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A. Landi et al.
Fig. 32.5 (a) The sequela of the skin scar at the postero-
lateral compartment. (b, c) At one-year neurolysis, of the
median and ulnar nerve, transfers of BR to wrist extensors
were carried out. (d–f) Follow-up at six years: good range
of motion at the elbow associated with active wrist extension. Grip strength measured by Jamar reduced to onethird compared to the unaffected side
believe that scar excision and palliative surgery
should be postponed to an age where one can rely
on the understanding and collaboration of the
young patient (Fig.32.6). In severe injuries, the
most affected compartment is usually the volar
one where often free muscle transplantation
should be carried out, while in the dorsal compartment, there is often the possibility of pallia-
cd
32 Compartment Syndromes (CS) and Volkmann’s and Upper Limb Vascular Pathology in the Peri…
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a b
403
Fig. 32.6 (a) The scar sequela of a sentinel lesion with
the underlining Volkmann’s contraction of the muscle of
the postero-lateral compartment. (b) No active contraction of the EDC while EIP and EDM are normal. FCU was
transferred early in life to EDC leading, during growth, to
tive surgery preceded by excision of the scar
tissue. In the severe Volkmann’s retraction of
both compartments, similar to the adult, there is
no alternative to the double free muscle transplant [21].
In the infant who suffered severe neonatal
compartment syndrome leading to both volar and
dorsal compartment muscle losses, a staged
approach is warranted. Exploration with debridement of nonviable muscle as well as neurolysis is
important, as this prevents xed contractures and
assists with re-gaining hand sensation and intrinsic muscle function. This should be performed
within the rst few months of life if the patient is
medically stable. It is important to suture the
ends of the FDP tendons to the radius in the forearm to prevent retraction into the carpal tunnel,
which can make secondary reconstruction much
more difcult. In addition, tagging of the ends of
the future donor nerves, most commonly anterior
a gradual dorsal contracture of the wrist. (c, d) Long-term
follow-up after tenolysis of the tendon transfer and dorsal
capsulotomy of the wrist. Normal function of
EDC.AROM at the wrist improved from −70° xed in
extension to +20° of active exion
and posterior interosseous nerves, can be very
helpful when performing the secondary functioning muscle transfer. Occupational therapy is also
an imperative adjunct in these patients to obtain
full passive range-of-motion and prevent xed
contracture. This allows secondary free functioning muscle transfer to be performed when the
patient is older, 1–3 years of age, and structures
are larger. Usually, the exor compartment is rst
to be reconstructed, followed by the extensor
compartment 1–2 years later (Fig.32.7) contrary
to Chuang who performed by rst reconstructing
the muscles of the extensor group followed by the
free transplant in the exor group when the rst
ones displayed signs of reinnervation [11].
Permanent nerve injuries can be prevented when
they are supercialized at an early stage.
Otherwise, the repair will be proportional to their
severity: neurolysis, nerve grafts or implementation of the Strange technique [53, 54].
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A. Landi et al.
c
Fig. 32.7 (a) Initial presentation two weeks after birth.
(b) After debridement and therapy, prior to free functioning muscle transfer. (c) Intra-operative debridement sur-
gery. (d) Postoperative gracilis transfer to volar and dorsal
compartment
32 Compartment Syndromes (CS) and Volkmann’s and Upper Limb Vascular Pathology in the Peri…
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d
Fig. 32.8 Vascular lesion of a newborn left upper limb
where multiple debridements were carried out. The whole
area covered by granulation tissue ready for skin grafting
405
Fig. 32.7 (continued)
32.6 Vascular Lesions
32.6.1 Spontaneous Vascular Lesions
Neonatal ischaemia of the upper limb represents
a rare occurrence, and to distinguish it from the
compartment and Volkmann’s syndromes
described in the previous section, it can be simply
assumed that for its treatment, a decompression
of the affected muscle compartments is not sufcient, whereas a substantial medical or surgical
intervention on the arterial or venous system is
indispensable [55].
A fasciotomy should be considered a complementary measure, but the gangrene of the newborn (Figs.32.8 and 32.9) can only be prevented
by intervening directly on the major vascular
axes.
Fig. 32.9 Clinical picture of a severe ischaemia of the
left upper limb
When this is not feasible for technical reasons
(avulsion of iliac vessels) or general health of the
newborn (coexisting hyaline membrane diseases), the residual survival of the limb is
entrusted to the establishment of collateral
circulation.
The experience on which this chapter is based
refers to the 32 cases already reported by GEOP
(Paediatric Orthopedic Group) collected in
France, Canada, Portugal and Belgium [29] in
addition to 3 cases, treated by the Modena Hand
Surgery Unit, for a total of 35 cases.
Vascular lesions of the upper limbs in the perinatal period are quite rare: it should be considered that less than 100 cases of ischaemia of the
limbs (upper and lower) at birth or in the rst
hours of life are reported in the literature [38].
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A. Landi et al.
Vascular lesions may be spontaneous and
often generated as a result of renal vein thrombosis where the presence of calcic thrombus stands
for an intrauterine genesis [56].
Vascular lesions may fall into group I
(Table32.2) due to impairment of a large arterial
vessel (phlegmasia alba dolens) or venous vessel
(phlegmasia cerulea dolens) or group III where
pre-existing systemic factors (meningococcal
septicaemia, epidermidis staphylococcal septicaemia, dehydration, etc.) are to be considered
responsible for the involvement of several limbs
with consequent necrosis of small and large
segments.
In utero ischaemia of the limb may be associated with necrosis of the ipsilateral hemiface generated by a placental thrombus likely migrated
through the oval foramen destined to the brachiocephalic arterial trunk [35].
Limb ischaemia manifests itself at birth or
within a few hours after birth. Several conditions
can cause ischaemic episodes: thromboembolism
could be a cause and be associated with maternal
diabetes, gestational hypertension (eclampsia
and pre-eclampsia), oligohydramnios and pressure of maternal pelvic structures. Venous
thrombosis and thromboembolism are wellknown complications in infants of diabetic mothers. Oppenheimer and Esterley, in their 4000
newborn autopsies, describe 45 cases of venous
thrombosis, 13 of which from diabetic mothers
[57]. Renal and adrenal veins are usually more
frequently involved.
Vallerame [58] published the rst case of
peripheral gangrene in a newborn from a diabetic
mother in 1972, hypothesizing that the adrenal
vein was the probable starting point of the
embolus occluding the right brachial artery, which
it reached through the patent foramen ovale.
Carr [41] hypothesized that the emboli resulting from a placental infarction could be transported in the right ulnar artery and in the right
internal carotid artery of the newborn through the
same foramen.
Extremity gangrene is a very likely complication of neonatal infections in the presence of
severe dehydration [6, 35] or meningococcal [34]
(Fig.32.10) or staphylococcus epidermidis septi-
caemia (Table32.2). There is also the sector of
multiple gangrene with self-amputation in association with severe dehydration, even in the
absence of occlusive vascular phenomena. The
most frequent cause is related to adverse underlying conditions, such as illiteracy and tribal practices, which for the treatment of mild intestinal
disorders resort to herbal enemas that also have a
cytotoxic effect on the kidney and liver. Herbal
enemas can have a phlebo-occlusive effect and
the vaso-constrictive effects typical of the alkaloids of ergot [6]. The clinical picture is characterized by dehydration, acidosis, hyperuricemia
and renal failure. Gangrene can affect all four
limbs, the tip of the nose, ears or perineum as in
case of meningococcal septicaemia.
On the neonatal side, the immaturity of the
coagulation systems and the presence of congenital thrombophilia play a relevant role [38].
Moreover, in the cardiovascular pathology, anatomical anomaly situations can be at the basis of
a paradoxical embolism that starting from the
maternal or foetal periphery (placental embolism
or renal and adrenal veins) is distributed to the
vascular district of the upper limb (an axillary or
humeral) and/or to the cerebral circulation
becoming responsible for the porencephalic
degeneration found in 9 of the 32 cases reported
by Romaña [29]. The most frequent cardiovascular malformations associated with upper limb
abnormalities are atrioventricular defects (atrial
septal defect (ASD) and ventricular septal defect
(VSD)), Fallot’s tetralogy, large vessel transposition and the presence of the common arterial
trunk [55, 59], but especially emboli that can
spread from the closing arterial duct.
32.6.2 Iatropathic Causes
The most frequent etiopathogenesis of vascular
lesions of the upper limb, however, is iatropathic,
linked to the more aggressive attitude in the treatment of infants at risk in intensive care units, and
it might include a false aneurysm caused by the
perforation of the lower third of the humeral
artery that required excision and microsurgical
repair [60].
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Fig. 32.10 (a) Four-limb amputation as consequence of
a meningococcal septicemia occurred after birth. Residual
trans-metacarpal amputation on the right side with minimal function. (b) Corresponding X-rays. (c-d) Deepening
of the rst web by a local plasty according to the
Ostrowsky technique. (e) Functional opening of the web.
(f) Possibility of performing bimanual activities
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A. Landi et al.
In a seemingly inexplicable way, even the
injection of an unknown substance, in a newborn
suffering from gastroenteritis into the anterior
belly of the deltoid muscle, resulted in severe
vascular suffering followed by an amputation of
the middle third of the arm [61].
Regarding the use of central venous catheters
(CVC), the access can be supercial or deep:
among the supercial access routes, it is worth
mentioning the cephalic, the basilic and the external jugular veins and among the deep ones, the
internal jugular and the subclavian veins.
Venous embolisms can pass to the arterial circle, always through the oval foramen, causing
peripheral ischaemia of the limbs.
Data from the Canadian Registry showed an
incidence of 3.5 events of thromboembolism
related to the presence of CVC for every 100,000
admitted patients. According to the same authors,
the placement of a central route, used for the
administration of liquids, drugs, parenteral nutrition or chemotherapy, would be responsible for
about 90% of DVT (deep vein thrombosis) and
pulmonary embolisms in the rst year of life [38].
The indication to use arterial catheters is based
on the need to perform frequent blood tests or to
continuously monitor blood pressure. In the neonatal age, within the rst 24–48 h of life, arterial
cannulation can be performed through the umbilical arteries.
In case of failure of the umbilical access or in
case of infants over 48 h of life, the radial artery
stands for the most used access; the criteria of
choice towards this artery are represented by its
supercial anatomical path and good secondary
circulation (validated by Allen’s test). The permanence of the catheter in the artery must not
exceed 72–96 h.
Ischaemia is not the only complication that
can occur after radial catheterization: there can
also be a bacterial infection. The most frequently
identied germs are staphylococcus and streptococcus, which are usually non-pathogenic but
might become so in the presence of debilitated
patients or those with an underdeveloped immune
system like the newborn.
32.6.3 Complications inStabilized
Outcomes
CS and Volkmann’s perinatal pathology are peculiar conditions connoted by scanty report and
unavailable long-term outcome studies.
There are substantial differences in the timing
of execution of fasciotomy, especially between
the newborn and adult. In the adult, fasciotomy
may sometimes be useless if performed for a
suprafascial oedema mainly in burns carrying the
side effects such as lack of strength at the hand
and fatigue in walking [15].
Late fasciotomy is often subject to bacterial
colonization, and when incomplete, it can be
accompanied by the brous evolution of the nondecompressed myotendinous components,
whereas late paediatric fasciotomy is not necessarily related to irreversible outcomes [18, 21]. In
traumatology, fasciotomy has been performed for
up to 88 h [7] 7 days [62] with an almost always
complete recovery of function [63].
This observation is conrmed by a retrospective analysis conducted at the Mayo Clinic by
Eichler and Lipscop [64]. This observation suggests that in these circumstances, there might be
a subliminal muscle circulation or early neoangiogenesis in children that guarantees their survival or that the muscles in the perinatal age
group have a greater tolerance to ischaemia than
in adulthood.
In the surgical treatment of Volkmann’s contracture, the possible complications are liaised to
exor origin sliding or functional free muscle
transfer. In this last scenario, the complications
can be categorized as acute or chronic: underlying ap loss (either skin paddle or partial muscle
loss). Long-term complications include scarring
and tendon attenuation at the repair site [5, 21].
In paediatric patients, bone growth may be
more rapid compared to the transferred muscle,
to which the following coercive aspects to the
surrounding scarring retractions can be added,
which can lead to joint contracture and tendon
adhesions despite excellent muscle contraction
[13, 21]:
32 Compartment Syndromes (CS) and Volkmann’s and Upper Limb Vascular Pathology in the Peri…
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– The slowly increasing tenodesis effect of a
premature tendon transfer, which might lead
during growth to an extension contracture of
the wrist.
– Ulnar deviation of the wrist usually occurs
when muscle sliding has been associated with
carpal tunnel decompression leading to a volar
dislocation of exor tendons which implies
that the tunnel should be repaired in the rst
instance.
– Joint stiffness mainly at the MPJ and wrist
level which can assume aspects of severity
that can only be encountered in arthrogryposis
and may require rst row carpectomy.
– Pseudoarthrosis, retracting scars and contrac-
ture of the rst web space (Fig.32.11) may be
present both in Volkmann’s outcomes and in
vascular lesions of the perinatal period [17].
32.6.4 Dierential Diagnosis
The almost constant skin lesion is the one that
has certainly aroused the most interest in the various medical disciplines. Effectively, this area of
the skin with the subcutaneous tissue with an
inammatory reaction can occur in different
elds. This description coincides with the one
given by R.Lightwood [2] and by Haymann [3]
where three cases of infants who had fat necrosis
were reported above the elbow were suggestive
of compression syndrome of the radial nerve in
the region of the spiral groove. C.V Feldmann
[65] also reported four cases, one of which was a
bilateral paresis of the radial nerve highlighted
three days after birth associated with fat necrosis
of the lateral surface. Function of both wrists was
fully restored after three months. A similar experience is reported by Rombouts [9].
Sclerema neonatorum is a systemic form
which manifests itself with a hardening of the
skin that suddenly appears in the third or fourth
day of life, often in malnourished, dehydrated,
hypotensive, hyponatremic or hypoglycaemic
infants. The thickening often starts from the
lower limbs and spreads to the trunk and cheeks.
Sclerema is probably the result of an enzymatic
neonatal dysfunction involved in the conversion
of saturated palmitic and stearic acid into unsaturated oleic acid [66].
Aplasia cutis represents a group of congenital
skin lesions ranging from the absence of skin to
small achromic scars. The preferred location of
these lesions, however, is the scalp but the forearm is also frequent. The classical pathogenetic
theory of aplasia cutis hypothesized an adhesion
between lesions affecting the teguments of the
foetus and amnion. It could also be a genetically
determined skin malformation because more than
50% of cases are associated with trisomy 13 [3,
17].
Analysing all the reported cases, the neural
damage, when present, is framed by a temporal
paralysis of the radial nerve that recovers
spontaneously and is therefore easily distinguishable from the muscle contracture associated with
a Volkmann’s syndrome of the lateral-external
compartment.
Moreover, the congenital deformity in exion
of long ngers might be connected to an aberrant
origin to exor digitorum profundus [67], a condition that belongs to the eld of congenital
malformations.
32.6.5 Long-Term Outcomes
As expected, long-term outcome following neonatal compartment syndrome is largely dependent on the severity of involvement. In cases with
a skin lesion but no muscle dysfunction or compartment ischaemia, near full function can be
expected although abnormal forearm growth is
still the norm and should be discussed with the
parents. This is in sharp contrast to the patient
that has exor and extensor compartment involvement who did not undergo emergent fasciotomy
at birth. Depending on the time delay until exploration, these patients may have severe joint and
skin contractures along with severe neurologic
decits in the hand that must be addressed. Even
with debridement, neurolysis +/- nerve grafting
and free muscle transfer hand function may be
limited. However, if timely staged reconstruction
is performed, good to excellent results can occur
even in the severely involved patient. As dis-
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b
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Fig. 32.11 (a) Skin necrosis at the dorsal aspect of the
wrist following extravasation of antirejection drug administration following a renal transplantation in the rst year
of life. (b) Surgical debridement and resurfacing by dermal graft. (c) Clinical appearance a still unclassied syndromic pattern. (d) Severe rst web contracture. (e)
Resurfacing by dermal graft. (f) Clinical result following
release of the rst web space initially caused by the
Volkmann’s contracture of the intrinsic muscle. (g)
Functional opening of the wrist web consenting a useful
key-pinch
32 Compartment Syndromes (CS) and Volkmann’s and Upper Limb Vascular Pathology in the Peri…
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e
g
Fig. 32.11 (continued)
cussed earlier, less involved cases can undergo
expectant management depending on the clinical
progression of deformity.
32.7 Prevention
f
acquired in 71.4%. Foetal morbidity was mainly
linked to the consequences of premature rupture
of the membranes (57.1%) and preterm birth
(28.5%). The AA concluded that fetoscopic band
release was encouraging in cases of ABS in the
limbs [69].
In the majority of cases, the newborn will be
affected by an irreversible established Volkmann’s
contracture. Therefore, it would be reasonable to
check if any preventive fetoscopic surgery could
be possibly undertaken during the intra-uterine
phase. Mentioning of this possibility is made
only for amniotic band syndrome to prevent
amputation caused by the attachment or entanglement of amniotic membrane remnants to the
foetus. The median gestational week at diagnosis
was 22 weeks/5 days [68]. A literature review
included a group of patients who had the fetoscopic release of the amniotic bands using a laser
(16 cases), scissors (6 cases) or both (5 cases) in
a series of 27 cases. In only one case, the hand
and ngers were affected; some of his ngers
were amputated. In the majority of cases, a single
entry was made and a functional limb was
32.8 Conclusions
The borderline between perinatal CS and
Volkmann’s contracture in the newborn and in
the vascular lesion appears to be very subtle; this
is the reason why these topics have been assembled in this chapter [23].
For the purpose of the classication of the
pathophysiological aspects, we relied on the
Holden classication, already described in 1958
by Perricone, to which we made added signicant modications [16]. Group A concerning the
lesion of a proximal major vessel, which we
divided into arterial and venous components
including also the distal pathology, is related to
the vascular compromise of a muscle with a single vascular supply (Table32.1).
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