Home Lateral Cutaneous Nerve Entrapment Syndrome (LACNES): A previously unrecognized cause of intractable flank pain
Article Publicly Available

Lateral Cutaneous Nerve Entrapment Syndrome (LACNES): A previously unrecognized cause of intractable flank pain

  • Robbert C. Maatman EMAIL logo , Nicole E. Papen-Botterhuis , Marc R.M. Scheltinga and Rudi M.H. Roumen
Published/Copyright: October 1, 2017
Become an author with De Gruyter Brill

Abstract

Background and aims

Chronic abdominal pain may occasionally be due to terminal endings of intercostal nerves (ACNES, abdominal cutaneous nerve entrapment syndrome) that are entrapped in the abdominal wall. Spontaneous neuropathic flank pain may also be caused by involvement of branches of these intercostal nerves. Aim is to describe a series of patients with flank pain due to nerve entrapment and to increase awareness for an unknown condition coined Lateral Cutaneous Nerve Entrapment Syndrome (LACNES).

Methods

Patients possibly having LACNES (constant area of flank tenderness, small point of maximal pain with neuropathic characteristics, locoregional altered skin sensation) presenting between January 2007 and May 2016 received a diagnostic 5–10 mL 1% lidocaine injection. Pain levels were recorded using a numerical rating scale (0, no pain to 10, worst possible). A >50% pain reduction was defined as success. Long term effect of injections and alternative therapies were determined using a satisfaction scale (1, very satisfied, no pain - 5, pain worse).

Results

30 patients (21 women, median age 52, range 13-78) were diagnosed with LACNES. Pain following one injection dropped from 6.9 ± 1.4 to 2.4 ±1.9 (mean, p < 0.001) leading to an 83% immediate success rate. Repeated injection therapy was successful in 16 (pain free n = 7, pain acceptable, n = 9; median 42 months follow-up). The remaining 14 patients received (minimally invasive) surgery (n = 5) or other treatments (medication, manual therapy or pulsed radiofrequency, n = 9). Overall treatment satisfaction (scale 1 or 2) was attained in 79%.

Conclusions and implications

LACNES should be considered in patients with chronic flank pain. Injection therapy is long term effective in more than half of the population.

1 Introduction

Flank pain may be defined as a sensation of discomfort that is located in the area between the axilla and iliac bone. Patients with acute flank pain often suffer from an underlying visceral disease such as gallbladder stones or kidney disease. However, chronic flank pain in patients with normal blood and urine tests and normal imaging is a diagnostic challenge for general physicians and medical specialists.

The department of General Surgery/SolviMax has gained ample experience in the treatment of chronic abdominal pain or groin pain of various causes [1,2,3]. A large portion of these painful patients are found to suffer from a neuropathic pain syndrome of anterior portions of the abdominal wall due to an anterior cutaneous nerve entrapment syndrome (ACNES) [3,4,5,6]. It was our impression that a subset of patients who were referred for possible ACNES actually presented with a lateral variant of an ACNES-like pain entity. These individuals reported a neuropathic pain that mimicked ACNES, although the location was far more laterally located in the flank region. As far as we know, only two cases of “entrapped” lateral branches of intercostal thoracic nerves causing flank pain have previously been reported by others [7,8].

The abdominal wall including the flank region is sensory innervated by anterior and lateral cutaneous branches of 6 paired thoracic intercostal nerves (Th 7th-12th) [9,10]. Anterior cutaneous nerve entrapment syndrome (ACNES) is a condition in which terminal (abdominal) parts of these cutaneous intercostal nerve branches are traumatized or triggered by a hitherto unidentified event leading to a local abdominal pain syndrome presenting in the area ofthe rectus abdominal muscle [9]. SpecificACNES characteristics include a small area (several square centimetres) of maximum pain, altered skin sensation covering this tender point, a positive pinch test and a positive Carnett’s test (increased local tenderness by tensing the abdominal muscles) [2,11]. Recently, a novel variant of ACNES was coined Posterior Cutaneous Nerve Entrapment Syndrome (POCNES). This POCNES syndrome is associated with severe localized (lower) back pain, and it was found that posterior branches of the intercostal nerve were entrapped in the region of the thoracolumbar muscle group [12].

The lateral cutaneous branches of an intercostal nerve pass the external intercostal muscles and the serratus anterior muscle towards the flank area. While piercing through these muscles they divide into an anterior and posterior branch (Fig. 1). This piercing site is possibly a preferred location of entrapment of this part of the nerve leading to a typical neuropathic flank pain that we propose to coin lateral cutaneous nerve entrapment syndrome (LACNES). Aim of the present study is to describe a case series of patients who were diagnosed and treated for possible entrapment of lateral branches of intercostal nerves, seen at our institution over the past 10 years.

Fig. 1 
            Anatomy of intercostal cutaneous nerve branch originating from the spinal cord with posterior (PCB), lateral (LCB) and anterior branches (ACB). VB vertebrae, RAM rectus abdominis muscle, EOAM external oblique abdominal muscle, 1OAM internal oblique abdominis muscle, TAM transverse abdominal muscle.
Fig. 1

Anatomy of intercostal cutaneous nerve branch originating from the spinal cord with posterior (PCB), lateral (LCB) and anterior branches (ACB). VB vertebrae, RAM rectus abdominis muscle, EOAM external oblique abdominal muscle, 1OAM internal oblique abdominis muscle, TAM transverse abdominal muscle.

2 Methods

The present study retrospectively evaluated patients who were analyzed for chronic abdominal wall pain between January 2007 and May 2016 in the department of General Surgery/SolviMáx, Maxima Medical Centre, the Netherlands, a 631-bed teaching hospital. The ethics committee of our hospital approved the study protocol (N17.009).The present study follows guidelines ofthe declaration of Helsinki (version October 19th, 2013). This manuscript adheres to the applicable STROBE (Strengthening the Reporting of Observational Studies in Epidemiology) guidelines [13]. In addition to the present case series, one illustrative patient is reported in greater detail with the clinical picture, pathology findings and intraoperative images to further support the proposed hypothesis of an alleged ‘entrapment neuropathy’ leading to localized neuropathic flank pain.

2.1 Patient selection and inclusion criteria

Surgeons of our hospital have a special interest in patients with chronic abdominal wall pain and groin pain. Outpatient department activities are performed in a centre of expertise (SolviMáx). During

Fig. 1.

the study period, the majority of LACNES patients was identified from a large population of patients presenting to SolviMáx with abdominal pain of unknown origin by two senior authors using a standard history and physical examination [4]. A subset of LAC-NES patients was diagnosed by one of these senior surgeons at a regular surgical outpatient clinic, occasionally as a follow-up after abnormal visceral imaging.

Physical examination includes a series of standard tests. The patient is asked to indicate the location of maximum pain. A cotton swab and alcohol soaked gauze are used to evaluate skin gnostic and vital sensibility covering this tender point. Somatosensory disturbances such as hypoesthesia, hyperesthesia, allodynia or altered cool perception are commonly found as compared to the contralateral flank. Furthermore, pinching the skin is often extremely painful compared to the opposite non-involved side (a positive Pinch test).

Inclusion criteria for the diagnosis LACNES are based on an extensive (but entirely subjective) experience in patients with ACNES [2,4]. Patients were eligible for inclusion if at least 3 out of 4 of the following criteria were met:

  1. A >3 month history of locoregional flank pain.

  2. A constant area of tenderness located in the flank covering a fingertip small point of maximal pain in the midaxillary line (Fig. 2).

  3. A larger area of altered skin sensation such as hypoesthesia, hyperesthesia or altered cool perception covering this maximal pain point, but not necessarily corresponding to a specific complete dermatome.

  4. A positive Pinch test (using thumb and index finger to ‘pinch’ and lift the skin around the tender point eliciting a painful response in comparison to the contralateral side).

Fig. 2 
              A patient with LACNES. The point of maximal pain (swab) is located in the right midaxillary line that is covered by an area of altered skin sensation (////), hyperesthesia, altered cool perception and positive pinch test. (Photos with permission).
Fig. 2

A patient with LACNES. The point of maximal pain (swab) is located in the right midaxillary line that is covered by an area of altered skin sensation (////), hyperesthesia, altered cool perception and positive pinch test. (Photos with permission).

Normal laboratory testing and imaging (ultrasonography, computed tomography) contributed to consideration of the diagnosis LACNES, but was not required per se. Exclusion criteria were surgical scar-related pain syndromes, thoracolumbar disease or impaired communication.

If diagnostic criteria were satisfied, the presumptive diagnosis LACNES was communicated to the patient and specifics of a diagnostic injection procedure were explained. Following verbal consent, 5–10 mL of 1% lidocaine was administered as follows. The exact amount of administered anaesthetic agent was based on the presumed weight of patients and/or the estimated thickness of subcutis covering the tender point. The patient was asked to sit or stand with a raised ipsilateral arm. After skin disinfection, the point of maximal pain was marked with a pencil. A 21 G 40 mm needle was used to administer the lidocaine. Needle tip placement was done 1–2 cm below the fascia of the external oblique muscle or serratus muscle, in proximity to the tender point using a free hand technique (Fig. 3).

Fig. 3 
              Free hand tender point-infiltration using 1% lidocaine combined with 40 mg of methylprednisolone.
Fig. 3

Free hand tender point-infiltration using 1% lidocaine combined with 40 mg of methylprednisolone.

2.2 Data accrual and outcome measurements

Fig. 3.

Pain was measured on a numerical rating scale [NRS, 0 (no pain) to 10 (worst possible pain)] immediately before and some 10–15 minutes after the injection. If levels of pain were considerably lower (e.g. >50% pain reduction), characteristics of the diagnosis were again communicated, and the patient received a control appointment after 2–3 weeks. If the pain had recurred by then, a combination of 5 ml of 1% lidocaine and 40 mg of methyl-prednisolone was injected. If levels of pain after this regimen remained unacceptable or if patients declined ongoing injection therapy, they were either referred to a pain clinic for alternative treatments such as medication, physical therapy, manual therapy or pulsed radiofrequency therapy (PRF) or treatment of a visceral source (if present) was performed. A hospital electronic search using a diagnosis code exclusively assigned to abdominal wall related pain syndromes was performed for identification of potential patients. All records were consequently identified hand searched to identify individuals suspected of having a neuropathic pain syndrome in the flank. Specifics including age, gender, body mass index, diagnostic delay, aetiology and NRS scores (if present) at baseline and time of follow-up.

A final follow-up evaluation was performed by the first author (RM) inJuly and August 2016 by phone assessing long-term clinical success and level of satisfaction as previously published (verbal rating scale, VRS, Table 1) [4]. Clinical success was defined as VRS 1-2 ([very] satisfied), while a VRS of 3 was defined as an attenuation of pain levels. The therapy had failed if a VRS 4-5 was reported.

Table 1

Level of satisfaction after treatment for ACNES using Verbal Rating Scale [a](4).

1 I am very satisfied; I have no pain anymore
2 I am satisfied; I occasionally experience some pain
3 I have improved, but experience some pain on a regular basis
4 The treatment did not change my pain level
5 My pain has worsened after the treatment

2.3 Data analysis

All analyses were performed using the Statistical Package for the Social Sciences (SPSS) version 21 for Windows. Categorical variables were described as frequencies. Continuous data were tested for normality and are presented as means with standard deviation (±SD) or median values (range) as appropriate. Changes in pain scores after lidocaine infiltration were evaluated using paired T-testing. A p-value of <0.05 was considered significant.

3 Results

3.1 Baseline patient characteristics

Thirty patients evaluated in MMC’s Department of General Surgery and SolviMáx retrospectively fulfilled inclusion criteria of LACNES during the almost 10 year study period. Characteristics are listed in Table 2. There were 21 (70%) females and 9 (30%) males. Median age was 52 years (range, 13–78). Patients were referred after a median 18 months (range, 3–360), indicating the long duration of this pain entity. Flank pain was more often located on the right than on the left side (80%, 24/30 vs 20%, 6/30). A positive skin pinch test was detected in 90% (27/30). All 30 patients had undergone imaging (e.g. US, CT or MR1 scan) to exclude organic disease. 1nterestingly, underlying visceral abnormalities were demonstrated in nine (liver adenoma (n = 2), liver cyst (n = 3), a spontaneous/congenital intrahepatic porto-caval shunt (n = 1); splenic cyst (n = 1) and kidney cyst (n = 1) and multiple gallstones (n = 1)). An expectative policy was used in the patients with liver cysts, porto-caval shunt and gallstones.

Table 2

Characteristics of patients with lateral cutaneous nerve entrapment syndrome (LACNES).

LACNES (n = 30)
Age (range) 52 (13-78)
Gender, F:M ratio 21:9
Body mass index, kg/m2 (SD) 23.7 (5.1)
Diagnostic delay, months (range) 18 (3-360)
Aetiology (n)
 Spontaneous 21
 Previous abdominal surgery 8
 Sports 1
Local sensory dysfunction covering pain point 24
Pain level at presentation (NRS, 0-10) 6.9 (1.4)
  1. Data are presented as mean with standard deviation (±SD), or median values with ranges. NRS, Numerical Rating Scale

3.2 Diagnostic and therapeutic injections for LACNES

Interventions for LACNES are depicted in Table 3. All patients consented to a diagnostic maximal pain point lidocaine infiltration. In these 30 patients, mean pre- and post-NRS scores were found to drop from 6.9 ±1.4 to 2.4 ±1.9 (p <0.001). Twenty five (83%) reported a >50% pain reduction following the diagnostic injection (Fig. 4).

Table 3

Characteristics and Success ofInterventions in LACNES patients (n = 30).

Patient characteristics Dx Injection Treatment Follow-up
Pt Age Location Pain area in corresponding dermatome NRS pre NRS post Number of injections MT PS Treatment visceral source Neurectomy Success IT[a] Final VRS[b]
1 72 Right Th10 - - 1 Yes 1
2 57 Right Th10 7 0 1 Yes 1
3 50 Right Th9 9 4 4 Yes 1
4 51 Right Th9 4 0 3 Yes 2
5 42 Right Th8 4 0 1 X Yes 1
6 13 Left Th9 - - 1 X Yes 2
7 66 Right Th9 5 0 3 X No 2
8 50 Right Th11 6 2 1 X No 2
9 46 Right Th11 - - 1 Yes 1
10 52 Left Th10 8 4 1 X No 2
11 32 Right Th9 8 3 2 Yes 1
12 71 Right Th7 - - 3 Yes 2
13 78 Right Th11 5 3 3 Yes 2
14 55 Right Th12 7 3 4 X No 1
15 26 Left Th7 8 2 1 X No 1
16 71 Right Th9 8 0 3 Yes 2
17 58 Right Th8 7 3 2 X Yes 1
18 48 Right Th9 6 5 2 No 2
19 59 Right Th9 9 4 5 Yes 2
20 48 Left Th8 8 0 1 Yes 1
21 54 Right Th9 7 3 1 Yes 2
22 64 Right Th11 6 2 2 Yes 2
23 23 Right Th7 8 3 2 X No 2
24 47 Left Th11 - - 1 X No 4
25 55 Left Th7 6 0 4 X No 3
26 52 Right Th10 7 5 1 X No 4
27 52 Right Th8 8 6 2 X No 4
28 68 Right Th11 8 4 2 X No 4
29 30 Right Th11 - - 1 X No 4
30[c] 61 Right Th8 - - 1 X No [c]
  1. Dx diagnostic, IT injection therapy, MT manual therapy, PS pain specialist, NRS Numerical Rating Scale pre/post injection. - Missing data

Fig. 4 
              Pain reduction before and after a diagnostic injection in LACNES.
Fig. 4

Pain reduction before and after a diagnostic injection in LACNES.

After this single diagnostic injection, 5 of the injected 30 patients (17%) were pain-free in the short term (median one month) as well as in the long term (median 60 months, range 2–103). Two additional patients were long term pain-free after 1–3 repeated injections (respectively 36 and 55 months FU). Thus, 7 of 30 (23%) were long term pain free by an injection regimen only (VRS = 1). Another nine patients reported substantial pain relief in the short term and opted for incidentally repeated injections, if required. Pain levels in this subset were acceptable in the long term (median 24 months, range 2–52; VRS = 2). As a consequence, the overall long term injection therapy success rate was 53% (16/30). Two patients received manual therapy simultaneously with injection therapy but they attributed pain relief to injections rather than to manual therapy. One patient who had short-term success with injection therapy experienced a recurrence of pain and was referred to a manual therapist leading to attenuated pain levels (VRS 3).

3.3 Alternative therapies for LACNES

Of the fourteen patients who did not successfully respond to injection therapy, six were referred to a pain service for medication or pulsed radiofrequency therapy (PRF) which led to acceptable pain levels in two (VRS 2). The other four still had residual pain at the final evaluation. Three other patients were referred to a manual therapist, with one achieving acceptable results (VRS 2).

Surgery was performed in the 5 remaining patients. Drainage of a spleen cyst (n = 1) and kidney cyst (n = 1) as well as embolization of a liver adenoma (n = 1) cured the neuropathic flank pain in all three. The fourth patient was referred to an academic hospital for embolization of liver adenoma but the pain remained (VRS 4). Therefore, visceral treatment had a 75% success rate (3/4; VRS –-2). One patient receiving a neurectomy is described in more detail as follows.

3.4 Case report

A 23-year old female presented with an 18 month history of flank pain located at the right mid-axillary region. Her complaints had presented spontaneously and were slowly progressive leading to a continuous stabbing pain that was provoked by physical effort. Diagnostics and treatments by an orthopaedic surgeon and a neurologist were to no avail. She also received TENS treatment, PRF treatment and cryoneuro ablation by a pain specialist but also without relief. Analysis by this pain specialist showed a DN4 (Douleur Neuropathique 4) score of 5, suggesting a neuropathic character of her pain symptoms. Physical examination revealed a constant area of hypoesthesia of approximately 5 × 5 cm overlying a small point of maximum pain located at the anterior axillary line at the level of the right seventh rib. Two Ultrasound guided infiltrations using local anaesthetics had a beneficial albeit temporary response. Following an extensive consultation, a local surgical exploration was proposed and consented. Prior to operation, the area of interest was marked onto the skin. Once general anaesthesia was administered, the sheath of muscle or serratus anterior was exposed via a transverse 7-cm skin incision. The neurovascular bundle was identified. The fascia was widened and this bundle and its branches were tracked down until it reached the caudal border of the rib edge and was consequently coagulated and removed (Fig. 5). Accompanying vascular structures were ligated or also coagulated. The sheath and the remainder of the wound were closed in layers using absorbable suturing material. Pathological analysis revealed normal nerve tissue. The patient was pain free at the 6 weeks postoperative outpatient control and remained up to the last control (6 months follow up).

Fig. 5 
              Intraoperative view of a neurovascular bundle (loop) and a nearby branch (loop) perforating the fascia of the serratus muscle. Point of view: inframammary on the right side of the supine patient. Black arrow pointing caudally showing the irradiation route of pain that was described by the patient.
Fig. 5

Intraoperative view of a neurovascular bundle (loop) and a nearby branch (loop) perforating the fascia of the serratus muscle. Point of view: inframammary on the right side of the supine patient. Black arrow pointing caudally showing the irradiation route of pain that was described by the patient.

3.5 Long term treatment satisfaction

A 100% response rate was attained after a median 40 months follow-up in the surviving 29 patients (range 2-103). One patient died of unrelated cause. More than three quarters (79%, 23 of 29) were satisfied with the therapeutic result (VRS 1-2). One patient (4%) reported improvement but regularly experienced pain (VRS 3). Therapy was unsuccessful in the five remaining patients (17%, VRS 4). No VRS 5 was scored.

4 Discussion

The present retrospective case series found that a small portion of patients who were referred to a tertiary referral centre for evaluation of a chronic abdominal pain syndrome were found to suffer from (possible) neuropathic pain in the flank. These patients demonstrated pain characteristics resembling ACNES although the point of maximal pain was situated far more laterally [4]. Aim of the present study was to describe a series of these patients who were diagnosed and treated for a possible entrapment of intercostal flank nerves. The most important finding is that this novel syndrome should be considered in the differential diagnosis of chronic flank pain. Injection therapy using a combination of local anaesthetic agents with or without steroids appeared long term effective in a little over half (53%) of the population. We propose to coin this syndrome lateral cutaneous nerve entrapment syndrome (LACNES).

It may well be that the set of symptoms of the present cases who are labelled as LACNES was previously described by others using different terminology. In the available literature, one case report was found describing a lateral cutaneous nerve entrapment syndrome [8]. Another case report described a case of ACNES although the pain actually was located more laterally in the flank [7]. More recently, a posterior version of ACNES was identified suggesting involvement of posterior branches of cutaneous intercostal nerves at the lower back [12]. The present study is the first case series reporting on specific features of entrapment of lateral branches of intercostal nerves that also discusses diagnostic and treatment protocols.

If one proposes a novel syndrome, a clear set of criteria is needed. All patients demonstrated a clinical picture that was characterized by a constant site of lateral abdominal tenderness located in the flank (mid axillary region between the costal arc and the iliac crest) with a fingertip small tender point (Fig. 2). Moreover, most (90%) had a positive pinch test defined as a disproportionally intense pain following skin pinching using thumb and index finger compared to the opposite flank. Similar to ACNES, the pain is accompanied by skin somatosensory disturbances (hypoesthesia, hyperesthesia, hyperalgesia and altered cool perception) that is limited to a discrete area in the flank, indicating some kind of local nerve involvement. 1n each future patient fulfilling these criteria, the presumptive diagnosis of LACNES should be considered and a diagnostic injection should be offered.

Abdominal cutaneous nerve entrapment syndrome is considered as an “entrapment” neuropathy of cutaneous branches of the 7-12th intercostal nerves [9]. Neuropathic pain has been defined by the International Association for the Study of Pain (1ASP) as pain caused by a (demonstrable) lesion or disease of the somatosensory nervous system [14]. A local neurectomy of the lateral cutaneous nerve branch in one patient resulted in complete and long lasting pain relief. A tissue analysis demonstrated normal nervous tissue. This finding supports the hypothesis that entrapment of cutaneous branches of intercostal nerves may possibly lead to a severe neuropathic flank pain entity.

It is remarkable that treatment of a visceral entity cured the neuropathic flank pain in 3 of 4 LACNES patients having parenchymal disease including liver, kidney and spleen. 1t is thought that a segmental relation between a visceral abnormality and the abdominal wall may explain somatosensory abnormalities and neuropathic pain of the latter. Such connections were described already more than one century ago by Head (“Head zones”) and MacKenzie [15,16]. MacKenzie proposed the term ‘referred pain’ that was defined as pain originating from internal organs that is projected onto a predictable skin area. Relevant to this discussion is an often cited theory that hypothesizes the presence of viscerocutaneous reflexes. Visceral afferent nerve fibres converge with cutaneous pain afferents at the level of the dorsal root ganglion and spinal cord [15,16,17]. Higher brain centres are possibly not able to distinguish neuropathic abdominal wall pain from visceral pain. One of the senior authors (RR) is also a liver surgeon skilled in the treatment of a range of liver pathologies. His subspecialty has likely contributed to the identification of neuropathic pain symptoms located in the right upper midaxillary line. The somewhat peculiar combination of visceral abnormalities and coinciding neuropathic pain strongly supports the presence of these segmental relations that modern-day clinical medicine, with its focus on high tech imaging, tends to ignore.

Most patients in our study were referred after a median of more than one and a half year diagnostic delay suggesting that this lateral variant is also frequently overlooked as a cause of chronic flank pain as also has been observed in other types of chronic abdominal wall pain [18,19,20]. However, there exist some differences between the abdominal myofascial pain syndrome (AMPS) and ACNES [18,21,22,23]. Contrary to ACNES, skin pinching in AMPS is usually normal, as is the local skin sensibility covering the painful area. Moreover, gender distribution is almost equal (females: males = 54:46) [22]. It is thus unlikely that the beneficial effect of the local injections can be seen as placebo effect. It is hoped that the present study contributes to an earlier recognition of patients with LACNES.

This study obviously harbours flaws including its retrospective character. 1t describes a patient series with a relatively small volume that is collected over a 10 years period by a selected group of clinicians. Referral bias is present due to the nature of our institution. Different types of treatment were proposed due to lack of knowledge in the early years of this case series. A standardized treatment regimen should prospectively be evaluated in future studies of flank pain. Furthermore, the diagnosis LAC-NES was not demonstrated by objective tests that are associated with somatosensory disturbances including laser evoked potentials, nerve biopsies (except the single operative case), Quantitative Sensory Testing or MR1 imaging [24]. We also did not standardly use specific questionnaires suggesting neuropathic pain, like DN-4, LANSS, Pain DETECT, or the Neuropathic Pain Syndrome Inventory [24]. Nevertheless we feel confident that the patients presented in this series were having neuropathic pain due to affected branches of intercostal nerves as also reported in similar syndromes such as ACNES and POCNES [4,12]. We are currently considering wider deployment of performing a neurectomy of the affected nerve as was done in the presented case, and as has previously been described in patients with recalcitrant ACNES [2,25].

In conclusion, lateral cutaneous nerve entrapment (LACNES) should be considered in the differential diagnosis of chronic flank pain. 1njection therapy using a combination of local anaesthetic agents with or without steroids is long term effective in over half of this population. Further research is needed to establish a standardized treatment protocol and investigate the effect of a local surgical exploration in treating LACNES patients.

Highlights

  • A common cause for chronic abdominal wall pain is the Anterior Cutaneous Nerve Entrapment Syndrome (ACNES).

  • Thoracic intercostal nerves divide in 3 cutaneous end parts: the anterior, lateral and posterior branches.

  • Spontaneous neuropathic flank pain can be due to entrapment of lateral branches of thoracic intercostal nerves.

  • This condition is coined Lateral Cutaneous Nerve Entrapment Syndrome (LACNES).


DOI of refers to article: http://dx.doi.org/10.1016/j.sjpain.2017.10.006.



Department of Surgery, Máxima Medical Center, P.O. Box 7777, 5500 MB Veldhoven, The Netherlands

  1. Author contributions: RM acquired and analysed the used data and drafted the manuscript. NB participated in analysing used data and critically revised the manuscript. MS participated in the design of the study and critically revised the manuscript. RR participated in the design of the study and critically revised the manuscript. All authors discussed the results and commented on the manuscript.

  2. Funding sources: None declared.

  3. Ethical issues: The ethics committee of our hospital approved the study protocol (N17.009). The present study follows guidelines of the declaration of Helsinki (version October 19th, 2013).

  4. Conflicts of interest: None declared.

References

[1] Boelens OB, Scheltinga MR, Houterman S, Roumen RM. Randomized clinical trial of trigger point infiltration with lidocaine to diagnose anterior cutaneous nerve entrapment syndrome. Br J Surg 2013;100:217–21.Search in Google Scholar

[2] Boelens OB, van Assen T, Houterman S, Scheltinga MR, Roumen RM. A doubleblind, randomized, controlled trial on surgery for chronic abdominal pain due to anteriorcutaneous nerve entrapment syndrome. AnnSurg 2013;257:845–9.Search in Google Scholar

[3] Loos MJ, Scheltinga MR, Roumen RM. Surgical management ofinguinal neuralgia afteralow transverse Pfannenstiel incision. Ann Surg 2008;248:880–5.Search in Google Scholar

[4] Boelens OB, Scheltinga MR, Houterman S, Roumen RM. Managementofanterior cutaneous nerve entrapment syndrome in a cohort of 139 patients. Ann Surg 2011;254:1054–8.Search in Google Scholar

[5] Loos MJ, Scheltinga MR, Roumen RM. Tailored neurectomy for treatment of postherniorrhaphy inguinal neuralgia. Surgery 2010;147:275–81.Search in Google Scholar

[6] Roumen RM, Scheltinga MR. Abdominal intercostal neuralgia: a forgottencause ofabdominal pain. Ned TijdschrGeneeskd 2006;150:1909–15.Search in Google Scholar

[7] Peleg R, Gohar J, Koretz M, Peleg A. Abdominal wall pain in pregnant women caused by thoracic lateral cutaneous nerve entrapment. Eur J Obstet Gynecol Reprod Biol 1997;74:169–71.Search in Google Scholar

[8] Sharf M, Shvartzman P, Farkash E, Horvitz J. Thoracic lateral cutaneous nerve entrapment syndrome without previous lowerabdominal surgery. J Fam Pract 1990;30:211–2, 214.Search in Google Scholar

[9] Applegate WV, Buckwalter NR. Microanatomy of the structures contributing to abdominal cutaneous nerve entrapment syndrome. J Am Board Fam Pract 1997;10:329–32.Search in Google Scholar

[10] Carnett J. Intercostal neuralgia as a cause of abdominal pain and tenderness. Surg Gynecol Obstet 1926;42.Search in Google Scholar

[11] Applegate W. Abdominal Cutaneous Nerve Entrapment Syndrome (ACNES): a commonly overlooked cause ofabdominal pain. Perm J 2002;6:20–7.Search in Google Scholar

[12] Boelens OB, Maatman RC, Scheltinga MR, van Laarhoven K, Roumen RM. Chronic localized back pain due to Posterior Cutaneous Nerve Entrapment Syndrome (POCNES): a new diagnosis. Pain Physician 2017;20:E455–8.Search in Google Scholar

[13] von Elm E, Altman DG, Egger M, Pocock SJ, Gotzsche PC, Vandenbroucke JP, Initiative S. The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) statement: guidelines for reporting observational studies. J Clin Epidemiol 2008;61:344–9.Search in Google Scholar

[14] Haanpaa M, Attal N, Backonja M, Baron R, Bennett M, Bouhassira D, Cruccu G, Hansson P, Haythornthwaite JA, Iannetti GD, Jensen TS, Kauppila T, Nurmikko TJ, Rice AS, Rowbotham M, Serra J, Sommer C, Smith BH, Treede RD. NeuPSIG guidelines on neuropathic pain assessment. Pain 2011;152: 14-27.Search in Google Scholar

[15] Head H. On disturbances of senation with especial reference to the pain of visceral disease, Part I. Brain: J Neurol 1893;16:1–133.Search in Google Scholar

[16] Mackenzie J. Contribution to the study of sensory symptoms associated with visceral disease. Med Chronicle 1892;XVI:293–322.Search in Google Scholar

[17] Hansen K, Schliack H. Segmentale Innervation; ihre Bedeutung fur Klinik und Praxis: Thieme; 1962.Search in Google Scholar

[18] Cimen A, Celik M, Erdine S. Myofascial pain syndrome in the differential diagnosis ofchronic abdominal pain. Agri: Agri 2004;16:45–7.Search in Google Scholar

[19] Lindsetmo RO, Stulberg J. Chronic abdominalwall pain-adiagnosticchallenge forthe surgeon. AmJSurg 2009;198:129–34.Search in Google Scholar

[20] Srinivasan R, Greenbaum DS. Chronic abdominal wall pain: a frequently overlooked problem. Practical approach to diagnosis and management.Am J Gastroenterol 2002;97:824–30.Search in Google Scholar

[21] Facco E, Ceccherelli F. Myofascial pain mimicking radicular syndromes. Acta Neurochir Suppl 2005;92:147–50.Search in Google Scholar

[22] Montenegro ML, Gomide LB, Mateus-Vasconcelos EL, Rosae-Silva JC, Candido-dosReis FJ, Nogueira AA, Poli-Neto OB. Abdominal myofascial pain syndrome must be considered in the differential diagnosis of chronic pelvic pain. Eur J Obstet Gynecol Reprod Biol 2009;147:21–4.Search in Google Scholar

[23] Nazareno J, Ponich T, Gregor J. Long-term follow-up of trigger point injections for abdominal wall pain. Can J Gastroenterol=J Can Gastroenterol 2005;19:561–5.Search in Google Scholar

[24] Graven-Nielsen T, Arendt-Nielsen L. Assessment of mechanisms in localized and widespread musculoskeletal pain. Nat Rev Rheumatol 2010;6: 599-606.Search in Google Scholar

[25] van Assen T, Boelens OB, van Eerten PV, Scheltinga MR, Roumen RM. Surgical options after a failed neurectomy in anterior cutaneous nerve entrapment syndrome. World J Surg 2014;38:3105–11.Search in Google Scholar

Received: 2017-10-03
Accepted: 2017-10-04
Published Online: 2017-10-01
Published in Print: 2017-10-01

© 2017 Scandinavian Association for the Study of Pain

Articles in the same Issue

  1. Observational study
  2. Perceived sleep deficit is a strong predictor of RLS in multisite pain – A population based study in middle aged females
  3. Clinical pain research
  4. Prospective, double blind, randomized, controlled trial comparing vapocoolant spray versus placebo spray in adults undergoing intravenous cannulation
  5. Clinical pain research
  6. The Functional Barometer — An analysis of a self-assessment questionnaire with ICF-coding regarding functional/activity limitations and quality of life due to pain — Differences in age gender and origin of pain
  7. Clinical pain research
  8. Clinical outcome following anterior arthrodesis in patients with presumed sacroiliac joint pain
  9. Observational study
  10. Chronic disruptive pain in emerging adults with and without chronic health conditions and the moderating role of psychiatric disorders: Evidence from a population-based cross-sectional survey in Canada
  11. Educational case report
  12. Management of patients with pain and severe side effects while on intrathecal morphine therapy: A case study
  13. Clinical pain research
  14. Behavioral inhibition, maladaptive pain cognitions, and function in patients with chronic pain
  15. Observational study
  16. Comparison of patients diagnosed with “complex pain” and “somatoform pain”
  17. Original experimental
  18. Patient perspectives on wait times and the impact on their life: A waiting room survey in a chronic pain clinic
  19. Topical review
  20. New evidence for a pain personality? A critical review of the last 120 years of pain and personality
  21. Clinical pain research
  22. A multi-facet pain survey of psychosocial complaints among patients with long-standing non-malignant pain
  23. Clinical pain research
  24. Pain patients’ experiences of validation and invalidation from physicians before and after multimodal pain rehabilitation: Associations with pain, negative affectivity, and treatment outcome
  25. Observational study
  26. Long-term treatment in chronic noncancer pain: Results of an observational study comparing opioid and nonopioid therapy
  27. Clinical pain research
  28. COMBAT study – Computer based assessment and treatment – A clinical trial evaluating impact of a computerized clinical decision support tool on pain in cancer patients
  29. Original experimental
  30. Quantitative sensory tests fairly reflect immediate effects of oxycodone in chronic low-back pain
  31. Editorial comment
  32. Spatial summation of pain and its meaning to patients
  33. Original experimental
  34. Effects of validating communication on recall during a pain-task in healthy participants
  35. Original experimental
  36. Comparison of spatial summation properties at different body sites
  37. Editorial comment
  38. Behavioural inhibition in the context of pain: Measurement and conceptual issues
  39. Clinical pain research
  40. A randomized study to evaluate the analgesic efficacy of a single dose of the TRPV1 antagonist mavatrep in patients with osteoarthritis
  41. Editorial comment
  42. Quantitative sensory tests (QST) are promising tests for clinical relevance of anti–nociceptive effects of new analgesic treatments
  43. Educational case report
  44. Pregabalin as adjunct in a multimodal pain therapy after traumatic foot amputation — A case report of a 4-year-old girl
  45. Editorial comment
  46. Severe side effects from intrathecal morphine for chronic pain after repeated failed spinal operations
  47. Editorial comment
  48. Opioids in chronic pain – Primum non nocere
  49. Editorial comment
  50. Finally a promising analgesic signal in a long-awaited new class of drugs: TRPV1 antagonist mavatrep in patients with osteoarthritis (OA)
  51. Observational study
  52. The relationship between chronic musculoskeletal pain, anxiety and mindfulness: Adjustments to the Fear-Avoidance Model of Chronic Pain
  53. Clinical pain research
  54. Opioid tapering in patients with prescription opioid use disorder: A retrospective study
  55. Editorial comment
  56. Sleep, widespread pain and restless legs — What is the connection?
  57. Editorial comment
  58. Broadening the fear-avoidance model of chronic pain?
  59. Observational study
  60. Identifying characteristics of the most severely impaired chronic pain patients treated at a specialized inpatient pain clinic
  61. Editorial comment
  62. The burden of central anticholinergic drugs increases pain and cognitive dysfunction. More knowledge about drug-interactions needed
  63. Editorial comment
  64. A case-history illustrates importance of knowledge of drug-interactions when pain-patients are prescribed non-pain drugs for co-morbidities
  65. Editorial comment
  66. Why can multimodal, multidisciplinary pain clinics not help all chronic pain patients?
  67. Topical review
  68. Individual variability in clinical effect and tolerability of opioid analgesics – Importance of drug interactions and pharmacogenetics
  69. Editorial comment
  70. A new treatable chronic pain diagnosis? Flank pain caused by entrapment of posterior cutaneous branch of intercostal nerves, lateral ACNES coined LACNES
  71. Clinical pain research
  72. PhKv a toxin isolated from the spider venom induces antinociception by inhibition of cholinesterase activating cholinergic system
  73. Clinical pain research
  74. Lateral Cutaneous Nerve Entrapment Syndrome (LACNES): A previously unrecognized cause of intractable flank pain
  75. Editorial comment
  76. Towards a structured examination of contextual flexibility in persistent pain
  77. Clinical pain research
  78. Context sensitive regulation of pain and emotion: Development and initial validation of a scale for context insensitive avoidance
  79. Editorial comment
  80. Is the search for a “pain personality” of added value to the Fear-Avoidance-Model (FAM) of chronic pain?
  81. Editorial comment
  82. Importance for patients of feeling accepted and understood by physicians before and after multimodal pain rehabilitation
  83. Editorial comment
  84. A glimpse into a neglected population – Emerging adults
  85. Observational study
  86. Assessment and treatment at a pain clinic: A one-year follow-up of patients with chronic pain
  87. Clinical pain research
  88. Randomized, double-blind, placebo-controlled, dose-escalation study: Investigation of the safety, pharmacokinetics, and antihyperalgesic activity of L-4-chlorokynurenine in healthy volunteers
  89. Clinical pain research
  90. Prevalence and characteristics of chronic pain: Experience of Niger
  91. Observational study
  92. The use of rapid onset fentanyl in children and young people for breakthrough cancer pain
  93. Original experimental
  94. Acid-induced experimental muscle pain and hyperalgesia with single and repeated infusion in human forearm
  95. Original experimental
  96. Swearing as a response to pain: A cross-cultural comparison of British and Japanese participants
  97. Clinical pain research
  98. The cognitive impact of chronic low back pain: Positive effect of multidisciplinary pain therapy
  99. Clinical pain research
  100. Central sensitization associated with low fetal hemoglobin levels in adults with sickle cell anemia
  101. Topical review
  102. Targeting cytokines for treatment of neuropathic pain
  103. Original experimental
  104. What constitutes back pain flare? A cross sectional survey of individuals with low back pain
  105. Original experimental
  106. Coping with pain in intimate situations: Applying the avoidance-endurance model to women with vulvovaginal pain
  107. Clinical pain research
  108. Chronic low back pain and the transdiagnostic process: How do cognitive and emotional dysregulations contribute to the intensity of risk factors and pain?
  109. Original experimental
  110. The impact of the Standard American Diet in rats: Effects on behavior, physiology and recovery from inflammatory injury
  111. Educational case report
  112. Erector spinae plane (ESP) block in the management of post thoracotomy pain syndrome: A case series
  113. Original experimental
  114. Hyperbaric oxygenation alleviates chronic constriction injury (CCI)-induced neuropathic pain and inhibits GABAergic neuron apoptosis in the spinal cord
  115. Observational study
  116. Predictors of chronic neuropathic pain after scoliosis surgery in children
  117. Clinical pain research
  118. Hospitalization due to acute exacerbation of chronic pain: An intervention study in a university hospital
  119. Clinical pain research
  120. A novel miniature, wireless neurostimulator in the management of chronic craniofacial pain: Preliminary results from a prospective pilot study
  121. Clinical pain research
  122. Implicit evaluations and physiological threat responses in people with persistent low back pain and fear of bending
  123. Original experimental
  124. Unpredictable pain timings lead to greater pain when people are highly intolerant of uncertainty
  125. Original experimental
  126. Initial validation of the exercise chronic pain acceptance questionnaire
  127. Clinical pain research
  128. Exploring patient experiences of a pain management centre: A qualitative study
  129. Clinical pain research
  130. Narratives of life with long-term low back pain: A follow up interview study
  131. Observational study
  132. Pain catastrophizing, perceived injustice, and pain intensity impair life satisfaction through differential patterns of physical and psychological disruption
  133. Clinical pain research
  134. Chronic pain disrupts ability to work by interfering with social function: A cross-sectional study
  135. Original experimental
  136. Evaluation of external vibratory stimulation as a treatment for chronic scrotal pain in adult men: A single center open label pilot study
  137. Observational study
  138. Impact of analgesics on executive function and memory in the Alzheimer’s Disease Neuroimaging Initiative Database
  139. Clinical pain research
  140. Visualization of painful inflammation in patients with pain after traumatic ankle sprain using [11C]-D-deprenyl PET/CT
  141. Original experimental
  142. Developing a model for measuring fear of pain in Norwegian samples: The Fear of Pain Questionnaire Norway
  143. Topical review
  144. Psychoneuroimmunological approach to gastrointestinal related pain
  145. Letter to the Editor
  146. Do we need an updated definition of pain?
  147. Narrative review
  148. Is acetaminophen safe in pregnancy?
  149. Book Review
  150. Physical Diagnosis of Pain
  151. Book Review
  152. Advances in Anesthesia
  153. Book Review
  154. Atlas of Pain Management Injection Techniques
  155. Book Review
  156. Sedation: A Guide to Patient Management
  157. Book Review
  158. Basics of Anesthesia
Downloaded on 8.9.2025 from https://www.degruyterbrill.com/document/doi/10.1016/j.sjpain.2017.10.007/html
Scroll to top button