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Server, V. Sánchez, Á. Mesas, J. Medel" "autores" => array:4 [ 0 => array:4 [ "nombre" => "A." "apellidos" => "Server" "email" => array:1 [ 0 => "anna.server@vallhebron.cat" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "V." "apellidos" => "Sánchez" ] 2 => array:2 [ "nombre" => "Á." "apellidos" => "Mesas" ] 3 => array:2 [ "nombre" => "J." "apellidos" => "Medel" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Anestesiología, Hospital Universitari Vall d’Hebron, Barcelona, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Técnica de radiofrecuencia pulsada en síndrome de ACNES invalidante en una paciente pediátrica" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1279 "Ancho" => 1675 "Tamanyo" => 215043 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0040" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Ultrasound-guided in-plane puncture.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Chronic abdominal pain (CAD) is highly prevalent (10%–19%) in the paediatric population.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> The aetiology of CAD can be classified as visceral (due to abdominal organ pathology), functional (evaluated by the Rome IV criteria) or parietal (originating in the abdominal wall).<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">ACNES (anterior cutaneous nerve entrapment syndrome) is a type of chronic abdominal wall pain that is frequently under-diagnosed,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3–5</span></a> and is responsible for 1 in every 8 cases of CAD among adolescents.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">ACNES occurs when the terminal branches of the intercostal nerves become trapped, possible due to mechanical compression or traction, although the specific pathophysiological mechanism remains unknown.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">The differential diagnosis of ACNES is very broad, and other causes of CAD must first be ruled out. In most cases, a detailed clinical evaluation must be made and complementary tests performed to rule out abdominal visceral pathology.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,6</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">Patients diagnosed with ACNES present intense, unilateral abdominal pain in a circumscribed area of less than 2<span class="elsevierStyleHsp" style=""></span>cm². Patients often present a positive Pinch Test or Carnett’s sign.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,4,7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">The therapeutic approach should be gradual, reserving the most invasive techniques for patients with refractory ACNES. Many different treatments are available, ranging from analgesics (which are largely ineffective due to the mechanical origin of the syndrome), local anaesthetic and/or corticosteroid infiltration, transcutaneous electrical nerve stimulation (TENS), infiltration of botulinum toxin, pulsed radiofrequency (PRF) and even neurectomy.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The success rate of these techniques is estimated to be 85% in adults, but few data are available from the paediatric population.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0035" class="elsevierStylePara elsevierViewall">We report the case of an 11-year-old girl, a high-level artistic gymnast, weighing 35<span class="elsevierStyleHsp" style=""></span>kg, with no pathological history of interest, who was referred to the Pain Unit in March 2021 by her paediatric surgeon due to abdominal pain.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The patient had a 6-month history of continuous, localized, stabbing abdominal pain in an area measuring 2<span class="elsevierStyleHsp" style=""></span>cm in diameter in the lower right hemiabdomen (about 3<span class="elsevierStyleHsp" style=""></span>cm medial to the anterior superior iliac crest). The pain had gradually increased to the point of becoming disabling, it was interfering with her sleep and had prevented her from going to school for 3 months.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Her family history was significant for the divorce of her parents, for which she had been receiving psychological therapy for 1<span class="elsevierStyleHsp" style=""></span>year. She had also had to change to a new school 1 month before the start of her symptoms.</p><p id="par0050" class="elsevierStylePara elsevierViewall">The case was initially diagnosed as non-specific abdominal pain. Ultrasound examination and serial lab tests were unremarkable, and an exploratory laparoscopy performed due to increased pain showed organic pathology.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Due to persistent pain that required multiple visits to the Emergency Department, and given the suspicion of inflammatory bowel syndrome, the patient was put on a dairy- and gluten-free diet, but her condition did not improve.</p><p id="par0060" class="elsevierStylePara elsevierViewall">Abdominal nuclear magnetic resonance imaging ruled out pathology and only revealed a slight oedema in the bilateral obturator externus and quadratus femoris muscles, probably related to mild bilateral ischiofemoral impingement.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The patient began physiotherapy in a private centre and received a series of treatments, all of which were ineffective; however, she responded very positively to local anaesthetic infiltration at the site of the pain, reporting over 50% pain relief lasting 1 week. TENS applied to the abdominal wall was partially effective, but of limited duration.</p><p id="par0070" class="elsevierStylePara elsevierViewall">When her history was taken other gastrointestinal symptoms were ruled out, and during the physical examination she showed altered gait due to the inability to fully support the right leg, adopting an antalgic hip gait and walking on tiptoes. On abdominal palpation, she reported local hyperalgesia, a highly positive Carnett’s sign (increased pain when tensing the abdominal muscles), and a positive Pinch Test (clamping the abdominal fat at the site of pain causes hyperalgesia). The intensity of the pain measured on the Visual Numerical Scale (VNS) was 6, with frequent daily exacerbations of up to 8 points.</p><p id="par0075" class="elsevierStylePara elsevierViewall">She had tried various pharmacological therapies, including analgesics, anti-inflammatories, neuromodulators, and muscle relaxants, all of which were unsuccessful, and she was currently receiving Bach flower therapy and magnesium.</p><p id="par0080" class="elsevierStylePara elsevierViewall">Given the diagnostic suspicion of ACNES based on her good response to local infiltration, and following a multidisciplinary evaluation involving a paediatric surgeon, a specialist in rehabilitation, and an anaesthesiologist from the Pain Unit, the patient was scheduled for ultrasound-guided infiltration in our Day Hospital.</p><p id="par0085" class="elsevierStylePara elsevierViewall">Since the paediatric psychologist had already diagnosed the patient with post-traumatic stress caused by the interventional procedures performed up to that moment, we decided to proceed directly with pulsed radiofrequency in order to avoid duplicating procedures and having to perform sequential sedation.</p><p id="par0090" class="elsevierStylePara elsevierViewall">The technique was performed with the patient in the supine position, under standard monitoring (SpO<span class="elsevierStyleInf">2</span>, BP, and HR), nasal prong oxygen, and intravenous sedation with propofol 180<span class="elsevierStyleHsp" style=""></span>mg (titrated doses).</p><p id="par0095" class="elsevierStylePara elsevierViewall">After applying lidocaine and prilocaine cream to the site of pain (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>), the right anterior rectus abdominis was identified on ultrasound using a Sonosite M-Turbo unit with a 13−6<span class="elsevierStyleHsp" style=""></span>MHz linear transducer. An ultrasound-guided in-plane puncture was performed using a 6<span class="elsevierStyleHsp" style=""></span>cm 23<span class="elsevierStyleHsp" style=""></span>G needle and a 5<span class="elsevierStyleHsp" style=""></span>mm active tip (Cosman electrodeCR™), placing the tip between the anterior and posterior fascia of the muscle (<a class="elsevierStyleCrossRefs" href="#fig0010">Figs. 2 and 3</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0100" class="elsevierStylePara elsevierViewall">Then, the patient's level of sedation was reduced, the needle was connected to the PRF generator (Cosman G4), verifying correct electrical impedance (256 Ω), and sensory stimulation was performed at 50<span class="elsevierStyleHsp" style=""></span>Hz, achieving a positive response at 0.4<span class="elsevierStyleHsp" style=""></span>V. This was followed by pulsed radiofrequency treatment of the anterior abdominal cutaneous nerve at 45<span class="elsevierStyleHsp" style=""></span>V, 42° for 6<span class="elsevierStyleHsp" style=""></span>min, with administration of a total volume of 5 cc of 0.2% ropivacaine. The procedure was uneventful.</p><p id="par0105" class="elsevierStylePara elsevierViewall">At the weekly follow-up visit, the mother reported complete pain remission, with no need for analgesics, and after 12 months of follow-up, the patient still has no pain and is able to live a normal life, return to school and continue with her sports activities. Both the patient and family report that the patient’s quality of life is now fully restored.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0110" class="elsevierStylePara elsevierViewall">We have reported a complex case of CAD in a paediatric patient who, despite various different treatments including advanced surgical techniques, still presented disabling pain. She was finally diagnosed with ACNES and her pain was resolved with PRF.</p><p id="par0115" class="elsevierStylePara elsevierViewall">CAD is a pathology that can lead to a significant decrease in quality of life that sometimes eludes an aetiological diagnosis. Paediatric ACNES is even more difficult to identify, and as many as 12% of patients are incorrectly diagnosed with functional pain.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,8</span></a></p><p id="par0120" class="elsevierStylePara elsevierViewall">Anatomically, the abdominal wall is innervated by the anterior and lateral cutaneous branches of the anterior ramus of the intercostal nerves (T8–T12), which course between the transverse abdominis muscle and the internal oblique muscle up to the junction with the rectus abdominis muscle. At this critical point, the fibres abruptly change direction to enter the rectus abdominis muscle and terminate on the external abdominal wall.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Although the pathophysiological mechanism is not yet clearly defined, ACNES is probably caused by mechanical compression or traction of the cutaneous branches of these intercostal nerves, although muscle contraction due to ischaemia or mechanical irritation could aggravate nerve compression at this level.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> The pain is located at these potential points of fibre compression, particularly in the right hemiabdomen. In the paediatric population, pain is more common in the right lower quadrant, 2−3<span class="elsevierStyleHsp" style=""></span>cm medial to the anterior superior iliac spine,<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> as was the case in our patient.</p><p id="par0125" class="elsevierStylePara elsevierViewall">In the 1920s, a surgeon named Carnett developed the diagnostic Carnett’s test to differentiate visceral pain from abdominal wall pain. This test is performed in 2 stages: first, the point of maximum pain is located by deep digital palpation, and usually corresponds to a very delimited area of less 2<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>2<span class="elsevierStyleHsp" style=""></span>cm. In the second stage, maintaining digital pressure, the patient is asked to contract the abdominal muscles by raising the head or legs. Skin sensitivity may also be altered, and the Pinch Test, which consists of pinching the painful subcutaneous fold between the index finger and thumb, may also be positive. In 1972, the term ACNES was coined by Dr. Applegate.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a></p><p id="par0130" class="elsevierStylePara elsevierViewall">Diagnosis in the paediatric population requires a detailed history and a comprehensive physical examination, maintaining a high index of suspicion once an organic origin has been ruled out. Sports has been identified as a trigger, particularly among adolescents, and incidence is higher in females.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0135" class="elsevierStylePara elsevierViewall">Based on established diagnostic criteria, our patient had a 6-month history of ACNES. She had received multiple lines of treatment, and had responded well to blind local anaesthetic infiltration and had reported transient improvement with TENS.</p><p id="par0140" class="elsevierStylePara elsevierViewall">Given the picture of disabling pain and evident disruption of the patient's biopsychosocial sphere, with significant emotional involvement, we discussed her case with a multidisciplinary team and proposed PRF as a less invasive alternative to surgical neurectomy, as no pathology had been identified on a recent exploratory laparoscopy.</p><p id="par0145" class="elsevierStylePara elsevierViewall">Radiofrequency or diathermy uses energy and heat to interrupt nerve stimuli and alleviate pain, improve quality of life, and even reduce or completely eliminate the need for analgesic treatment. In some cases the procedure will need to be repeated because the nerves usually regenerate over time. The indications for PRF include trigeminal neuralgia, radiculopathies or spinal pain, among others.</p><p id="par0150" class="elsevierStylePara elsevierViewall">PRF is a relatively new approach to the treatment of various neuropathic pathologies, and studies in adults with ACNES have shown it to be both safe and effective.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The use of ultrasound to guide needle placement has been shown to increase the success rate and reduce complications, particularly in children, in whom the target site is more superficial.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0155" class="elsevierStylePara elsevierViewall">In conclusion, ACNES should be considered in paediatric patients presenting with CAD, and innovative techniques such as PRF could be effective. A multidisciplinary approach is essential, as ACNES can greatly affect the quality of life of this population group.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Ethical considerations</span><p id="par0160" class="elsevierStylePara elsevierViewall">Informed consent was obtained from the patient’s mother for publication of this case report.</p><p id="par0165" class="elsevierStylePara elsevierViewall">All the authors have contributed to data collection, writing, and final approval of the manuscript.</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Conflict of interests</span><p id="par0170" class="elsevierStylePara elsevierViewall">None.</p><p id="par0175" class="elsevierStylePara elsevierViewall">No type of financial support has been received for writing the manuscript.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1967301" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1692678" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1967302" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1692679" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Case report" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Ethical considerations" ] 8 => array:2 [ "identificador" => "sec0025" "titulo" => "Conflict of interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2021-07-06" "fechaAceptado" => "2022-10-07" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1692678" "palabras" => array:3 [ 0 => "ACNES" 1 => "Abdominal wall pain" 2 => "Carnett" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1692679" "palabras" => array:3 [ 0 => "ACNES" 1 => "Dolor de pared abdominal" 2 => "Carnett" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Chronic abdominal pain is a highly prevalent entity in the paediatric population and represents a diagnostic challenge for professionals. It is frequently underdiagnosed, and must be treated by a multidisciplinary team after a detailed clinical evaluation has been performed to rule out other pathologies. Anterior cutaneous nerve entrapment syndrome (ACNES) occurs when the anterior cutaneous abdominal nerves become pinched or trapped, causing intense, unilateral, circumscribed abdominal pain. Patients often present a positive Pinch test or Carnett’s sign. A stepwise therapeutic approach should be used, reserving the most invasive techniques for patients with refractory ACNES. Among the many different treatments available, local anaesthesia infiltration has shown a high success rate, and surgery should only be performed in the most refractory cases. We report the case of an 11-year old girl with a 6-month history of ACNES that severely affected her quality of life, who responded well to pulsed radiofrequency ablation.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">El dolor abdominal crónico es una entidad muy prevalente en la población pediátrica y supone todo un reto diagnóstico para los profesionales, siendo frecuentemente infradiagnosticada. Requiere un abordaje multidisciplinar y una minuciosa evaluación clínica para descartar otras patologías. El ACNES consiste en un atrapamiento de los nervios cutáneos anteriores de los nervios intercostales y origina un dolor abdominal intenso, unilateral, circunscrito y frecuentemente presenta un Pinch test y un Test de Carnett positivos. El planteamiento terapéutico debería contemplarse desde un enfoque gradual, reservando las técnicas más invasivas para los pacientes con ACNES refractario. Entre los múltiples tratamientos posibles, las infiltraciones locales presentan una alta tasa de éxito, reservando las técnicas quirúrgicas para los casos refractarios. Presentamos el caso clínico de una niña de 11 años, con ACNES de 6 meses de evolución con grave afectación de su calidad de vida y con respuesta favorable a la técnica de radiofrecuencia pulsada.</p></span>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1666 "Ancho" => 1675 "Tamanyo" => 231567 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0035" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Delimitation of the site of pain in the lower right hemiabdomen.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1279 "Ancho" => 1675 "Tamanyo" => 215043 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0040" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Ultrasound-guided in-plane puncture.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1142 "Ancho" => 1675 "Tamanyo" => 186711 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0045" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The needle (identified with the red arrow) is advanced until the target site is located in the anterior rectus muscle.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Treatment strategies for anterior cutaneous nerve entrapment syndrome in children: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:6 [ 0 => "J. 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Case report
Pulsed radiofrequency in a paediatric patient with disabling ACNES
Técnica de radiofrecuencia pulsada en síndrome de ACNES invalidante en una paciente pediátrica
A. Server
, V. Sánchez, Á. Mesas, J. Medel
Corresponding author
Servicio de Anestesiología, Hospital Universitari Vall d’Hebron, Barcelona, Spain