Abdominal Cutaneous Nerve Entrapment Syndrome: The Cause of Localized Abdominal Pain in a Young Pregnant Woman

Abdominal Cutaneous Nerve Entrapment Syndrome: The Cause of Localized Abdominal Pain in a Young Pregnant Woman

The Journal of Emergency Medicine, Vol. -, No. -, pp. 1–4, 2018 Ó 2018 Elsevier Inc. All rights reserved. 0736-4679/$ - see front matter https://doi...

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The Journal of Emergency Medicine, Vol. -, No. -, pp. 1–4, 2018 Ó 2018 Elsevier Inc. All rights reserved. 0736-4679/$ - see front matter

https://doi.org/10.1016/j.jemermed.2018.01.035

Clinical Communications: Adult ABDOMINAL CUTANEOUS NERVE ENTRAPMENT SYNDROME: THE CAUSE OF LOCALIZED ABDOMINAL PAIN IN A YOUNG PREGNANT WOMAN Sody A. Naimer, MD Family Health Center, Clalit Health Services, Eilon Moreh, Israel and Department of Family Medicine, Siaal Research Center for Family Medicine and Primary Care Research, Faculty of Health Sciences, Ben-Gurion University of the Negev, Beer-Sheva, Israel Reprint Address: Sody A. Naimer, MD, Department of Family Medicine, Faculty of Health Sciences, Ben-Gurion University of the Negev, P.O. Box 653, Beer-Sheva 84105, Israel

, Keywords—torso wall pain; abdominal pain; cutaneous nerve entrapment; soft tissue pain; chest wall pain

, Abstract—Background: Despite the broad differential diagnosis in any patient referring with symptoms involving the chest or abdomen, a small number of conditions overshadow the rest by their probability. Chest and abdominal wall pain continues to constitute a common and expensive overlooked source of pain of unknown cause. In particular, cutaneous nerve entrapment syndrome is commonly encountered but not easily diagnosed unless its specific symptoms are sought and the precise physical examination undertaken. Case Report: A primigravida woman with unbearable abdominal pain was referred repeatedly seeking a solution for her suffering. Numerous laboratory and imaging studies were employed in order to elucidate the cause of her condition. After numerous visits and unnecessary delay, the diagnosis was finally made by a physician fully versed in the field of torso wall pain. The focused physical examination disclosed abdominal cutaneous nerve entrapment syndrome as the diagnosis, and anesthetic infiltration led to immediate alleviation of her pain. Why Should an Emergency Physician Be Aware of This?: Cutaneous nerve entrapment is a common cause of abdominal pain that is reached on the basis of thorough history and physical examination alone. Knowledge dissemination of the various torso wall syndromes is imperative for prompt delivery of suitable care. All emergency physicians should be fully aware of this entity because the diagnosis is based solely on physical examination, and immediate relief can be provided in the framework of the first visit. Wider recognition of this syndrome will promise that such mishaps are not repeated in the future. Ó 2018 Elsevier Inc. All rights reserved.

INTRODUCTION This report relays the case of a pregnant woman with pain involving the right anterior torso. Multiple medical personnel lacking experience confronting such cases failed to make the correct diagnosis, despite the ability to reach a conclusive diagnosis with history and physical examination alone. After appropriate intervention, significant symptomatic improvement was observed and much of the patient’s fears were allayed. This case raises the importance of knowledge of the torso wall as a common source of pain. CASE REPORT A previously healthy 22-year-old primigravida woman presented to her general practitioner after a week-long hospitalization. Three weeks before, at 19 weeks of gestation, she awakened at night with severe pain. It originated between the right lower chest and upper abdomen radiating downward. It was not associated with trauma or activity. She denied any change in bowel habits, fever, or urinary symptoms. Evaluation at the emergency department she was referred to that morning disclosed mild pyuria only. She returned home with oral antibiotics. The next day her pain persisted and

RECEIVED: 29 June 2017; FINAL SUBMISSION RECEIVED: 4 December 2017; ACCEPTED: 25 January 2018 1

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appeared constant as opposed to previously episodic and she was sent to the hospital. Upon examination there was mild tenderness without rebound over her right abdomen. She was admitted, analgesics were administered and a workup commenced. Laboratory values were normal. Abdominal ultrasound revealed a normal fetus and a small uterine myoma. Fetal monitoring was normal and the abdominal pain was found to be nonspecific. Days after her release she was sent to the emergency department for a third time by another general practitioner for persistent pain. On this last visit, she was seen by an experienced family physician. Only then did it become obvious that the tenderness originated from the torso wall. Informed consent was received and then a mixture of 5 mL 1% ezracaine and 1 mL triamcinolone acetonide (10 mg) was infiltrated into the fascial plane in a fanning fashion from the superolateral aspect into the tender zone, applying the modified technique. Immediately after the procedure, the patient reported significant improvement, which reinforced the current diagnosis. After treatment the condition resolved. DISCUSSION In any patient referring with symptoms involving the chest or abdomen, torso wall syndromes should be

considered (1). A wide array of conditions should be considered in a patient presenting with chest pain. Despite the broad differential diagnosis (Table 1), a small number of conditions overshadow the rest by their probability. At this visit, thorough examination revealed classic ‘‘dough-rolling’’ or ‘‘pinch’’ tenderness of the abdominal wall, elicited in the region of the right costal margin, progressing inferiorly toward the midline (Figure 1A). Abdominal cutaneous nerve entrapment syndrome (ACNES) is a pain syndrome thought to be the result of entrapment of cutaneous branches of an intercostal nerve at the lateral edge of the rectus abdominis that causes severe, refractory, and chronic pain (2). The anterior cutaneous branches of the thoracoabdominal (T7–11) and subcostal (T12) nerves are the most susceptible to entrapment. These sensory nerves run in a plane between the internal oblique and transverses abdominis muscles. The thoracic nerves advance to the posterior wall of the rectus sheath and each enters a neurovascular channel in the rectus muscle to supply the skin. Each of the neurovascular channels in the rectus muscle contains a fibrous ring that should allow the anterior cutaneous nerve to pass through freely; this ring, however, can also become a site of nerve compression and ischemia resulting in symptoms of ACNES (3).

Table 1. Diagnoses to Consider as the Source of Torso Wall Pain I. Chest or abdominal wall pain  Cutaneous nerve entrapment syndrome, one of the most common conditions leading to this symptom  Fibromyalgia, widespread musculoskeletal pain and tenderness, associated with depression and anxiety  Herpes zoster neuralgia, more often with cutaneous clusters of vesicles  Focal induration, panniculitis, fasciitis  Nodular lesion, leiomyoma, glomangioma, metastatic carcinoma II. Chest wall pain  Costochondritis, multiple areas, usually in the upper costal cartilages there is no swelling  Slipped rib syndromes, typically costal margin region  Tietze’s syndrome, costosternal, sternoclavicular, or costochondral joints, most often involving the second and third ribs, swelling usually apparent  Sternalis syndrome, localized tenderness directly over the sternum or overlying sternalis muscle  Inflammatory disease: rheumatoid arthritis, psoriatic arthritis, systemic lupus erythematosus, relapsing polychondritis, each usually involve additional body regions  Xiphoidynia, typically over midline: sternal/epigastric region  Fractures: stress, usually sports injuries from extreme biomechanical load Pathological, associated with metastatic infiltration Osteoporotic, may be associated with chronic corticosteroid use  Infection, post-thoracotomy and sternotomy osteomyelitis or fistulae, septic arthritis of chest wall  Sternoclavicular hyperostosis, swelling may be noted in designated region  Spontaneous sternoclavicular subluxation, specifically over the sternoclavicular joint III. Abdominal wall pain B Abdominal wall defects:  Abdominal wall hernias, epigastric, Spigelian, or umbilical  Surgical scars, full laparotomy or trocar insertion sites after laparoscopic surgery B Referred pain:  Thoracic nerve radiculopathy, disease in the T7 to T12 nerve roots  Thoracic spinal conditions disc prolapse or spinal cord tumors.  Pain generated from the ribcage and chest wall, source actually more superior anatomical sites B Infiltration of abdominal wall  rectus sheath hematoma, especially suspected when under anticoagulant therapy  abdominal wall endometriosis B Mechanical:  ribs on pelvis syndrome, mechanical friction more common with old and frail in subject with low body mass index.

Abdominal Cutaneous Nerve Entrapment Syndrome

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Figure 1. (A) Manipulation of trigger point: ‘‘dough rolling’’ will illicit exquisite pain. (B) Raising the soft tissue up and away from the torso wall enables safe penetration without fear of thoracic or viscus penetration.

This patient was informed that her condition is an established entity and improvement is expected with a simple intervention. She was delighted to learn that the problem is not dangerous and that her symptoms had an identifiable cause. Abdominal wall pain continues to constitute a common and costly overlooked source of abdominal pain of unknown cause (2–8). Its prevalence in patients presenting to the emergency department with acute abdominal pain reaches 2% (9). This entity should be considered, especially in pregnancy, which is a known risk for this condition (10). There are usually no specific triggers or associated pathology or history related to this syndrome. Abdominal pain may occur in pregnancy, after recent weight gain or after surgery (11). The pain is initially vague, without a clear focus and eventually

worsens and becomes constant. It may present either as localized pain in the vicinity of the midclavicular line or lateral boundaries of the rectus abdominis muscle or have a more diffuse character (3). Physical examination must include identifying the precise location of maximal intensity of pain. Palpation can accurately demarcate the limits of the region involved and assists assessment of both the consistency of the painful zone and its local temperature. Elicited tenderness may lead to apprehension, guarding, or resistance. The primary objective is to differentiate between tenderness that stems from any of the underlying skeletal or visceral structures from the soft tissue itself. Classic signs include: ‘‘hover sign,’’ which refers to patients who guard the affected area from the examiner’s hands, and ‘‘Carnett sign,’’ which refers to the

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disappearance of abdominal tenderness to palpation when the anterior abdominal muscles are contracted, indicating pain of intra-abdominal origin, while persistence of tenderness suggests a source in the abdominal wall (10). Dough rolling or pinching the area of maximal intensity (trigger point) will result in an intensely painful sensation (Figure 1A). A single injection combining an anesthetic and a steroid agent helps secure the diagnosis and can provide immediate and long-term relief of symptoms (12). There is no literature comparing steroid agents or their doses. We normally select either triamcinolone (Kenolog; Sandoz Canada Inc., Boucherville, Quebec, Canada) or betamethasone acetate and betamethasone sodium phosphate injection of 1–2 mL (Celestone; Merck Sharp & Dohme, Kenilworth, NJ). The favorable safety profile of these agents and their minimal expected systemic absorption allow their use freely with this procedure during gestation. A safe precaution to reassure that the intervention will not result in vital internal organ or thoracic cavity injury can be provided by injecting into a raised mound of the affected tissue (modified technique) (Figure 1B) (13). A further technique that has been reported to facilitate needle penetration for medication infiltration is ultrasound guidance with a vascular probe (14,15). A comprehensive analysis of the current literature on management by Chrona et al. exposes the apparent benefit of steroid in the infiltration, although this has not been compared to injection of anesthetic alone (16). For refractory cases with intense and far more extended periods of pain, there are those that have even resort to surgery, such as neurectomy (17–19). However, it seems that in the common cases, such procedures are far from necessary. WHY SHOULD AN EMERGENCY PHYSICIAN BE AWARE OF THIS? This patient is not unique. She demonstrates that some patients are difficult to diagnose despite repeat referrals and investigations unless the correct examination is performed and the exact diagnosis is sought. Cutaneous nerve entrapment is a common cause of abdominal pain, reached on the basis of thorough history and physical examination alone. Knowledge dissemination of the various torso wall syndromes is imperative for prompt delivery of suitable care. All emergency

S. A. Naimer

physicians should remain fully aware of this entity because the diagnosis is based solely on physical examination and immediate relief can be provided in the framework of the first visit. REFERENCES 1. Langdon DE. Abdominal wall pain will be missed until examinations change!. Am J Gastroenterol 2002;97:3207–3208. 2. Kopell HP, Thompson WA. Peripheral Entrapment Neuropathies. Malabar, FL: Robert E. Kreiger Publishing; 1976:85–88. 3. Applegate WV. Abdominal cutaneous nerve entrapment syndrome (ACNES): a commonly overlooked cause of abdominal pain. Perm J 2002;6:20–27. 4. Habib PA, Huang GS, Mendiola JA, Yu JS. Anterior chest pain: musculoskeletal considerations. Emerg Radiol 2004;11:37–45. 5. Bo¨sner S, Becker A, Hani MA, et al. Chest wall syndrome in primary care patients with chest pain: presentation, associated features and diagnosis. Fam Pract 2010;27:363–369. 6. Suleiman S, Johnston DE. The abdominal wall: an overlooked source of pain. Am Fam Physician 2001;64:431–438. 7. Srinivasan R, Greenbaum DS. Chronic abdominal wall pain: a frequently overlooked problem. Practical approach to diagnosis and management. Am J Gastroenterol 2002;97:824–830. 8. Costanza CD, Longstreth GF, Liu AL. Chronic abdominal wall pain: clinical features, health care costs, and long-term outcome. Clin Gastroenterol Hepatol 2004;2:395–399. 9. van Assen T, Brouns J, Scheltinga M, Roumen R. Incidence of abdominal pain due to the anterior cutaneous nerve entrapment syndrome in an emergency department. Scand J Trauma Resusc Emerg Med 2015;23(19):2–6. 10. Carnett JB. Intercostal neuralgia as a cause of abdominal pain and tenderness. Surg Gynecol Obstet 1926;42:625–632. 11. Roderick E, Normal B. Anterior cutaneous nerve entrapment syndrome: an unusual cause of abdominal pain during pregnancy. Int J Obstet Anesth 2016;25:96–97. 12. Greenbaum DS, Joseph JG. Abdominal wall tenderness test. Lancet 1991;337:1606–1607. 13. Naimer SA. Modified infiltration technique for cutaneous nerve entrapment syndrome. Br J Med Med Res 2015;8:672–677. 14. Kanakarajan S, High K, Naaraja R. Chronic abdominal wall pain and ultrasound guided abdominal cutaneous nerve infiltration: a case series. Pain Med 2011;12:382–386. 15. Adoni A, Kostopanagiotou G, Saranteas T, Batistak C. Ultrasoundguided anterior abdominal cutaneous nerve block for the management of bilateral abdominal cutaneous nerve entrapment syndrome (ACNES). Pain Physician 2013;16:E799–801. 16. Chrona E, Kostopanagiotou G, Damigos D, Batistaki C. Anterior cutaneous nerve entrapment syndrome: management challenges. J Pain Res 2017;10:145–156. 17. Boelens OB, van Assen T, Houterman S, Scheltinga MR, Roumen RM. A double-blind, randomized, controlled trial on surgery for chronic abdominal pain due to anterior cutaneous nerve entrapment syndrome. Ann Surg 2013;257:845–849. 18. Boelens OB, Scheltinga MR, Houterman S, Roumen RM. Management of anterior cutaneous nerve entrapment syndrome in a cohort of 139 patients. Ann Surg 2011;254:1054–1058. 19. Zganjer M, Bojic D, Bumci I. Surgery for abdominal wall pain caused by cutaneous nerve entrapment in children—a single institution experience in the last 5 years. Iran Red Crescent Med J 2013; 15:157–160.