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Case Report
2024
:3;
100342
doi:
10.1016/j.jorep.2024.100342

Unfused sternum as a rare cause of gradual onset chest pain – Case report

Department of Orthopedic Surgery, Medical Center Leeuwarden, the Netherlands

⁎Corresponding author: Joris Raessens. joris.raessens@mzh.nl

Disclaimer:
This article was originally published by Reed Elsevier India Pvt. Ltd. and was migrated to Scientific Scholar after the change of Publisher.

Abstract

Abstract

There is a wide range of orthopedic differential diagnoses for sternal pain. Incomplete development of the sternum, resulting in unfused sternebrae, may gradually become symptomatic after repetitive stress and can mimic a stress fracture. A symptomatic unfused sternum is a rare cause of sternal pain.

A healthy young man presented with 3 years of chest wall pain with sudden onset. He performed physical labor on a fish farm and also worked as a gasfitter. Physical examination revealed mild pain when palpating the sternum. Conventional X-rays did not show any abnormalities. Subsequent bone scintigraphy and single-photon emission computer tomography (SPECT) revealed an unfused sternal body with an image similar to a sternal fracture, which was confirmed by conventional computer tomography (CT).

To our knowledge, this is the first case describing complaints of a previously asymptomatic unfused sternum. Developmental disorders of the sternum should be considered in the differential diagnosis of chest wall pain, especially when there is habitual physical activity of the upper extremities.

Keywords

Unfused sternum
Sternum pain
Chest pain
1

1 Introduction

Sternal fractures are most commonly caused by direct trauma. Spontaneous fractures of the sternum are rare. Our case involves a healthy young man with sternal pain without any preceding trauma, caused by microtrauma to unfused sternebrae of a previous asymptomatic congenital bony deformation.

2

2 Case report

A 22-year-old man presented to our orthopaedic outpatient clinic with complaints of anterior chest pain for a period of 3 years. There was no history of trauma. The pain was described as sharp and was located in the center of the chest. The pain had started while performing strenuous physical labor on a fish farm. Initially, physical movements did not provoke any complaints. Subsequently, the patient noticed a direct relationship between physical effort and the intensity of pain. Because of these complaints, he quit his job and quit exercising for a period of two months. Nonetheless, the pain in his chest never fully subsided during this time. The patient worked as an electrician and gasfitter for following two years. The chest pain was especially aggravated by performing horizontal shoulder abduction. Resting and hunching over temporarily reduced the complaints. Over the last few years, the pain gradually increased, and conservative treatment (non-steroid anti-inflammatory drugs and physical therapy) did not give sufficient relief. His medical history was not remarkable, nor had he undergone surgery in the past. The patient did not experience fevers, nocturnal sweating, weight loss, shoulder pain, neck pain, or radiculopathy. He had never used corticosteroids. None of his family members were familiar with spontaneous fractures, nor were there any known congenital disorders.

3

3 Clinical findings

Physical examination revealed a slender young man with a minor pectus excavatum. Palpation over the sternum was slightly painful, although he was able to exactly point out the punctum maximum of the pain. At the moment of examination, his symptoms were mild. No excessive thoracic kyphosis was observed. There was no muscle atrophy. Bilateral shoulder range of motion and strength testing were normal. There were no clinical signs of cardiovascular or pulmonary disease.

4

4 Diagnostic assessment

Chest X-rays did not show any abnormalities. A technetium bone scan plus single-photon emission computer tomography (SPECT) of the thorax showed increased osteoblastic activity in the midline of the thorax (Fig. 1). Furthermore, there was suspicion of a sclerotic disruption of the cortical bone on the dorsal side of the sternum. This was confirmed by additional conventional computer tomography (CT), which revealed an image suggestive of a sternum fracture with a sclerotic reaction (Fig. 2).

Bone scintigraphy (A), low diagnostic CT (B) and SPECT (C) of the thorax showed increased activity in the sternum. The dorsal side of the sternum showed sclerosis with suspection of cortical dis-ruption.
Fig. 1 Bone scintigraphy (A), low diagnostic CT (B) and SPECT (C) of the thorax showed increased activity in the sternum. The dorsal side of the sternum showed sclerosis with suspection of cortical dis-ruption.
Sagittal (A) and coronal (B) views of CT of sternum showing the sternomanubrial joint (black arrows) and the transverse cleft of the sternal body(white arrows).
Fig. 2 Sagittal (A) and coronal (B) views of CT of sternum showing the sternomanubrial joint (black arrows) and the transverse cleft of the sternal body(white arrows).
5

5 Differential diagnosis (if any)

There is a wide range of orthopedic differential diagnoses for sternal pain, such as sternal fracture, costochondritis, sternoclavicular osteoarthritis, costal fracture, pectoralis major tendon rupture, osteomyelitis, and congenital abnormalities.1,2

Incomplete development of the sternum, resulting in unfused sternebrae, may gradually become symptomatic after repetitive stress and can mimic a stress fracture. A symptomatic unfused sternum is a very rare cause of sternal pain. Most causes of sternal pain can easily be differentiated from a symptomatic unfused sternum by conventional diagnostic modalities such as X-rays, sonography, bone scintigraphy, and/or CT. In contrast, a stress fracture of the sternum can fully mimic a symptomatic unfused sternum and be mistakenly regarded as the cause of the pain.

6

6 Therapeutic intervention

To our knowledge, no prior studies have examined the management of a symptomatic unfused sternal body. The more commonly described isolated stable sternal fracture with minimal dislocation usually does not require invasive treatment and heals spontaneously over the average course of 10 weeks.3 The management of other symptomatic unfused bony segments in the body, such as os acromiale or os trigonum is often invasive and includes simple excision or operative fixation.

As our patient currently only experienced mild symptoms, and had experienced a variable degree of severity over the course of multiple years, invasive treatment was undesirable.

Although a true fracture is different in nature than a symptomatic unfused bony segment, as the unfused sternal body was previously asymptomatic we decided that initial treatment should be similar to management of a sternal stress fracture.

Our patient initially reduced his physical effort while working.

7

7 Follow-up and outcomes

Nine months after load reduction the patient reported that he experienced little to no pain and was satisfied with the result of the treatment. In the future, open reduction and internal fixation may still be a viable option if complaints return and repeated conservative treatment would prove insufficient.

8

8 Discussion

8.1

8.1 Anatomy and development of the sternum

The adult sternum can be divided into three segments connected by cartilaginous joints: the manumbrium, mesosternum, and xiphoid process.

The development of the sternum starts during early gestation, when the cartilaginous precursor of the sternum is formed from a pair of longitudinal mesenchymal bars. These sternal bars migrate to the midline and eventually fuse, starting cranially and gradually progressing to the caudal end. Fusion is finished around the 9th week of gestation.4

The sternebrae, the ossification centers formed by the fusion of both sides of the sternal bars, continue to merge until early adulthood, eventually shaping the adult sternum, as seen in Fig. 3. By the age of 25 years, the calcification and fusion of the sternum is usually completed.

Schematic representation of the development of the sternum. A. unfused infant sternum with (partially fused) ossification centers forming sternebrae. B. adult sternum with fused sternebrae. C. adult sternum with unfused bar in the middle of the sternal body.
Fig. 3 Schematic representation of the development of the sternum. A. unfused infant sternum with (partially fused) ossification centers forming sternebrae. B. adult sternum with fused sternebrae. C. adult sternum with unfused bar in the middle of the sternal body.

Failure in the process of fusion of the sternal bars results in longitudinal sternal abnormalities, such as foramina, clefts, tubercles, and bifid-xiphioid process, which have been previously described.5 These anatomical variations of the sternum are relatively frequent, usually asymptomatic, and often detected incidentally.

8.2

8.2 Stress fracture of the sternum

Stress fractures are fatigue-induced fractures of the bone caused by repeated submaximal loading.6 As stress fractures most commonly occur in weight-bearing bones, a stress-related fracture of the sternum is rare. Repetitive contraction of the anterior thoracic musculature (mainly the pectoralis major and rectus abdominis) increases stress on the sternum. If these forces exceed the tensile strength of the bone, fractures can occur.1 In literature, 10 cases of sternal or manubrial stress fractures were reported.7–14 Of these reports, almost all were associated with physical exercise and had an acute onset of sharp pain. In our case, the complaints did not occur acutely.

The location of the suspected sternal fracture and the deformed manubriosternal joint on CT forced us to consider congenital factors or variations. Pain caused by common unfused skeletal variations, such as the os trigonum in the foot and the os acromiale of the shoulder, has been widely described. Given the gradual onset of the pain and the age of the patient, we hypothesize that a previously unfused sternal ossification center had become symptomatic due to the repetitive, strenuous exercises. To our knowledge, this is even rarer than a stress fracture, which highlights the uniqueness of this case. Non-fusion of sternal body segments is a far less described variation, with only one case report describing a rare variation of a combination of failed intra- and intersegmental sternal fusion in a skeleton from an anatomical collection.15

With respect to other possible causes of chest pain, thoracic kyphosis has been described as an underlying cause of increased load on the sternum, which could lead to a stress fracture, especially in elderly osteoporotic patients.16,17 Our patient did not show any abnormalities of his spine.

Furthermore, costo- and sternoclaviculair causes were unlikely, since the SPECT-CT clearly revealed the anatomic origin. Moreover, the circumscript location of the increased scintigraphic activity in the sternum was atypical for osteomyelitis. The patient had no history of sternal surgery. Lastly, cardiopulmonary causes of spontaneous chest pain were considered in the differential diagnosis. Based on history and physical examination, coronary artery disease was deemed very unlikely when the patient was presented to us. In addition, pulmonary symptoms were excluded (as coughing also stresses the sternum). Corticosteroid intake for chronic obstructive pulmonary disease was shown to be related to a spontaneous sternal fracture.18 Our patient, however, had never used corticosteroids. In addition, abdominal and gastrointestinal pathology like gastroesophageal reflux disease, pancreatitis, and peptic ulcers have to be kept in mind as a cause of sternal pain. However, medical history in combination with the findings on the bone scintigraphy made the existence of gastrointestinal problems less likely in this case.

In conclusion, considering our patients age and the normal development pattern of merging and calcification of the sternum, the sternal pain in this case was probably based on pre-existing unfused sternum, which gradually became symptomatic due to repetitive strenuous exercise. Diagnosis can be confirmed with bone scintigraphy, SPECT and CT.

Developmental disorders of the sternum should be considered in the differential diagnosis of chest wall pain, especially when there is habitual physical activity of the upper extremities.

Credit author statement

JR and EZ were directly involved in patient care and wrote the report with input of all authors. WZ and PR were overseeing patient care and revised and approved the final version to be published.

Financial support and sponsorship

None.

Informed consent

Patient consented for images and other clinical information relating to his case to be reported in a medical publication.

Authors contribution

JR and EZ were directly involved in patient care and wrote the report with input of all authors. WZ and PR were overseeing patient care and revised and approved the final version to be published.

Ethical Statement for solid state ionics

Hereby, I Joris Raessens consciously assure that for the manuscript “Unfused sternum as a rare cause of gradual onset chest pain – Case report” the following is fulfilled.1)This material is the authors' own original work, which has not been previously published elsewhere.2)The paper is not currently being considered for publication elsewhere.3)The paper reflects the authors' own research and analysis in a truthful and complete manner.4)The paper properly credits the meaningful contributions of co-authors and co-researchers.5)The results are appropriately placed in the context of prior and existing research.6)All sources used are properly disclosed (correct citation). Literally copying of text must be indicated as such by using quotation marks and giving proper reference.7)All authors have been personally and actively involved in substantial work leading to the paper, and will take public responsibility for its content.

The violation of the Ethical Statement rules may result in severe consequences.

To verify originality, your article may be checked by the originality detection software iThenticate. See also http://www.elsevier.com/editors/plagdetect.

Funding statement

The authors of the manuscript “Unfused sternum as a rare cause of gradual onset chest pain – Case report” hereby declare they recieved no financial support or sponsorship of any kind in the course of study, research or assembly of the manuscript.

Consent to publication

Authors declare that patient has given full permission for the publication, reproduction, broadcast and other use of photographs, recordings and other visual and textual material. Patient has declared, in consequence of granting this permission, that he has no claim on ground of breach of confidence or any other ground in any legal system against — (author's/developer's name) — and its agents, publishers, successors and assigns in respect of such use of the photograph(s) and textual material (case histories). Patient agrees to release and discharge the authors, and any editors or other contributors and their agents, publishers, successors and assigns from any and all claims, demands or causes of action that I may now have or may hereafter have for libel, defamation, invasion of privacy, copyright or moral rights or violation of any other rights arising out of or relating to any use of his image or case history.

Declaration of competing interest

The authors whose names are listed immediately below certify that they have NO affiliations with or involvement in any organization or entity with any financial interest (such as honoraria; educational grants; participation in speakers’ bureaus; membership, employment, consultancies, stock ownership, or other equity interest; and expert testimony or patent-licensing arrangements), or non-financial interest (such as personal or professional relationships, affiliations, knowledge or beliefs) in the subject matter or materials discussed in this manuscript.

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