

once but instead passes beneath head. The artery then courses head to from in veins which the the with branches sternal pterygoid the common are head extended appears formed vein.

interval between posterior border to of the the supply superior clavicular and

are

branches

head). eventually thyroid the and

The

vein

terminates lingual

plexus facial

The clavicular head derives cleidomastoid artery by means

thyrocervical artery. are trunk or from one

its arterial of a small the of branches through the

from the inferior which comes this aspect

portion of either directly which

sternofrom the

The

branch
20

runs exact has supply

upward

trunk, the transverse of the muscle to

scapular branches muscle

extended. The Fitz Simmons receive their blood artery.

According from five or more

path of the written that from the and

sources

venous return the sternal sternocleidomastoid the its ; (2) supply

from this head is uncertain. and middle portions of the branch of the superior thyroid arterial supply of the posterior insertion the sternocleidomastoid portion of the ascend and descend is extensive auricular ; this artery muscle in each branch and

to Chandler main

Altenberg, occipital near artery artery or branches

comes frequently are belly;

( 1) : the

branch aponeurotic either the

supplies or musto which muscle

the
cular

upper
branches

portion with the

NO. 5,

of

the

muscle

anastomoses attached
VOL. 39-A,

the occipital ofthe occipital large

1957

many
OCTOBER

maj

which

1170 (3) portion the sternocleidomastoid and part

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

branch

of

the

superior

thyroid

artery

supplies

the

middle

of the lower third of the sternocleidomastoideus muscle and sends two main branches, aswell as many small branches, down the chief muscle division ; (4) a branch of the transverse scapular artery supplies the lower portion of the muscle by means of many secondary branches ; and (5) an arterial branch which arises from the ascending branch of the transverse cervical artery supplies the lower lateral portion of the muscle. The
are even

vessels
more

which
profuse

provide
than

the

venous

drainage

of

the

sternocleidomastoideus

muscle the return

areas venous
of

of

venous channels,

drainage both

but within is

the are arterial also there the muscle carried by the anterior

vessels. Not only is there overlapping of are numerous communications between the main substance and on the surface. The venous all the jugular, major the veins of transverse the neck-the scapular, the

the

jugular,

sternocleidomastoideus the external

jugular,

internal occipital, are also fre-

the

posterior quent and

between

auricular, the posterior facial, and the anterior facial veins. There profuse communications not only between the main venous channels but their branches both on the surface and within the muscle tissue itself.
NERVE SUPPLY

from foramen ing

In 1915, Fitz Simmons the spinal accessory. and

under

wrote that The external

the sternocleidomastoideus portion of this nerve to the about

receives leaves

its nerve the skull

supply by the

jugular mastoid

descends the muscle

obliquely outward at a distance of supplying arise also the chiefly receives The spinal bellies of the spinal

sternocleidomastoideus two inches from

muscle, disappearthe apex of the

process. The The The branches motor branches sternocleidomastoideus sternocleidomastoideus from the visceral motor and are both visceral and somatic. fibers of the spinal accessory sensory fibers a deep portion from course of the in the from of the vagus which second, third, and the nerve.

and fourth it usually middle artery.

Arey

cervical nerves. lies between the third, has

regarded

accessory follows the cleido-occipital course of the nerve and muscular

this region, muscle and the

following written innervates

as of

closely that the the

branchial-arch

branches a part

occipital complex. are

accessory
origin. FUNCTION

is really trapezius

This
usually

complex

sternocleidomastoideus

muscles,

According is to

to

Morris

Human neck

Anatomy, toward the

the

function and

of to

the rotate

sternocleidomastoideus them toward the opposite When same the

bend

the

head

and

shoulder

side. When the trapezius also acts, the head is fixed, the two muscles may further in the simplest terms, the sternocleidomastoideus side and the chin to the opposite side.

neck is flexed and the chin is raised. increase the degree of hyperextension. ctually, A rotates the occipital bone to the

ETIOLOGY In remote, regard a discussion cause to the ofthis of the etiology condition must

of congenital be considered probably matters

muscular as well little

as

torticollis, the the immediate it is contraction fibrosis of the in the length

ultimate, cause. considered

or With to be

immediate

cause,

it

whether

fibrosis to fibrotic mastoideus

during

( 1)

within the sternocleidomastoideus scar contractions elsewhere with relative shortening

muscle, with in the body, or due to a rapid

subsequent (2) static increase

similar sternocleidoof the

neck

the the remote which the fibrous-tissue

stages of accelerated growth, cause which has stimulated least is known. If it is postulated replacement
inquire as

or (3) a combination the most interest that the presence is the tumor. immediate fibrous

of both mechanisms. through the years of a fibrous tumor cause of

It is and about resulting it is

in

of origin

the of

muscle the

torticollis,

tempting

to

the to

CONGENITAL

MUSCULAR

TORTICOLLIS

1171 more breech difficult. position, It might it

Determination

be

argued that On
tend wry-neck

that breech closer to be

to

of the because

predisposing some babies

factors with

is apt torticollis

to

be even are in born the

follows wry-neck. for records

presentation attention to a causal factor latter as to

can be considered a predisposing the problem, however, we find that in the breech presentation. Recent interpretation. the cause It of congenital muscular of interest attributed at the

factor in congenital it would be possible studies of clinical have in 1670 condition the the was giving antenatal theory revived. rise of to birth Strovan to evolved Roonab-

A
in

number past
in

confirm the of theories

torticollis that this outset,

the

two
one

hysen, normal

and ofthe
on

one-half centuries. first recorded works

or position

is a matter on torticollis, utero. 1838, before is Thus,

pressure

theof

head

in

existence
injury, meyer

of and believed

torticollis it was to that

was postulated. In be nearly a century the sternocleidomastoideus he

Stromeyer formulated the antenatal theory ruptured during labor, the

the

sternomastoid
following

develops

deformity.
of large

From hematoma part of the

tumor, which the organization a pathogenetic is followed substance of the

contracture congenital

felt is in reality a hematoma; of this hematoma is responsible standpoint, Stromeyer thought which replacing may it

contracture which for the wry-neck that the development a of

the

by the

myositis muscle, deformity.

involve and ultimately destroy with fibrous tissue ; contraction experimentally by the tearing of tumors of the sternocleidomascontributed to the to disproving Stromeyers of true adhesions a position falls

this
muscles toideus

muscle In 1883,
leave seen

causes Witzel
no in

wry-neck wrote that and

hematomata in no way

produced resemble the

findings which

muscular

torticollis,

the

hematoma Petersen,
theory between

theory. in 1884,
which
was

hematoma

1886, and 1892, widely accepted.

reported Petersen

his

attempts stated that He these of the

combat formation

hematomata
form of in of of

is not followed the amnion and which

when

by the development the face of the embryo with the development face adheres to the this side the situation, of the opposite the changes
of sections

of torticollis. and that of one amnion,

believed that bring about

the the

head
for

interferes
oneside

muscles, cleidomastoideus insertion development

of the In on the on

the sternocleidomastoideus body of the embryo but

opposite
ofthe on

direction.
muscle

he postulated, adhesion is not side are brought theory the

of

the development interfered with closer of are congenital the result

from

of the sternothe points a lack torticollis. inflammatory

patient

muscle

together, muscular of
a

causing

that
in 1885,

side. initiated that the finding results in 1890, the the

studies

Volkmann, This
process,

infectious in of some the fibrosis returned the

author
for

believed
microscopic

muscle
a

an

sternocleidomastoideus

with He and

wry-neck believed that A few that torticollis years some to

revealed this later, variations. explain

presence indicated from the Schmidt

of

the

characteristics of an develops the of old infection subsequent intra-uterine,

of

infectious in intra-uterine to antenatal liver upon presented the

myositis. life myositis. theory, the by uterus breech.

occurrence which to pressure of wry-neck

but added as sufficient

Conditions

He regarded development

the mothers in an infant

sentation, fluid. That that life,

truly foundation

he regarded as favoring the development of pelvic deformity, primiparity, and the presence same year, in 1890, Golding-Bird originated cause of which
writing is laid

wry-neck included breech preof a small amount of amniotic the neurogenic theory. He felt lesion during of type that intra-uterine the the three is arrested. two types and the considered

the
Anderson,

the

deformity development in 893, 1 an

is the of believed

occurrence

of a cerebral

following
congenital

the sternocleidomastoideus that there are really intra-uterine parturition. arrest the of the side of life, He

torticollis, for which there are

type, which

by of the

arises accident

during during

possible causes (2) disease of

VOL. 3-A, NO. 6,

the former type: (1) nerve centers affecting

1957

development the neck

of the secondarily

face and neck; ; and (3) mal-

OCTOBER

1172
position of the

R.

T.

L1DGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

head of

of the muscle

the

foetus in per

utero.

The

second he did been ischaemia

type Although bring thought as

of out

torticollis Anderson

he

felt added

might little

be to

due

to

injury

se during

parturition.

the conception of the sternocleidomastoideus, probably not syphilitic Mikulicz, actually involved sternocleidomastoideus duces
passage

causes of torticollis, which formerly had in origin. in 1895, suggested general He reasoned while muscle, and thus
relaxed

that the so-called by some to be due cause of anoxia) of the torticollis.

tumor of the to lues, is This theory to the in-

writing two

factors, muscle.

ischaemia that

(actually compression

and trauma umbilical cord

forced
rupturing infection muscle,

forced respiratory movements ; the sternocleidomastoideus inspiration, is contracted

more is a easily cause of than torticollis. a

the fetal head is still engaged in the maternal being one of the muscles involved in the is more vulnerable to rupture-a rigid muscle muscle. Mikulicz also accepted the theory that 1897, thought origin and traumatic today theory, done that if it and infection occurs infectious in of the a muscle theories. expressed theory, theories. the it patholis imposthe muscle

especially has Most to been

if

the

infection is a factor

Kader, is ; he the

of

writing in hematogenic combined of

which prior (4) During ogy sible shorten might Because

muscular

inj ured,

thus

the

of the theories on 1896 : (1) intra-uterine theory, time, however, using and (5) this

cause theory, experimental as

torticollis accepted (2) birth-trauma and work (6) had been

were first (3) infectious of the above concerning that unless

neurogenic of In to

ischaemia little dogs

theory,

combinations

the condition. 1898, Heller, cause by when be myositis

experimental of a muscle addition, are favor of the presence of muscle possible a year

animals, by means he showed permanently

demonstrated of trauma dogs that in brought theory to the

contracture

is infected

pyogenic organisms. In their points of attachment as in and evidence the relation as a in the area in

muscles ultimately nearer together. This malposition. in congenital that the muscle that an muscle the same

considered of his belief torticollis of the as

of the old hematoma of his of actual by means cause earlier. of

of intra-uterine myositis found which showed

consequence

experiments

contraction infection through conclusions

occurred only the traumatized blood stream Kader had and facts of or the

infection, Heller assumed of organisms reaching Thus, he reached

was a reported

wry-neck.

In 1899, Bradford but added no new which hematomata results did those
in torticollis

Lovett summarized interpretations. They sternocleidomastoideus is interpreted pathological

.

the main etiological described experiments were produced and to mean made at Three probably in to mechanics either of characteristics had ruptured trauma. were that the

theories upon stated wry-neck lesions and studies had did

then rabbits that

known in the

not
whom

were negative ( negative develop or that the seen the in congenital muscular sternocleidomastoideus

deformities not resemble of infants found that 1902, V#{246}lcker He

torticollis) They had been this

follow-up birth

reported described, pregnancy. obstruction clear, writing an adult microscopic the

did not develop as that the so-called for the first The reason of however. that the In they blood time, for supply

a result of hematomata the the presence ischaemia, to the

years later, in postobstructive oedema.

of torticollis according muscle and of ; the

infants following extra-uterine V#{246}lcker, was prolonged partial of the cells the obstruction were not by in no that infarct. occurred describe the of four club-foot,
SURGERY

1903, Gallavardin had observed atrophy The the following belief that the excised also

Savy reactivated the anterior horn (1904), Kempf myositis was is that a case that with side of in he

neurogenic theory in the cervical cord that ; he hemorrhagic torticollis first to there stated was

with torticollis. evidence for appearance writing extra-uterine congenital a typical

year infectious muscle It in

showed responsible which is the

microscopic

of

a healed

Joachimsthal, following an presence

one-half

in 1905, pregnancy.

described is believed

of other years had

deformities case of wry-neck

association on the right

THE

torticollis. as well
OF BONE

A child as bilateral
AND JOINT

and

JOURNAL

CONGENITAL

MUSCULAR

TORTICOLLIS

1173 Joachimsthal muscular was also the was first

deformed to report In 1906,

toes and hereditary a new

fingers,
theory

and

a deformity
of torticollis.

of etiology

the of

head. congenital

transmission

concerning

the

torticoffis

developed-the theory of arterial occlusion, as conceived These men based their theory chiefly on the work infants. They reported that their anatomical studies blood
pendent cate

by Nov#{233}-Josserand and Viannay. which they had done with stillborn had demonstrated three systems that each system does not arteries. the venous is an indeconare and which surrounding reasoned that communiFrom the systems

of

supply with either

of the

to

the of

sternocleidomastoideus. or

They
ofthe

stated muscle the they

unit which

supplies its own portion the other systems supply then can to the They heads proceeded be occluded

with

figuration

arterial

muscle,

also
sternal such serand of the

independent.
and as occurs clavicular during

to show that in the foetus the arteries of if the head is in a position of extreme rotation head is in of supply this position, where they interference torticollis by Morse stated the have that was in the pass tensed beneath with arterial 1915. the the

the

mastoideus

may

labor, compress believed

for when the the vessels their may

sternocleidoit. Nov#{233}-Josblood occlusion. presented was supply The the not muscles

and Viannay sternocleidomastoideus Schloessmann, recorded

of an

work showed that occur during labor. the was cause of described and

first
case caused

case

in 191 1, stated that of bilateral torticollis delivered injury. extended wrote a result of the platysma being by He caesarian thought position

He

infant

section

condition shortened

by the
Bevan,

was

a birth partially
in 1918,

that the cause for the fetal head may

bilaterally occupiedutero. in by an in and for injury around

that
during

cleidomastoideus

muscle

congenital birth, followed of this trauma, limited is not

wry-neck is caused by hemorrhage histological to the muscle itself,

of the sternothe muscle in the structure cervical fascia of

and the
which

its muscle.

sheath. The
forms

As

changes

occur the

contracture

deep

marked is caused
of the 1920,

the sheath change in the by the head

muscle is also extensively involved. In addition muscle. This is not due to fibrous degeneration held on one side, which causes marked reduction no alteration theory. to in or the in its histological at or ischemia of the muscles

there is a but rather in the size structure. presuperior affected. in torticollis. by abnormal evidence deformity. for although cases wrythe In

platysma, although Sippel re-emphasized

Meyerding, in 1921
,

as a rule it causes the intra-uterine thought that which pressure trauma results upon into

sternocleidomastoideus myositis. The branch of the

ceding
may,

birth causes ischaemia he wrote, result from artery in 1923, or from a

chronic interstitial the sternomastoid around the sheath

thyroid Schubert, In intra-uterine

fact

hematoma

the same year, wrote that Jones and Lovett supported position or hematomata are not by increased due must followed to by that club-foot,

heredity may the theory intra-uterine the also the a to be origin, same be cause, recognized development

play an important part that torticollis is caused pressure. is as of often a They an possible used associated cause, in muscle theory anomaly is the other as

that added some does

congenital that they occur. Krogius, torticollis itself. He process

continues for

They in
neck

cases

wry-neck,

ruptured. muscular
blastoma

They emphasized in 1924, is thought has an endogenous believed which

years

previously shortened the first to suggest the being the result of an of this condition

may easily be that congenital in the muscle an anatomicalthis process theory. torticollis uterus. In this expense He felt degeneration. active

that

the

development

pathological
of muscle-tissue process

formation,

consists in connective-tissue or tendon formation at the tissue originating from the perimysium. and that it is not a sequel to ischaemic muscle believe birth. year, case that gave his to support of the the development reasons the foetus for theory and to of torticollis is

He
lasting

was Stern, reported to

thus
many

the
years

first

to

an

following

in an

He

that same an unusual abnormal

supporting the that congenital a lack of space

intra-uterine muscular in the

is due
voL.
39-A.

position
1957

NO.

5.

OCTOBER

1 174

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

case
Bauman

the the
based

infant
stated

was that

in

the

breech

position had

and

was

delivered

by

caesarian been majority

section. followed of infants

In

1925,

by with
was

development congenital
upon a some

in no case of torticollis. the

of study carried more

injury to the Fitz Simmons stated 21

than 500

sternocleidomastoideus that in the

torticollis had been

condition the

is not cases.

due to Some during

obstetrical trauma. of the infants with pregnancy following majority

in same

This observation torticoffis in had received delivery the flatly origin that of Petersen, the developColonna, of or the

this
no injury condition

series
trauma was

outside

uterus

and

some strenuous

; in

the in

not followed

Rugh,
is

writing
traumatic

development of by the development 1925, felt strongly

rather than congenital.

hematomata of torticollis. that in the

Sever,the

of year, following

cases stated the He of of

he
a

had
hematoma.

never

seen Aberle, Spitzy,

a case in and

in 1927, others

which

torticollis reported

had the

developed heredity that, in

development

Friedberg, ment in the that study

vanced of

emphasized who had been that

factor. a number

quoted cases,

of hematomata same year,

had stated his

not belief

followed abnormal

by the development position in utero is a

cause.

torticollis. Also during

year, Hellstadius stressed the of the case of a child delivered by Petersen, who maintained condition and the intra-uterine by the flexion pressure exerted with rotation and of in basis of the
conditions

heredity theory. Rossi, in 1928, by caesarian section. He referred that there is a connection between life of walls. neck. the The This foetus, pressure position, describing the forces the in turn, of

made a complete to the theory adthe pathogenesis mechanism foetus causes as an into a faulty an abnormal and posi-

the

abnormal position shortening resulting

on

uterine of the on

the distance between torticollis. Petersen, of his clinical is related observations, to foetus

the points the basis had

of insertion of numerous concluded that to to

the sternocleidomastoideus, experiments on animals the faulty intra-uterine to various of thromecchymosis to two of his since, closed veins with the

the

tion
other

amniotic anomalies. he was He

adhesions, unable

oligohydramnios, and the

Middleton, bosed
tumor

which also produce in 1930, stated that in the sternocleidomastoideus. not have the characteristics

demonstrate that

presence

veins does

pointed out of a hematoma,

the sternocleidomastoideus for there is never any cartilaginous. of one Excision 1931, wrote presented that be

or

fluctuation and from the onset more, the process is diffuse instead
before

the mass of localized is not

is firm, hard, and feels and there is an interval with hemorrhage. Bargellini, in in 1933, sternocleidomastoid it would seem that supply being the same expressed and the Abels, cause. theory. the origin by by scar of in the

Furtherweeks a tumor reasons under

the
supporting

tumor proved the

appears, it was heredity neck at not

which

consistent

definitely
for to may

due to theory.

hemorrhage. Fitz Simmons,

experimental
injection

condition,
when the

be

shut

off

during

the middle and inferior is in a certain position, labor without the arterial there is Abels, year, which the foetus the rupture the observers The produced in 1934, malposition held is due heredity

arteries can the thin-walled interrupted, as is that and head. article

result experiin the trauma. in 1936, Schmid

that,
mentally

temporarily by venous occur as

least, ligation.

condition his belief are aggravated view, same clavicular in an

obtained changes by birth Jahss In 1937, written

muscle

a result

of intra-uterine

Dc Gaetano, in described a type

repeated

the following of torticollis that

in 1939,

essentially to a shortened factor.

the that
Kastendieck,

theories the position that

during

stress of
supported

in

1938,

stated
in

1940,

stated
fibers

some birth.

is a direct intra-uterine believe that is accompanied muscle development his belief of

Shands and of torticollis formation of

Raney, writing is rupture of a hematoma and

the of

muscle

subsequent the hematoma mastoideus. Christian, neck the

replacement of a part of the may be followed by the .Jan#{233}k,he next t year, affirmed in Oslers Principles sternocleidomastoideus
and

tissue ; incomplete regression contracture of the sternoeleidotraumatic factor-birthinjury. that and

OF BONE

is

Practice shortened,

Medicine, stated hard, and firm

THE JOURNAL

in congenital is atrophied
AND JOINT

to

wrya con-

SURGERY

CONGENITAL

MUSCULAR

TORTICOLLIS

1175 from local at that but In their thickening

siderable cleidomastoideus
produce

degree.
an

This due

condition to rupture, Chandler

must a and of act

be

distinguished which in or may 1944,

sterno- the of
which may torticollis number of conmal-

induration.

condition Altenberg, pathological

occur stated

birth and muscular from a

results, tributory

not

from factors

a which

single may

type

disturbance, together.

separately of

opinion

intra-uterine

position and the local ischaemia contribute to the development maldeveloped, fibrous, shortened, may also Johnson, be a factor. the next year,

and pressure which may be caused by muscular torticollis by rendering the and ischaemic. They felt that trauma that the muscle may become

this malposition muscle atrophic, during delivery

or

wrote

contracted utero in

may undergo fibrosis and shortening as a result He added that in many cases a hematoma can week after birth. Chandler, in 1948, stated that

of stretching be palpated it is recognized

and tearing during in the muscle during that contracture same year, was on the head than into tumor tumor two formation. and 1950, caesarian
to the

delivery. the first of the

sternocleidomastoideus may exist prior to birth. Stevens, in the congenital muscular torticollis in identical twins. The condition both infants and there was greater involvement of the clavicular head. He considered the condition to be a congenital anomaly. In 1950, Hulbert reviewed 100 cases. He classified the patients
those

described right side in of the sternal groups, and He felt

with with

postural muscular

torticollis torticollis

in in

which which did the

there there not cause

was was support of the the

no

associated

formation

those that other

observed

associated

the findings prenatal

a case

of histological studies factor was probably

of congenital

the ischaemic theory condition. Also in

in an

that some Greenstein

muscular

torticollis

delivered infant labor, weeks two

by
prior

section.
expected

The date

operation of delivery.

was

performed

before

onset

of

PATHOLOGY

The
in

earliest latter white

as

known part male

induration

description the upon nineteenth whom

of the

pathology, Taylor, been

as in done.

seen

microscopically, a he

was

written a sixthe

the week-old
condition

of

century. autopsy had

1875, presented In this report

case of described

tumor. histological had

and

He

stated character
to

that,

the of to of the the

a

st#{233}rnocleidomastoideus muscle his knowledge, his case was lesion had been ascertained. bundles in tumor of muscle fibers, some parts the showed dense,

or sternocleidomastoideus the first on record It was found that was and destroying double its connective

in which the fibrous tissue these fibers thicktissue or, in

developed
extending

between
such

degree that

displacing muscle white

usual

ness. many dense be

on

Study

of

a section fibrous fibrous

of

the tissue. tissue

fibrous

places, white and nodular, upper Some

In the middle portion alone was present and tissue normal and quantity Deeper of fibrous in

of the muscle, where most it was no striated muscular could fibers was in were of the tissue only moderately appearance and less fibers distinctly and elastic of abundant size, while striated tissues, than but

seen. At the the surface. were All were muscle were

end of the muscle, the muscular of the fibers of this tissue were twisted, and by of varying a considerable growth. amount breadth

others normal. there the served fibrous

narrow, no features there was

surrounded

of early cell an excessive

substance in which

the upper end of tolerably well prenew growth of

muscle fibers lay isolated in the midst of the waxy bands. The tissue was neither completely localized nor uniformly diffuse. One of the most interesting secondary changes in congenital muscular asymmetry ofthe skull and face-was carefully described by Witzel in 1883. that that the
and
VOL.

torticollisHe reported reveal is deflected, been offered,

it onis

the the normal the

39-A,

affected distance side. cheek

NO. 5.

side of the from the He is less

OCTOBER

head external reported No

and

face canthus that

becomes the toangle the eyebrow explanation

atrophic
of the

and
mouth

that
is less

measurements
than

also full.
1957

is less of this

arched, condition

the

nose has

satisfactory

1176

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

but ishes the New had by

have

Witzel thought it is probably due to imperfect blood if the torticollis is treated early. In 1891, in a discussion of a paper by Whitman, Hadra open operation which he stated he first described had in York shown cicatricial
occurred

supply. claimed a paper cases that its extensive of

The

asymmetry for

dimindeveloping in the examination taken could

priority published microscopic place

Medical that the tissue.

unless

Record

in

1884.

He

said

that

in that which

two and such

muscle had entirely He found it hard the condition reported but caused Spencer
in

disappeared to believe

had been degeneration sternocleidomasHe claimed

In toideus that
been

1891, muscle

Whitman was present is not H.

at

was of intra-uterine seven cases in no that

of

origin. swelling

the muscle.

in which by reported
fifteen

torticollis of the
nearly 300

subsequently

developed.

torticollis In 1892,
observed

a hematoma

sternocleidomastoideus

R.

autopsy

hemorrhage children had and

in the sternocleidomastoideus who either had been been in

had stillborn posi-

or
tion

had
; in

died two

shortly cases

after forceps to

birth. had

Ten been

of the infants used in delivery

in the breech or footling two no instruments been had cases muscle. and

used. into
inch

According the anterior hemorrhage the thick at separating

in large

Spencer, portion muscle was the edge. the

amounts

the microscopic of the upper dark red, had

sections in nine two-thirds of the a firm consistency showed fibers.

revealed hemorrhage As a result of the was one-third of an tissue, view infiltrated showed with blood large sublatter

blood,
effusion

The vessels more

low-power view and muscular

fasciculiand

connective The high-power

between

Some bulbous stance often these that by

fibers

appeared

granular in fibers and

than some showed

also, others

in places, and many still

between individual fibers. were ruptured. Some had muscle the

ends. The sarcolemma had given way. The being indistinct, areas the fine the distribution in wavy, transverse of the

cases was longitudinal

intact, although the and transverse striations, swollen Spencer similar

coarse. striations extravasated

In many fibers there were were usually more uniform. blood in these infants was children. of the infant

areas and in concluded to that seen and cut Chirurinto the it been

other authors In a discussion gical Society in swelling same had

converted

the fibrous of torticollis 1893, Parker sternocleidomastoideus W. G. Spencer that

cartilaginous

tissue of older at a meeting said that in an and stated and

one-half

British Royal Medical five weeks old he had resembling performed of the muscle blood. by sheath

of the discussion been revealed found

into

had that

found liothing in an operation layers fibers

In this Volkmann had masses In concludblood or even tissue totally had clot

the

anterior
masses

posterior
inch thick.

had

that

they

contained that

some

muscle

Microscopic study of these and ordinary scar tissue. trace that that normal of pigment had been or altered

ing the discussion within the muscles Mikulicz, in excised in parts

Power said of new-born 1895, studied

he had found no infants with torticollis. specimens of muscles and appeared said

partially

from twenty-one of muscle which

infants with wry-neck microscopically

changes were present and that connective

formed in place of the muscle fibers. Hildebrand, in 1897, reported that study of microscopic sections had indicated that marked decrease in the muscle fibers in the region of the tumor had occurred in an eight-week-old child. Many fibers still remained functional, however, and no blood pigment had been demonstrated. The diagnosis was interstitial myositis. In
oped had with with been

1903,
after

VSlcker
prolonged

described
partial microscopically

a peculiar
obstruction

degeneration the of circulation

of of muscles hematomata the

centimeters

muscle the

fibers muscle. excised reported

which These

had changes

devel-

observed

congenital

torticollis.

in sternocleidomastoideus V#{246}lcker believed that the 1905,

muscle

from patients were probably

Postpartum showed

oedema. a swelling the

In size

Heusinger
which

reported
was nine

case

of two
OF BONE

a sternocleidomastoideus

long. Pathological

fourteen-day-old infant study after

JOINT

of a hazelnut

which

had
THE

developed
JOURNAL

days
AND

birth.
SURGERY

Micro-

CONGENITAL

MUSCULAR

TORTICOLLIS

1177 no trace of blood pigment. life the head, and lateral Henthat a

scopic

study

revealed

chronic condition not have

wrote

interstitial must existed.

as a

myositis have originated the abnormal

with

singer felt that this true hematoma could In 191 1 Jacobs ,

curvature away from

during position

intra-uterine of

that

result

of

ofthe cervical spine the affected side. in the lower thoracic region. by several authors: V#{246}lckerhad had noted that the diseased

is always present, the convexity of the curve being directed Occasionally, there is also a secondary compensatory curve In 1915, Morse summarized the pathological findings reported demonstrated degenerative changes in the muscle, Koester muscle had been almost entirely replaced by fibrous tissue, the changes Kader same
contracted

Volkmann changes
enous study Zenker ing of

had as
spread excised

interpreted myositis,
infection. portions of

as had year,

inflammatory, Mikulicz stated Fitz

muscles had

had

designated are due to pathological

the hematog-

fibrous

In

and the
of

that Simmons

the changes 20 stated that

which had of the muscles the body

to of cause

been described with subsequent theory of

myositis

as

a sclerotic shortening. to myositis

contracture in

shown interstitial He also : (1)

except

the waxy degeneration myositis culminating gave the following rupture of muscle ; (2) Heller by injections

of in hardenreasons for in other was parts im-

considering of the
possible pyogenic dence toideus anterior,

Stromeyer by
and

be

implausible and
animals

is never
organisms;

followed (3)
or

contracture

showed it of actual

most by

of

hematoma is not followed trapezius,

injury; and

the (4)

torticollis.

infants in most In this

with torticollis seen after birth show no eviinstances hematoma of the sternocleidomassame article he pointed out that the scalenus involved this in condition contracture represents conditions a type of

and splenius capitis are frequently affecting the sternocleidomastoideus. Three years later, in 1918, Bevan stated that
fibrous degeneration

in

resulting in contracture. sternocleidomastoideus

scaleni,

which In muscle

the 1921, but

muscle cells are Albee stated that that in long-standing shortened. that in torticollis

replaced by connective-tissue cells, this condition is usually limited severe cases, the platysma, the sternocleidomastoideus tissue may the occur muscular findings in contains in would 1925, patches tissue. seem wrote

to the the

In
abnormal throughout

and the spelini 1923, Jones and

amounts of

may also be Lovett said

fibrous

tissue.This

scarlike

fibrous

is also to

the adaptive the

muscle or shortening presence

in severe cases it may of the other soft parts of fibrous myositis and

largely replace of the neck. These perimyositis.

There that

indicate

Clark,

as a result of the pathological studies made and others, the character of the muscle change is a substitution of fibrous-connective tissue for
replacement fibers is almost complete, resulting

by Bouvier, Krogius, Volkmann, had been determined. stated He that there the muscles and that sometimes of of four the insertion. muscle, tissue.
more

Mikulicz, the

the in

formation three below

a fibrous cases of the injury;

band.

The

muscle

In muscle
however, rhage there

which 1929, was the

or was

are left lose von Lackum apparently scar ran

their cross reported normal above

striations. that in and

sternocleidomastoideus in one of these cases, fourth case the hemorin the outer aspect muscle mass or side. of the The tissue fibers

site

injury

into both divisions extended the entire length

of the of the

In the although In
extensive

was
tumor

still distributed
as the

what appeared through the muscle proper that

of the

to be normal muscle mass scar, of becoming at either end severe degeneration, and more extensive may solve that dense the other the problem

all

four cases, thein muscle at one

was

approached

or,

in

one the

case,

in

the

Lackum several prevention fibers replaced removed

neck added
months in the

showed a closer

apparently study of of sections The dense

as a result muscle in and make

pressure. Von early period, possible the muscle apparently the fibers mass and

after birth,

etiology showed muscle scar of

lesion. midst of He

He stated relatively tissue. noted in

microscopic scar tissue. tissue and

degenerating fibers were composed muscle

by
at

connective
operation.

Connective muscle

tissue some

homogenous fibers,

appearance indicating

the

presence

of

vacuoles

degeneration.

1178

R.

T.

LIDGE,

R.

C.

BECHTOL,

AND

C.

N.

LAMBERT

In
seen in

1930,
later

Middleton, childhood that it The has would

in

a survey

of clinical rational

ninety

cases,

wrote

that

the

torticollis are present as thatin attributable at

condition seen to to a

infancy
common two of weeks

and

the same appear

and pathological to expect that of infancy

appearance all cases is usually

cause.

sternocleidomastoideus

tumor

one

the

frequently

after birth ; it first appears as a spindle-shaped sternocleidomastoideus muscle, occasionally involving involving both sternal and clavicular heads.

swelling occupying the position only the sternal head but The uppermost portion of the affected. is present six months torticollis one begins side as The for two after while to the enlarged or three birth. the muscle months It tumor is not is is

muscle hard and at

tumor uncommon

close and then feels is

for

to

the

mastoid

process when

is touched.

seldom, The of

if

ever,

cartilaginous gradually a hild c development.

in size, the

tumor four to temporary

to

absorbed, disappearing show a mild degree In some head to original about the tumor collections somewhat in result
fibrous

its

fullest
is

diminishing

cases being be

a true drawn

torticollis over to

develop tumor

while the is abresembles tissue no nuclei reveals is no degensmaller and show it the

sorbed. Grossly,
a soft
in

the are

tumor
Microscopic

appears
study

fibroma.

shows that

composed of glistening it consists of young muscle fibers.

Microscopic

fibrous tissue and and cellular fibrous of

study

which
vacuolation

remnants
and are

of in
or

the
undergoing

Many with
there

or

degeneration.

mastoideus that
erating

tumor its tissue, terminal with and stigmata the not

not begin muscular

a child stage
young

eight has
tissue;

years the of size of

instead

old following muscle and the

fully
are

these fibers have of the sternocleidodeveloped torticollis There

of non-cellular

in

characteristics.
swathes adult,

fibrous than
none

scattered varying

fibers. outline, absorption are

These living of This this the

fibers, and tumor

although healthy in the

normal
of the

Although usually
infant does

of degeneration. torticollis is the become

to

infant, of

does

does
abnormal lags

apparent assume the

for three proportions on

or four years. of a child until

is because the neck time. When growth has been normal partly muscle.

commence,

by

the tissue behind

muscle will in

substance not elongate growth and

the affected to the same relatively

side which extent as shortened,

replaced Thus, the classic

the

muscle

becomes

producing

deformity. Middleton described a number of pathological changes which skeleton in association with congenital torticollis. One such change may develop in the clavicle when there is fibrosis in the clavicular Exostosis is never seen at the sternal attachment because in this region fibers
vicular

may appear in the is that an exostosis head of the muscle. the fibrosed muscle insertion. bone. which In the clathe Apparently are inflicted

are head, new-bone the Pathological marked

muscle

separated however, formation changes asymmetry

tends to

from the

the

bone

by comes by the bone

the

normal

fibrous

tissue with traumata

of

the
upon

scar tissue is caused of the occur of the

into direct contact minute recurring through the shortened

subperiosteal

layers

muscle. face and skull. There is on the side of the shortthe level of the eye side than eminence other side, the the the is

well
ened

slope

especially in the skeleton of the bone in the skull, and the eyebrow downward. The portion of the face below On the the on

appears to corresponding
flattened and

be

shorter portion
there

from above downward of the normal side.

is well a

and to be wider from side to the affected side the frontal occipital region, while on the

marked

bulge and back

in

the vault

eminence of the as side. long

is unduly skull has

prominent been thrown

occipital region the affected side

is rather flat. In short, and pushed forward on severity of to diminish early, all was
AND JOINT

opposite curve
after

surgical

The deformity of the skull varies according to the as growth continues ; thus the facial asymmetry tends correction the of deformity. If treatment is undertaken may disappear. reported several cases. In
THE

the

cervical gradually traces of upon

asymmetry In

1 93 1 Holloway ,

one

case
JOURNAL

the
OF

patient
BONE

operated
SURGERY

CONGENITAL

MUSCULAR

TORT1COLL1S

1179 literature. by fibrous patient was found in weeks definitely degenin this in 1842. old,

twelve

days

after birth,

In this case the tissue and study

operated upon

which is the earliest time of operation recorded in the muscle fibers of the tumor were found to have been replaced of sections showed that there were no blood cells. Another five weeks afterbirth; in this patient thrombus formation was changes. marked quoted composed
Hough

addition showed In
proved erative country

to even 1934, that

muscle were

fibrous more Hough it is

fibers. made by

Still fibrosis. Middleton of Warren

another as
wrote that

patient, saying the stated entire two that fibrous

operated excision tissue

upon of the containing

when tumor

seven has scattered

young
also

cellular of Boston

John

first reports in 1841 and that muscle to six in

of cases of John Brown

torticollis of Boston

Chandler had showed heads. The

one and

and Altenberg, in 1944, a fusiform swelling of the swollen muscle was from
centimeters

their series gross pathological study including both sternal and clavicular centimeters in length and from one at times, cartilaginous. child, after disappearance fibrous Instead replaced of tendinous it showed by dense and all there further more so contains
and five

to

one-half

On
of

section, the tumor,

the

tissue the

appeared was study

width in and felt firm, white and glistening. found no muscle many calcification and fibroblasts. and the tissue hyaline resembles
findings

fibrous, and, In an older by a dense hematoma. had been stages of

muscle

to

have been evidence cells were nuclei),

replaced

band. complete tissue.

swelling, staining masses

Microscopic disappearance The few

vacuolation characteristics. of young

showed of the fibers with some,

tissue

of which in

almost fibrous showing in

muscle
(some

remaining

varying absence

degeneration, changes

striations, and in Altenberg

In
fibrous

was present, Chandler and tumor becomes connective tendon.

in infants

were great reported more finally of the all

that
muscle

as

the
cells

patient
vanish rows

grows and the

older fibrous

begins dense

to

disappear, and acellular which

three,

and that nuclei

months

that
in

remains
parallel Reye, congenital

is dense, and which

reported muscular

inelastic thus
autopsy

tissue
one,

arranged
of age

in 1951,

with
sections.

torticollis.

He

was

the

first

to

relate

studies muscle masses arrangement

of with

longitudinal hyaline of the fibrous between

These that fibrous

In a have

degeneration.

studies revealed They also revealed the and in cells that fibrous the
concerning

the

presence that there and attachment.

of

of multinucleated was a regular that there was no

tissue this
cells

replaced tissue
textbook been seen

affected the tendinous

muscle
diseases

line

of demarcation

muscle, dams A

described
representing

these
early

the muscles as masses

at

of of

patients sarcolemmic
of

attempts

regeneration

and associates stated that giant congenital muscular torticollis. They nuclei and interpreted their presence muscle fibers. In 1955, Kiesewetter and with cases they of atrophy cent of the duration. had cases reviewed, being present but Evidence was not of the muscle bundles in the surviving conspicuous, degeneration of dogs. completely various changes

as

associates
were muscle replaced

reported by that

in 85 tissue, lesions

per cent varying

was

of the degrees of long

fibers. of

Dense striated

collagen muscle

found 20 in per

suggesting
atrophy

were
was

not

and

seen all in cases.

kinds
Among

Some has
his

experimental been mentioned

in muscle findings as wasthe

work
a

regarding previously.
of

changes Brooks
disturbances in

occurring

result

in muscle following 1922, studied the circulation. He did his in leading from a muscle the

trauma pathological work on was

fact

that

when

the

vein

occluded

resulted.
acute in the

but These
inflammation,

the

artery changes
and

was left intact, pathological changes in included hemorrhage, oedema, degeneration fibrosis ofthe muscle. The surrounding tissues so that that
and
1957

muscle consistently of muscle fibers, were also involved in a fibrous of an tissue

fibrosis Brooks also

in
NO. 5,

process, found
gangrene
OCTOBER

eventually permanent
necrosis but

the obstruction will not

muscle of cause

became the fibrosis

embedded arterial and supply

mass. extremity of the

may
VOL.

result
39-A,

substitution

1 180
muscles with of arterial fibrous or in 1926,

R.

T.

LIDGE,

R.

C.

BECHTOL,AND:C.

N.

LAMBERT

tissue. Simple nerve in supply did an experimental

ence
ing
animals

hermorrhage not cause study

the

into the muscle fibrosis or changes produced fibrotic work a

with or which changes little

without interferwere seen followin later the and muscles arrived of at

venous
.Jepson,

obstruction. by venous the same occlusion. conclusions Middleton as Brooks. did similar

essentially

REFERENCES 1.

2. 3. 4.
5. 6.

7. 8.
9.

H. : Die Entstehung des sogenannten angeborenen Schiefhalses. Beitrag zu einer Problemloeung dursh Zusammenwirken verschiedener Sonderf#{228}cher. (Geburtshilfe, Kinderheilkunde, Orthopadie, Pathologie, Erbkunde.) Wiener med. Wochenschr., 84: 1091-1097, 1934. ABELS, HANS: Ueber die Entstehungsweise des sogenannten angeborenen Schiefhalses, seine konstitutionsund Erbpathologischen Beziehungen. Ann. Paedit., 152: 4-34, 96-116, 1938. ABERLE, WALTHER: Atiologisches zum Schiefhals. Zeitschr. f. Orthop. Chir., 49: 27-43, 1927. ADAMS, R. D. ; DENNY-BROWN, D. and PEARSON, ; C. M. : Diseases of Muscle. A Study in Pathology. New York, Paul B. Hoeber, Inc., 1953. ALBEE, F. H. : Orthopaedic and Reconstruction Surgery. Industrial and Civilian. Ed. 1, p. 930. Philadelphia, W. B. Saunders Company, 1921. ANDERSON, WILLiAM: Clinical Lecture on Sterno-mastoid Torticollis. Lancet, 1 : 9-12, 1893. AREY, L. B. : Developmental Anatomy. Ed. 5. Philadelphia, W. B. Saunders Company, 1946. BARGELLINI, D. : Intervento precoce nel torcicollo muscolare congenito. Chir. d. Org. di Movimento, 16: 415-428, 1931. BAUMAN, G. I. : Discussion of Birth Injuries Requiring Orthopedic Treatments by S. W. Boorstein. J. Am. Med. Assn., 85: 1866-1870, 1925.
ABELS,

10.
12. 13. 14.

BEVAN,

A. E.

D. M. E.

: Congenital :Source H., and

Wry-Neck.

Surg. W. : Treatise in

Clin.

Chicago,

2:

895-900,

1918. Wilkins and Ed. 2. Company, New York, of 1948. William Circulation. Wood Arch.

1 1. BICK,

Book
LovErr, Pathologic

of Orthopaedics.
R.

Ed.
on Muscle

2.

Baltimore,
Orthopedic as a Result

Williams
Surgery. of

BRADFORD,

and Surg., 15.

16.
17.

Company,
BARNEY:

1899.
Changes Disturbances

BROOKS,

5:

CHANDLER,
CHANDLER,

188-216, 1922. F. A. : Muscular

F. H. A., 1944. A. : Oslers and

Torticollis.

ALTENBEEG,

ALFONS:

J. Bone and Congenital

Practice of

Joint

Surg., Muscular
Ed.

30-A: 566-569, Torticollis.

5. New York,

J.

July Am.
D.

1948. Med.

Assn.,

125:

476-483,

CHRISTIAN,

Principles

and

Medicine.

Appleton-Century

Company,
CLARK,
COLONNA, EDGEWORTU,

18.
19.

1944. W. A. : Torticollis. P. C. : Congenital

F. H. : Cited

California Torticollis.
by Straus

and
and

Western Virginia
Howell.

Med., 23: Med. Month.,

1429-1432, 794-796, 53:

1925. 1927.

20.
21. 22.

H. J. : Torticollis. Frrz SIMMONS, H. J.: Discussion J. Am. Med. Asan., 85: 1866-1870, FITZ SIMMONS, H. J. : Congenital 209: 66-71, 1933.
FITZ SIMMONS,
FURBRINGER,

J. Am. Med. Assn., 64: 645-649, 1915. of Birth Injuries Requiring Orthopedic Treatments 1925. Torticollis. Review of the Pathological Aspects.
and e cura Howell. chirurgica del torcicollo muscolare negli

by New

S. W. England

Boorstein. J. Med.,

23.
24. 25.

M.

: Cited
LUIGI:

by

Straus

DE

GAETANO,

Patogenesi

adulti.

Riforma

Med.,

51:

1463-1466,

1935.

GALLAVARDIN,

26. 27. 28. 29. 30.

31.

LOUIS, and SAvy, P. : Sur un as de torticollis c congenital avec autopsie et examen histologique du syst#{232}me nerveux. Lyon MM., 101 : 767-778, 1903. GOLDING-BIRD, C. H. : Congenital Wry-Neck. With Remarks on Facial Hemiatrophy. Guys Hospital Reports, 47: 253-273, 1890. GRANT, J. C. B. : A Method of Anatomy. Descriptive and Deductive. Ed. 4. Baltimore, Williams and Wilkins Company, 1948.

GREENSTEIN:

Personal

communication.

32. 33.
34.

E. : Discussion of article by Royal Whitman H Moarrz : Experimenteller Beitrag zur Aetiologie des Angeborenen muskularen Zeitschr. f. Chir., 49: 204-241, 1898. HELLSTADIUS, ARVID: Torticollis congenita. Acta Chir. Scandinavica, 62: 586-598, 1927. HEUSINGER: Cited by Joachimathal, p. d432 HILDEBRAND: Ueber doppelseitiges Caput Obstipum. Deutsche Zeitschr. f. 45: 584-594, Chir., HOLLOWAY, L. W. : Caput Obstipum Congenitum. Southern Med. J., 24: 597-601, 1931.
THE JOURNAL OF BONE AND JOINT

HADRA, HELLER, Deutsche

B.

Schiefhalses.

1897.

SURGERY

CONGENITAL

MUSCULAR A Review and

TORTICOLLIS

1181
Study. Surg., Gynec., Feb. III. and Obstet.,

:s. :36. 37. 38.

39.

HOUGH,

0.

DEN.,

Ja.

: Congenital

Torticollis.

Result

58: 972-981,
HULBERT,

1934.

40.
41. 42.

F. : Congenital Torticollis. J. Bone and Joint Surg., 32-B: 50-59, Untersuchungen #{252}ber die Heredit#{228}t orthopadischer Leiden. Schiefhals. Arch. f. Orthop. u. Unfall-Chir., 30: 459-494, 1931. JACOBS, C. M. : Congenital Torticollis. Illinois Med. J., 19: 727-735, 1911. JAHSS, S. A.: Torticollis. J. Bone and Joint Surg., 18: 1065-1068, Oct. 1936. JANrK, JAROMIR: Beitrag zur traumatischen Atiologie der Tortikollis. Zeitschr.
ISIGK.EIT, EDUARD:

K.

1950. Der Angeborene

f. Orthop.,

71 : 290-295,

19:1.
JEPSON,

P.

N. : Ischaemic

Contracture.

Experimental

Study.

Ann.

JOACH1MSTHAL,

Edited 43.
44.
JOHNSON, Frederick JONES,
pany,

G. : Schiefhals. In Handbuch der Orthopadischen by G. Joachimsthal. Jena, Gustav Fischer, 1905-1907. R. W. : Congenital Deformities. Part In A Textbook I. Christopher. Philadelphia, W. B. Saunders Company, ROBERT, and Lovsrrr, R. W. : Orthopedic Surgery. Ed.
BRONISLAUS: HELMUT:

84: 785-795, Surg., 1926. Chirurgie. Vol. I, Sect.

Surgery. Ed. 4, p.

2, pp.

423-486. by and Corn-

of

492. EdIted

1945. 2. New z. KIn.

York, Chir.,

William 18: 173-322,

Wood 1897.
Zentralbl. Zeitschr.

1923.

45.
46.

KADER, KASTENDIECK,

Das

Caput obstipum Uber die Atiologie

1939. und Behandlung P. 1955. K. PALLADINO, ;

musculare.
des des
\.

Beitr.

Angeborenen Caput

muskularen obstipum musculare.

Schiefhalses. Deutsche

f. f. Cb.ir.,

Gynak.,

63:

727-729, Ursache \V. 157:

ALl: 1904.

47. 48.
49.

KEMPF, 73:

F. : Uber

351-387, Assn.,

KIESEWETTER,

B. ; NELSON, 1281-1285,

S. ; tnd

Koop,

C. E. : Neonatal Acta Chir.

Torticollis. 56:

J. Am. 497-512,

Med.
KEOGIUS,

Zur

Pathogenese

des communication.
of

muskul#{228}ren Schiefhalses.

Scandinavica,

1924.

50.
51. 52. 53.

LAMBERT,

C.
W.

N. : Personal
H. H. D. : Development

LEWIS,

484-485.
MEYERDING,

Edited

by
\V.

Franz
: Congenital

Keibel

54.
55.

S. : The Pathology MIKULICZ, J. : Uber die Exstirpation des zur Pathologie dieses Leidens. Centralbl.
MIDDLETON,

System. Manual In of Human Embryology. P. Mall. Philadelphia, J. B. Lippincott Torticollis. J. Orthop. Surg., 3: 91-97, Jan. 1921. of Congenital Torticollis. British J. Surg., 18: the Muscular

and

F.

Vol. Company, 188-204,

nebst

I, pp. 1910. 1930.

Bernerkungen Company,

Kopfnickers

beim

Muskularen

Schiefhals, The

f. Chir.,
by J. P.

22:

1-9,

1895.
Philadelphia, Blakiston

MORRIS

HUMAN

ANATOMY. Ed.

10.

Edited

Schaefler.

1942. 56. 57.

58.

59. 60. 61.

62. 63.

A. H. : Bilateral Congenital Caput Obstipum. H. W. : Cited by Straus and Howell. NOV-JOSSERAND and VIANNAY, CHARLES: Pathog#{233}nie Rev. dOrthop., 7: 397-425, 1906. PARKER, R. W. In a discussion : of the Relationship
MORSE,

Surg.,
du torticollis

Gynec.,

and

Obstet.,

20: 1915. 74-77,

ischemique.)

Nounis,

congenital. Wryneck and

(Th#{233}orie

between and

Congenital
Society.

Haematoma
Lancet, 1 : 247,

of
1893.

the

Sternomastoid

at

a meeting

of

the

Royal

Medical

Chirurgical

PETERSEN,

FERDINAND:

Caput

obstipum.
des

(Zur

Aetiologie

und

Behandlung.)

Arch
Zentralbl.

f.

Klin.
f.

Chir.,
Gynak.,

30:

781-798, 1884. PETERSEN, FERDINAND: 10: 777-778, 1886.

PETERSEN,
FERDINAND:

Zur Frage

Kopfnickerh#{228}matoms

bei Neugeborenen.

Ueber
a discussion at a meeting

den
of of

angeborenen
the the Relationship Royal

muscul#{228}ren Schiefhals.
between Medical and Wryneck Chirurgical

Zeitschr.
and Society.

f.
Congenital

Orthop.

Chir.,
Haematoma

1:
of

86-113,
POWER, the REYE,

1892.
DARCY:

In

Sternomastoid

Lancet, 1 : 247,

64. 65.
66.

R.

D.
1951.

K. : Sterno-mastoid
HEINRICH:

Tumor

and

Congenital

Muscular

Torticollis.

Med.

1893. J. Australia,

1:

867-870,
VAN

ROONHYSEN,

Cited

Rossi, del
RUGH,

D. : Un
torcicollo.)

caso
Riv.

di torcicollo
dOstet.

by Joachimsthal . congenito in un feto

Prat., 10:

estratto
277-282,

con
1928.

parto

cesareo. by

(Contributo S. W. Beitr.
1937.

alletiogenesi Boorstein. z. KIm. J. Chir., Am.

e Ginec.

67. 68.
69.

J. T. : Discussion of Birth Injuries Assn., 85: 1866-1870, 1925. SCHLOESSMANN, H. : Die Entstehung des Med.
71: 209-253, 1911. L#{233}tiologie Zum Die du torticolis des der
SCHMID, SCHMIDT, WERNER: MEINHARD:
ALFRED:

Requiring angeborenen
musculaire.

Orthopedic

Treatments

muskul#{228}ren Schiefhalses.
Presse Med., 45: 1189-1191,

70. 71. 72.

Kapitel Ursachen

Schiefhalses. Angeborenen

Zentralbl. f. Chir., Schiefhalserkrankung.

17:

570-572,
Deutsche

1890.
Zeitehr. f. Chir.,

SCHUBERT,

167: 32-59, 1921. SEvER, J. W. : Discussion of Birth Injuries Med. Assn., 85: 1866-1870, 1925.

Requiring

Orthopedic

Treatments

by

S. W.

Boorstein.

J. Am.

1 182 73. 74. 75. 76. 77.

SHANDS,

R. A. R.,
JR.,

T.

LIDGE, R.

R.

C.

BECHTOL,

AND of Orthopaedic

C. N.

LAMBERT Surgery. Zeitschr. Ed. 2. St. Louis, 155: The 1-48, C. 1920. V.

and
1940.

RANEY,

B. : Handbook muskulare

Mosby

Company,

PAUL: Der angeborene H. R.: On Haematoma Bacteriol., 1: 112-116, 1892. SPENCER, W. G. In a discussion :
SPZNCIIE,

SIPPEL,

Schiefhals.

Deutsche

f. Chir.,

of the
of the
of the

Sterno-mastoid
Relationship
Royal

Muscle

in New-Born

Children.

J. Pathol. and

the

Sternomastoid

at

a meeting

Medical and

STzRN, AFREED: Zur Atiologie des Angeborenen 179-180, 1923-24. 78. STEVENS, A. E.: Congenital Torticollis in Identical Twins. Lancet, 2: 378, 1948. 79. STaAUS, W. L.,JR., and HOWELL, A. B. : The Spinal Accessory Nerve and Its Musculature. Quart. Rev. Biol., 11: 387-405, 1936. 80. STaEETER, G. L.: Development of the Nervous System. In Manual of Human Embryology. Vol.II, p. 139. Edited by Franz Keibel and F. P. Mall. Philadelphia, . B. Lippincott J Company, 1910. 81. STROMETER, 0. F. : Beitrage zuroperationen orthopaedik, oder erfahrungen ueber die subcutane durchscneidung verkurzter muskein une deren sehnen. Hannover, Helwing, 1838. 82. TAYLOR, FREDERiCK: Induration of the sterno-mastoid muscle. Trans. Pathol. Soc. London, 224-227, 26:
1875.

between Wryneck and Congenital Haematoma of Chirurgical Society. Lancet, I : 247, 1893. Sehiefhalses. Monatschr. f. Geburtsh u. Gynak., 65:

83. 84. 85. 86. 87.

V#{246}LCKER,

FRIEDRICH:

Das

Caput

obstipum-eine

intrauterine

Belastungsdeformitat.

Beitr.

z.

lOin.

Cb.ir.,

33:

1-71,

1902.

RICHARD: Das sogenannte angeborene Caput obstipum und die offene Durchschneidung des M. sternocleido-mastoides. Centralbi. f. Chir., 12: 233-236, 1885. VoN LACKUM, H. L. : Torticollis. Removal in Early Life of the Fibrous Mass from the Sterno-mastoid Muscle. Surg., Gynec., and Obstet., 48: 691-694, 1929. Wmmta.N, ROYAL: Observations on Torticollis, with Particular Reference to the Significance of So-called Haematoma of the Sterno-mastoid Muscle. Trans. Am. Orthop. Assn., 4: 293-307, 1891. WrrzzL, Oscii: Beitrage zur Kenntniss der secund#{228}ren Veranderungen beim muscularen Schiefhalse. Deutsche Zeitschr. f. Chir., 18: 534-578, 1883. VOLKMANN,

the

THE

JOURNAL

OF

BONE

ANDJOINT

SURGERY