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European Journal of Plastic Surgery

https://doi.org/10.1007/s00238-021-01790-9

CASE REPORT

Medial cutaneous nerve neuroma-in-continuity within the ulnar


nerve: a report of 2 cases
Jantine PosthumaDeBoer 1 & Raf Sciot 2 & Ilse Degreef 1

Received: 25 December 2020 / Accepted: 19 January 2021


# The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature 2021

Abstract
Cubital tunnel syndrome is the second most prevalent entrapment neuropathy in the upper extremity and, therefore, surgical
decompression of the ulnar nerve is regularly performed. During this procedure, the medial antebrachial cutaneous nerve
(MACN) branches are at risk for iatrogenic injury because of their proximity to the medial incision used. Injuries to the
MACN may lead to painful neuroma formation and, more seldomly, to formation of neuromas-in-continuity.
Neuromas-in-continuity are challenging to treat as the neuroma needs to be dissected from nervous tissue that must remain
functional. In this paper, we present two cases of MACN neuromas-in-continuity within the ulnar nerve. We provide a detailed
description of the clinical features of painful MACN neuromas-in-continuity and provide a detailed description of two different
treatment options. This work highlights the advances made in microsurgery which provides better treatment possibilities for these
highly complex sequelae of ulnar nerve decompression surgery.
Level of evidence: Level V, risk / prognostic study.

Keywords Intraneural neuroma . Neuroma-in-continuity . Ulnar nerve . Microsurgery

Introduction Although persistent or recurrent pain after surgical ulnar


nerve decompression may be due to various reasons, injury
Cubital tunnel syndrome is the second most prevalent entrap- to branches of the MACN is one of the most common causes.
ment neuropathy in the upper extremity with surgical decom- Moreover, painful neuromas that arise from injured MACN
pression by opening the Osborne fascia as regularly per- branches may mimic recurrence or persistence of the ulnar
formed operative treatment. The medial antebrachial cutane- nerve compression syndrome, and neuromas can be
ous nerve (MACN) with its anterior and posterior branches misdiagnosed as such [2–4]. For the treating physician, it is
lies in close proximity to the incision used for ulnar nerve important to be aware of both the relevant MACN branches as
decompression surgery which makes it susceptible to injury. well as the diagnosis and treatment of painful neuromas. Here,
Consequently, iatrogenic injury of the MACN is a notorious we report 2 particular cases with a painful MACN
complication after ulnar nerve decompression. It can lead to neuroma-in-continuity within the ulnar nerve and the success-
scar pain, painful neuroma formation, hypaesthesia and ful treatment thereof.
hyperalgesia both locally and in the area of the sensory distri-
bution of the MACN with a significant negative impact on the
quality of life for the patient [1–4].
Case reports

Case 1
* Jantine PosthumaDeBoer
Jantine.posthumadeboer@uzleuven.be In 2012, a 40-year-old otherwise healthy male patient present-
ed to our outpatient clinic after having had multiple surgical
1
Department of Orthopaedic Surgery, University Hospitals Leuven, procedures to his left elbow within a 3-year period following a
KU Leuven, Herestraat 49, 3000 Leuven, Belgium complex elbow fracture dislocation. He had a radial head im-
2
Department of Pathology, KU Leuven and University Hospitals plant with secondary capitellum erosion and concurrent
Leuven, Leuven, Belgium cubital tunnel syndrome. Therefore, a radiocapitellar joint
Eur J Plast Surg

arthroplasty was performed with ulnar nerve decompression Case 2


in situ. One year after this procedure, he had a painful
ulnohumeral joint osteoarthritis and was treated with a total In June 2020, a similar case presented at our outpatient clinic.
elbow arthroplasty. Although initial postoperative recovery A 46-year-old male requested a second opinion regarding
was uneventful, the patient developed hyperaesthesia at the persisting ulnar-sided forearm pain. His medical history re-
medial elbow scar in the course of the following year, which vealed a cubital tunnel release with anterior transposition of
progressed to unbearable and debilitating pain. He had a pos- the ulnar nerve in 2013. In 2017, because of recurrent symp-
itive Tinel sign over the scar without altered sensibility in the toms, a neurolysis and anterior transposition of the ulnar nerve
ulnar nerve distribution or impaired motor function in the was performed. Although his complaints appeared to resolve
ulnar two digits. initially, the patient suffered from recurrent medial-sided arm
It was suspected that the patient suffered from ulnar pain by the end of 2018, with additional reduced motor func-
nerve or MACN adhesion to the scar, and after informed tion and force of the left hand. In his file, it was mentioned that
consent, a surgical exploration with × 4 loupe magnifi- clinical examination revealed a sensitive string-like structure
cation was scheduled. However, during surgery, we at the medial side of the elbow which was thought to be the
found that the MACN ran alongside the ulnar nerve ulnar nerve. Electromyographic examination (EMG) of the
and formed a neuroma which seemed to enter the ulnar ulnar nerve was normal at that time. Early 2019, a third ex-
nerve. The MACN calibre had increased to 4 mm over a ploration and neurolysis was performed, and here, the surgeon
length of approximately 5 cm and at the location where had found a neuroma. It was judged that dissection was im-
this nerve attached to the ulnar nerve, the ulnar nerve possible, and therefore, a partial resection of the neuroma was
had an irregular and swollen morphology for a length performed. After this last procedure, the patient experienced a
of 2 cm (Fig. 1a). At this time, it was judged that exci- decrease in pain severity, but local scar tenderness persisted as
sion or biopsy of the neuroma would pose too high a well as reduced sensation in his little finger and the ulnar side
risk of damaging the ulnar nerve which was clinically of his hand.
fully functional, and therefore, this neuroma- Upon presentation at our clinic in July of 2020, his com-
in-continuity was left intact. Instead, the nerves and plaints had again recurred, with debilitating pain at the medial
neuroma-in-continuity were wrapped in a cellulose sheet elbow, paraesthesias in the 4th and 5th digits and a slight loss
(Divide™, DePuy Mitek, Johnson & Johnson Medical in strength. The patient mentioned irradiating pain upon pal-
Inc., New Brunswick, NJ, USA) and were then trans- pation of the medial side of the left elbow which increased
posed underneath the flexor pronator muscle group during elbow flexion. Ultrasound investigation revealed a tra-
(Fig. 1b, c). Postoperatively, the arm was immobilized jectory of the ulnar nerve in the subcutaneous fat with an
in a sling for 3 weeks after which full arm use was increased neural diameter and surrounding oedema. A repeat
allowed. The patient reported that the hyperaesthesia at EMG of the ulnar nerve was again normal.
the medial elbow had diminished immediately postoper- After informed consent, we proposed a fourth microsurgi-
atively. One year postoperatively, the patient remained cal exploration of the nerve with a sub-muscular transposition.
very satisfied without symptoms, and he had normal ul- During surgery, we found the ulnar nerve to be severely buck-
nar nerve sensation and motor function. led underneath the fascia. As we dissected further proximally

a b c
UN
MACN

UN
UN

Fig. 1 a Neuroma-in-continuity (N) sprouting from the medial cutaneous antebrachial nerve (MACN) running into the ulnar nerve (UN). b Wrapping of
the neuroma-in-continuity in a cellulose sheet and (c) subsequent submuscular transposition
Eur J Plast Surg

to where the ulnar nerve had a normal morphology, we also digits had ceased, and sensation returned to normal. Three
found the MACN that ran alongside the ulnar nerve which months later, the patient was extremely satisfied with a
then, similar to the first case, formed a neuroma attached the full and pain-free range-of-motion of the elbow joint and
ulnar nerve (Fig. 2a). This time, we proceeded to excise the normal sensation and motor function.
neuroma by an intraneuronal neurolysis with x6 to × 20 mi-
crosurgery magnification. The MACN neuroma was released
completely from the ulnar nerve (Fig. 2b–e, Supplementary Discussion
film 1) and was then excised with the proximal stump buried
deeply subcutaneously above the elbow (Fig. 2f). The ulnar The MACN is the second branch of the medial cord, aris-
nerve had a neuropathic appearance with a slim diameter for ing from the C8 and T1 nerve roots. This purely sensory
several centimetres. It was further released distally up to nerve innervates the distal part of the medial upper arm,
where its morphology and calibre appeared normal again. cubital fossa, posterior olecranon and volar ulnar side of
Finally, the ulnar nerve was transposed to underneath the the forearm. About 4–6 cm proximal to the medial
common flexor origin (Fig. 2g). epicondyle, the MACN divides into an anterior and a pos-
Histopathological examination of the excised tissue re- terior branch. The anterior branch pierces the brachial
vealed a haphazard proliferation of Schwann cells as is typi- fascia anterior to the elbow and then runs subcutaneously
cally seen in a traumatic neuroma (Fig. 2h, i). in the forearm. The posterior branch runs in close prox-
Postoperatively, a compressive bandage was kept on imity to the basilic vein with terminal branches that cross
for 10 days, and the patient used a sling for 4 weeks. ulnar and posterior at variable distances from the medial
The patient mobilized the limb as pain allowed. He re- epicondyle, which puts them particularly at risk for tran-
ported an immediate alleviation of his complaints after section during cubital tunnel release surgery [1–6].
surgery. At 3 weeks, the paraesthesias in the 4th and 5th Reports on the exact position and numbers of these

a UN b UN c
UN
MACN MACN

N
N N

d e f
UN
UN
UN
MACN MACN

N
N

g UN h i

Fig. 2 a, b Neuroma-in-continuity (N) of the MACN enveloping the ulnar nerve (g). h, i Histopathological views of the tissue after S100
ulnar nerve (UN) and the start of the microdissection. The ulnar nerve staining for Schwann cells. The remnants of the MACN are indicated
is released from the surrounding neuroma (c) and has a very thin calibre at with asterisks (*), and the arrows indicate irregular proliferation of nerve
the level of the neuroma (d). e The ulnar nerve is completely released, the branches
neuroma is excised (f) followed by a submuscular transposition of the
Eur J Plast Surg

posterior MACN branches vary. Race and Saldana [6] sensation, mostly in the olecranon or posteromedial forearm
performed a cadaver study on 20 arms and found 1 to 3 region, which is noticed only upon touch and is generally well
divisions of the anterior branch and 3 to 7 divisions of the tolerated [2–4, 6]. However, when the transected branch ad-
posterior branch that cross between 6 cm proximal and heres to the scar, it may induce scar pain. This is usually sharp
6 cm distal to the medial epicondyle. The authors there- and local but sometimes radiates distally to the sensory ulnar
fore warn that when using the standard incision for ulnar nerve distribution. Scar tethering is reported to lead to a longer
nerve surgery it is highly likely that terminal branches are length of nerve damage and neuroma formation. Once patients
injured unless they are carefully dissected. develop such elongated types of neuroma, they often have
Benedikt et al. [1] found 23% of crossing branches proxi- neuropathic pain which is notoriously difficult to treat. Also,
mal and 77% distal to the medial epicondyle in 40 cadavers. neuromas can become so severely painful that upper limb
On average, there were 2.95 crossing branches distal to the function is heavily impaired and patients are debilitated
medial epicondyle with an average distance of 2 cm to the [2–4, 8]. Furthermore, these painful neuromas often cause
epicondyle. Likewise, Manoukov et al. [4] found 1 to 3 pos- symptoms highly similar to cubital tunnel syndrome and
terior branches of which 18% emerged proximally and 72% may be misdiagnosed as recurrent ulnar nerve compression
distally to the medial epicondyle in a study of 13 cadavers. [4]. Again, vigilance is important to set the correct diagnosis
The average distance from the medial epicondyle was in persisting pain after longitudinal incisions at the ulnar as-
2.53 cm, which is around 1 cm distal to where the ulnar nerve pect of the elbow.
penetrates the heads of the flexor carpi ulnaris muscle (FCU). Neuromas develop after nerve injury as a result of fail-
Also, they reported that the distance to the distal crossing ure of a regenerating nerve to reach its peripheral targets,
increased in elbow flexion, a potentially protecting manoeuvre and as a consequence, degeneration and fibrosis of the
to the posterior branches from damage during surgery. nerve fibres occur. Neuromas-in-continuity are usually
The fact that vigilance for the MACN is warranted in ulnar the consequence of partial nerve injury. They may devel-
nerve surgery is further underlined by Lowe et al. [2] who op within one nerve after internal damage to the nerve
studied its position in 97 patients undergoing ulnar nerve re- fascicles, and usually, the distal part of this nerve no lon-
lease. They found that the posterior branch crossed at an av- ger functions properly [9]. Alternatively, after transection,
erage distance of 1.8 cm proximal to the medial epicondyle in the axonal sprouts of the proximal portion of the damaged
61% of patients and that all patients had at least 1 branch nerve fascicles may grow out of their own neurotubular
crossing distally at an average 3.1 cm distance. Indeed, we sheaths and show aberrant in-growth into a nervous struc-
may derive from these studies that most, if not all, patients ture that lies within close proximity to the damaged end,
undergoing cubital tunnel release surgery have at least one which is not its own distal end. As such, here we report 2
posterior MACN branch crossing the operative field and that cases with a neuroma-in-continuity of the MACN within
caution is required not to damage them. the ulnar nerve. Sprouting and invasion of axons of dam-
Persistent pain after ulnar nerve decompression surgery is aged nerve endings into intact nerves has been studied in
not uncommon. It is reported to occur in about 10% of patients the context of nerve bypass grafting and is referred to as
who suffered a moderate compression; however, in patients neurotisation. It is stated that regenerating axons can form
treated for severe ulnar nerve compression, it can be as high as from both ‘recipient’ and ‘donor’ nerves [10]. In the cases
20–35% [7]. The pain may be attributed to pressure on the we described here, however, it appears that the MACN
ulnar nerve by scar formation, kinking, and compression of a sprouts failed to achieve a functional neurotisation to the
transposed nerve by fascial flaps or tendinous bands, but also ulnar nerve and painful neuromas-in-continuity
to the formation of MACN neuromas due to iatrogenic injury. developed.
Reoperation is often considered in case of persistent or recur- The treatment of painful neuromas is considered challeng-
rent pain. Neuroma formation is estimated to occur between ing and is not always successful. In general, the neuroma is
40 and 73% of patients with persistent pain after ulnar nerve dissected from its surrounding tissues followed by capping the
decompression surgery. Mackinnon et al. [3] reported a very distal nerve with bipolar cautery and transposition deep into
high incidence of MACN neuromas of 73% in patients need- the subcutis or a local muscle belly. Wrapping the affected
ing secondary procedures due to persistent pain after surgery nerve end with autologous tissue or artificial materials has also
to the ulnar nerve in a chart study of 100 patients. been described [2–4, 8].
Iatrogenic injury to a branch of the MACN may cause a Management of a neuroma-in-continuity is considered to
variety of symptoms. Most benign is a simple loss or reduced be even more challenging than the treatment of a neuroma,
Eur J Plast Surg

since it needs to be dissected from nervous tissue that must this may mimic recurrent cubital tunnel syndrome.
remain unharmed. Treatment options include neurolysis, neu- Treating physicians should however keep in mind this al-
roma resection with interposition nerve grafting, wrapping in ternative differential diagnosis. We reported 2 surgical
local flaps, submuscular transfer with wrapping with cellulose treatment options for painful neuroma-in-continuity with
membrane, or resection of the neuroma using microsurgical a time interval of 8 years which highlights the technical
techniques [3, 4, 8]. Technical advancements in the field of advancements in the field of microsurgery over the past
microsurgery have allowed for meticulous dissection with years. Whereas in earlier days, we opted for wrapping
high magnification; however, risk always remains in terms and submuscular transposition of the neuroma-in-
of microvascular damage to the nerve, damage to remaining continuity; we were now able to microsurgically dissect
viable axons and the formation of intraneuronal scars. and excise the neuroma followed by submuscular transpo-
With the aim to avoid surgery in such highly complex sition of the ulnar nerve, yielding good clinical outcome in
sequelae in the future, basic scientific work is undertaken our patients.
to investigate the application of molecular treatments
against neuroma formation. Pre-clinical research on the Supplementary Information The online version contains supplementary
material available at https://doi.org/10.1007/s00238-021-01790-9.
use of targeted antibodies is performed to selectively stop
neuroma-in-continuity formation by selective destruction
of aberrant neurons [9]. This designated ‘molecular neu- Declarations
rosurgery’ seems highly appealing; however, results are
Conflict of interest Jantine PosthumaDeBoer, Raf Sciot and Ilse
still very preliminary and the feasibility of such innova- Degreef declare no conflict of interest.
tive and even promising future strategies is yet unclear.
Therefore, at present, (microsurgical) treatment of painful Ethical approval All procedures performed in studies involving human
neuromas-in-continuity is still recommended. Elliot et al. participants were in accordance with the ethical standards of the institu-
tional and/or national research committee and with the 1964 Helsinki
[8] reported on 14 neuromas-in-continuity, 2 of which in
Declaration and its later amendments or comparable ethical standards.
the ulnar nerve. Treatment included external neurolysis of No ethical approval was necessary for this case report.
the neuroma and circumferential wrapping with local fas-
cial flaps. In 8 patients, the symptoms resolved complete- Patient consent Written informed consent was obtained from the pa-
ly. Furthermore, they obtained a significant pain reduction tient regarding publishing his data and photographs.
in all patients in various categories of pain, except in
Informed consent The authors confirm that human research participants
hypersensitivity pain. Notably, the neuromas-in-
provided informed consent for publication of their data and images.
continuity studied in this work lie more distal than the
cases that we described here, so these results may not be
exactly comparable. Nonetheless, these results indicate
that patients may be successfully treated, although a con-
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