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JAMA Clinical Challenge |

Episodic Abdominal and Chest Pain in a Young Adult FREE

Marcello Migliore, MD, PhD; Maria Signorelli, MD, PhD
[+] Author Affiliations

Author Affiliations: Thoracic Surgery, Department of Surgery (Dr Migliore), and Unit of Psychiatry (Dr Signorelli), Policlinic Hospital, University of Catania, Catania, Italy.


JAMA. 2012;307(16):1746-1747. doi:10.1001/jama.2012.506.
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A 22-year-old man has experienced intermittent severe chest and abdominal pain for a year. He stopped playing water polo because of recurrent severe abdominal pain in his right and left upper quadrant. He was evaluated for suspected pancreatitis, but all diagnostic results were unremarkable. A few weeks later he was admitted for acute chest pain, and again all diagnostic results were unremarkable. In 1 year the patient had 7 hospital admissions for abdominal or chest pain with no episodes of fever, chills, sweats, or cough. There is no history of trauma. The patient is currently treated with ketoprofen, oral morphine, and anxiolytic drugs. He spends almost all day in bed but is unable to sleep in a prone position and has pain in his throat. He has taken a leave of absence from college and reduced his social activities. The patient has received injections of cortisone and ketoprofen in the xiphoid area administered 10 days apart; however, pain persists. Physical examination is remarkable for considerable pain over the xiphoid on palpation. Slight pain is elicited on examination of the right second and third sternocostal cartilages. Chest computed tomography (CT) has been performed several times with interpretations ranging from osteochondritis to xiphoid fracture (Figure 1).

Figure 1. Computed tomography scan showing heterotopic ossification or chondrification (arrow) erroneously interpreted as xiphoid fracture.

Grahic Jump LocationImage not available.

  • A. Do nothing; the abdominal and chest pain will disappear over time

  • B. Perform ambulatory esophageal pH and pressure monitoring

  • C. Perform xiphoidectomy

  • D. Prescribe different anxiolytic drugs and oral steroids

Hypersensitive xiphoid syndrome (so-called xiphodynia)

C. Perform xiphoidectomy

The key clinical feature in this case is making the diagnosis in a young patient with chest and abdominal pain of unknown origin. The key to making the diagnosis of hypersensitive xiphoid syndrome is the reproduction of pain when the tender part of the xiphoid is subjected to very moderate hand pressure.

The word xiphodynia comes from the Greek xiphos for sword (the xiphoid process) and odyne for physical pain, often of an acute and sudden nature. First described in 1712, xiphodynia is a rare syndrome capable of producing myriad symptoms that can mimic several common thoracic and abdominal diseases. Common symptoms of xiphodynia may include cardiac chest pain; pain radiating into the back, neck, shoulders, arms, and chest wall; epigastric pain; nausea; vomiting; and diarrhea. Although Lipkin et al1 found the syndrome present in about 2% of the population of a general hospital ward and stated that xiphodynia is “far more common than is generally appreciated,” no data are available on the incidence or prevalence of xiphodynia.

The diagnosis of xiphodynia is mainly clinical. Careful examination of the xiphoid should be part of a routine examination of any patient presenting with chest and abdominal pain of unknown origin. A simple provocative test, moderate pressure on the xiphoid process, may uncover a symptomatic xiphoid process and establish the diagnosis of xiphodynia. In some circumstances the prominence of the xiphoid process under the skin is evident,2 but in others the cause of xiphodynia remains unknown.

Differential diagnosis includes a variety of conditions, such as esophagitis, angina, cholecystitis, pleuritic chest pain, and inflammation of the condral cartilage. Frequently, despite extensive investigations, no definitive diagnosis can be established. The presence of refractory and recurrent pain may be erroneously treated with analgesics and psychotropic drugs.

Patients with xiphodynia are usually treated with analgesics, an elastic rib belt,3 topical heat and cold, and a course of ultrasound and laser,4 but conservative physical therapies may not be effective. However, local injection with an anesthetic-steroid combination may provide symptomatic relief.5 In the few reported patients for whom medical treatment has failed, xiphoidectomy provides resolution of symptoms.2,4 Ambulatory esophageal pH and pressure monitoring, to rule out possible pathologic gastroesophageal reflux, should not be necessary with a proper physical examination. The decision to perform a xiphoidectomy in this case was supported by the persistence of symptoms after injections of cortisone and ketoprofen.

In this patient, CT demonstrated some heterotopic ossification/chondrification with no gross deformity of the xiphoid (Figure 1). Intraoperatively, a 2-cm cylindrical piece of cartilage was found along the insertion of the anterior rectus abdominis with the xiphoid, and this cartilage was removed. The pathology report revealed a cartilaginous process (Figure 2). The patient outcome was remarkable, and the first night following the operation the patient slept in a prone position and symptoms resolved. Postoperatively the patient was symptom free. At 10 months' follow-up, the patient remains symptom free and has resumed studying and playing water polo.

Figure 2. Microscopic examination shows cartilagineous tissue with fibers of striated muscle. Sampled from the cartilagenous tissue along the insertion of the rectus abdominis with the xyphoid (hematoxylin-eosin, original magnification × 200).

Grahic Jump LocationImage not available.

Corresponding Author: Marcello Migliore, MD, PhD, Thoracic Surgery, Department of Surgery, University of Catania, Via S Sofia, 95124, Catania, Italy (mmiglior@unict.it).

Conflict of Interest Disclosures: The authors have completed and submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest and none were reported.

Additional Contributions: We thank the patient for providing permission to publish his information. We also thank Rosario Caltabiano, University of Catania, Department of Pathology, for his assistance with the microscopic imaging of Figure 2.

Lipkin M, Fulton LA, Wolfson EA. The syndrome of the hypersensitive xiphoid.  N Engl J Med. 1955;253(14):591-597
PubMed   |  Link to Article
Maigne JY, Vareli M, Rousset P, Cornelis P. Xiphodynia and prominence of the xyphoid process: value of xiphosternal angle measurement: three case reports.  Joint Bone Spine. 2010;77(5):474-476
PubMed   |  Link to Article
Simpson JK, Hawken E. Xiphodynia: a diagnostic conundrum.  Chiropr Osteopat. 2007;1513
PubMed  |  Link to Article   |  Link to Article
Ergin M, Yeginsu A. Thalassemia minor presenting with xiphodynia.  J Clin Anal Med. 2011;2(3):119-120
Howell JM. Xiphodynia: a report of three cases.  J Emerg Med. 1992;10(4):435-438
PubMed   |  Link to Article

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References

Lipkin M, Fulton LA, Wolfson EA. The syndrome of the hypersensitive xiphoid.  N Engl J Med. 1955;253(14):591-597
PubMed   |  Link to Article
Maigne JY, Vareli M, Rousset P, Cornelis P. Xiphodynia and prominence of the xyphoid process: value of xiphosternal angle measurement: three case reports.  Joint Bone Spine. 2010;77(5):474-476
PubMed   |  Link to Article
Simpson JK, Hawken E. Xiphodynia: a diagnostic conundrum.  Chiropr Osteopat. 2007;1513
PubMed  |  Link to Article   |  Link to Article
Ergin M, Yeginsu A. Thalassemia minor presenting with xiphodynia.  J Clin Anal Med. 2011;2(3):119-120
Howell JM. Xiphodynia: a report of three cases.  J Emerg Med. 1992;10(4):435-438
PubMed   |  Link to Article
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