pISSN 2384-1095 iMRI 2020;24:55-60 https://doi.org/10.13104/imri.2020.24.1.55 eISSN 2384-1109

Dynamic Contrast-Enhanced MR Imaging of Tietze’s Syndrome: a Case Report

Dong Chan Kim, Sang Yoon Kim, Bong Man Kim Department of Radiology, Dankook University Hospital, Cheonan, Korea

Tietze's syndrome is an inflammatory condition associated with painful swelling of the costochondral, costosternal, and sternoclavicular . Tietze's syndrome has been mostly attributed to microtrauma until now; however, this etiology is currently disputed. The diagnosis is based on clinical findings, although a few studies suggest Case Report the advantages of imaging. We report a case of Tietze's syndrome with a review of radiological findings, especially magnetic resonance imaging (MRI) with dynamic contrast enhancement.

Received: December 15, 2019 Revised: January 16, 2020 Keywords: Tietze’s syndrome; diseases; Magnetic resonance imaging; Accepted: February 17, 2020 Dynamic contrast enhancement

Correspondence to: Sang Yoon Kim, M.D. Department of Radiology, Dankook University Hospital, 201, INTRODUCTION Manghyang-ro, Dongnam-gu, Cheonan 31116, Korea. Tietze’s syndrome is an inflammatory condition of unknown etiology characterized by Tel. +82-41-550-6921 , non-suppurative , and swelling of costochondral, costosternal, Fax. +82-41-552-9674 and sternoclavicular joints (1, 2). In most cases (80%), only a single is E-mail: [email protected] involved, especially the second or the third ; however, other joints may be involved concurrently (1). The etiopathogenesis of Tietze’s syndrome is often explained by the theory of microtrauma, which is still disputed (2). The histological examination of Tietze’s syndrome reveals significant pathological changes involving costal cartilage, including increased vascularity and degenerative changes associated with patchy loss This is an Open Access article distributed of ground substance resulting in a fibrillary appearance (3). The diagnosis of Tietze’s under the terms of the Creative Commons syndrome is based on clinical findings. Multiple imaging modalities have been proposed Attribution Non-Commercial License (http://creativecommons.org/licenses/ to diagnose this condition; however, few studies investigated the role of conventional by-nc/4.0/) which permits unrestricted radiography, computed tomography (CT), ultrasonography (US), bone scan, positron non-commercial use, distribution, and reproduction in any medium, provided emission tomography (PET)-CT, and magnetic resonance imaging (MRI). Here, we the original work is properly cited. present a female patient with Tietze's syndrome involving the left 3rd and right 4th costochondral joints. We report the findings of Tietze’s syndrome based on various imaging modalities, especially MRI with dynamic contrast enhancement (DCE).

Copyright © 2020 Korean Society of Magnetic Resonance in Medicine (KSMRM) CASE REPORT

A 35-year-old female patient with right breast cancer was pathologically diagnosed

www.i-mri.org 55 DCE MRI of Tietze's Syndrome | Dong Chan Kim, et al. with an invasive carcinoma following lumpectomy. The enhancement ratio were computed based on the pre- patient underwent adjuvant, concurrent chemo-radiation contrast and five post-contrast phases. The time-signal therapy for right breast at 3 months after surgery. During intensity curves (TICs) of multiple post-contrast phases the follow-up, 99mTc-methylene diphosphonate (MDP) were constructed. The subchondral bone marrow and the bone scan was performed at 7 months post-surgery, which capsular component of the left 3rd costochondral joint showed focal MDP uptake in the left 3rd and right 4th exhibited an early strong enhancement with a steep slope . The physician at the Department of Nuclear Medicine and a maximum, followed by a transition to a stable level or suspected that the lesions were bone metastases (Fig. 1). a slight decrease in signal intensity. The subchondral bone Chest CT (CT) scan was performed on the same day. marrow of the contralateral costochondral joint showed The CT showed enlargement of subchondral bone with relatively early enhancement, followed by a rapid wash-out sclerotic changes in the left 3rd and right 4th ribs (Fig. 2). phase (Fig. 3c). Morphologically, the enhancing portion of The enhanced scan revealed uncertain enhancement of the the periarticular capsular component eventually thickened costochondral lesions. These findings were not adequate to after gadolinium injection (Fig. 3d, e). confirm the bone metastases of breast cancer. Furthermore, a comparison with a CT scan performed at 3 months after surgery, which was conducted for the planned post- operative radiotherapy, revealed no significant changes between the findings of the costochondral lesions. A review of the patient's images revealed that these lesions were also scanned in the breast MRI, which was conducted 1 week before the operation. The MR images showed enlarged costal cartilage and the subchondral bone of the rib, similar to the findings on the CT, as well as subchondral bone marrow edema on the T2-weighted image (Fig. 3a). Enhanced T1-weighted image showed homogeneous enhancement of the subchondral bone marrow and capsular component of the costochondral joint. The DCE images were included in the breast MRI protocol, and thus were available for analysis. During the dynamic scan, the five phases were scanned continuously in a total scan time of 5 min. Regions of interest (ROIs) were manually placed in the axial slices, subchondral bone marrow, and the capsular component of the left 3rd costochondral joint (Fig. 3b). For comparison, the subchondral bone marrow of the contralateral costochondral joint was also selected Fig. 1. The 99mTc-MDP bone scan shows a focal increase in as an ROI. The kinetic variables including wash-in slope, MDP uptake at the anterior end of left 3rd rib (arrow) and wash-out slope, peak enhancement, and maximum relative the right 4th rib (open arrow) on the anterior static image.

Fig. 2. Axial CT images (bone window setting) show enlargement of subchondral bone with sclerotic changes at the anterior end of the (a) right 4th (open arrow) and (b) left 3rd (arrow) ribs. a b

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Moreover, the PET-CT was also performed at 1 (SUV) was not significantly high. week, preoperatively. The PET-CT revealed a mild US was performed for further evaluation. The patient fluorodeoxyglucose (FDG) uptake at the costochondral showed a sonographic probe-induced tenderness in junctions (Fig. 4). The value of standardized uptake value the affected lesions, and a chronic clinical history of

a b

c d

Fig. 3. Conventional MRI combined with dynamic contrast-enhanced images and kinetic curves. MR fat-suppressed T2-weighted images (a) show enlargement of subchondral bone with bone marrow edema in the anterior end of left 3rd rib (arrow). Regions of interest (b) are placed in the subchondral bone marrow and the capsular component of the left 3rd costochondral joint to calculate the kinetic variables in the DCE-MRI. Kinetic curves (c) of the capsular component (◆) and the subchondral bone marrow (■) suggest an early steep and strong enhancement, followed by a transition to a stable level or a slight e decline. In contrast, the kinetic curve in the subchondral bone marrow of the contralateral joint (▲) shows early enhancement followed by a rapid wash-out phase. Precontrast axial T1-weighted fat- suppressed images (d) and the 5-min delayed enhanced T1-weighted fat-suppressed images (e) show thick enhancement at the capsular component of left 3rd costochondral joint (arrows). www.i-mri.org 57 DCE MRI of Tietze's Syndrome | Dong Chan Kim, et al.

Fig. 4. 18F-FDG PET/CT images reveal mild FDG uptake in the (a) right 4th costochondral junction (open arrow) and (b) left 3rd costochondral junction (arrow). The value of SUVmax was not significantly high (Left SUV max value, 2.1; Right SUV max value, 1.9). a b

a b

c d Fig. 5. Ultrasound images show enlargement of the costal cartilage and the end of rib in the (b) left 3rd costochondral joint (arrow) and (c) right 4th costochondral joint (open arrow) compared with the (a) right 3rd and (d) left 4th costochondral joints.

58 www.i-mri.org https://doi.org/10.13104/imri.2020.24.1.55 intermittent pain and tenderness involving those joints. of hyaline cartilage was not significant in our patient. The US showed enlarged costal cartilage and subchondral The US investigation of Tietze’s syndrome is rapid, but the bone at the left 3rd and right 4th costochondral junctions. findings are highly dependent on the operator’s skill. However, there was no significant change in echogenicity of Third, the bone scans show increased radioactive uptake the affected cartilage (Fig. 5). in the costochondral joint in Tietze’s syndrome. In general, A review of all the imaging studies suggested chronic bone scans are considered highly sensitive but nonspecific inflammatory lesions, with a low possibility of malignancy. indicators of Tietze's syndrome, due to similar findings in A diagnosis of Tietze's syndrome was established based on various benign and malignant bone and joint disorders (7). the chronic intermittent symptoms, characteristic location, Next, the PET-CT findings of Tietze's syndrome are rarely and various imaging findings showing long-standing reported. The PET-CT scan usually shows a mild FDG uptake inflammatory features. The patient was not treated of the affected costochondral joint; however, occasionally it for Tietze’s syndrome due to the absence of significant may show intense uptake and mimic a malignant lesion (8). symptoms at the time. However, the patient’s breast cancer Finally, the MRI findings of our patient were similar was subsequently treated with concurrent chemo-radiation to those reported previously by Volterrani et al. (2): therapy. enlargement of costal cartilage and subchondral bone of the rib, focal or widespread edema of cartilage and subchondral bone, gadolinium uptake of cartilage, subchondral bone DISCUSSION and periarticular capsular component, except for poor enhancement of the costal cartilage. It was probably due Tietze’s syndrome is defined as a self-limiting disorder of to the relatively low vascularity of the costal cartilage unknown etiology. It is characterized by non-suppurative compared with the subchondral bone. painful swelling of the costochondral, costosternal, or We had a good opportunity to analysis DCE MRI findings sternoclavicular joints. The etiology of Tietze's syndrome of Tietze's syndrome. In DCE MRI image, the affected is still disputed, although it has been mostly attributed subchondral bone marrow and the capsular component to microtrauma until now. The costochondral joint is a exhibited an early stronger enhancement with a steep slope, hyaline that is immobile normally and and slower wash-out of contrast media than the normal has no synovial capsule. It is avascular and nourished only bone marrow. It is presumed that the rapid and intense via vascular supply to the tightly adherent perichondrium enhancement of the lesion is a result of angiogenesis and periosteum. The histological examination of the caused by mechanical stress-induced proliferative activity costochondral joint in Tietze’s syndrome generally shows of Tietze's syndrome. Although only a few studies reported hypervascularization and degenerative changes associated DCE image findings of the articular disease, a few studies with partial calcification of the cartilage, or peripheral reported the use of DCE in synovial inflammatory arthritis, hypertrophic changes (4). such as rheumatoid arthritis and psoriatic arthritis (9, 10). The diagnosis of the syndrome is mainly based on clinical van der Leij et al. (10) reported that even though most of findings. However, the differential diagnosis of Tietze’s TIC shape of synovial tissue in inflammatory arthritis on syndrome including various articular diseases is usually DCE study showed a ‘continuously slow enhancement’ difficult because of ambiguous clinical presentations. type, the second largest number of cases still revealed ‘fast Therefore, the diagnosis of Tietze’s syndrome has been enhancement followed by washout phase’, similar to our attempted using different imaging modalities. case study. Schwenzer et al. (9) also reported strong early First, CT generally shows chronic features such as enhancement followed by a slower wash-out of contrast subchondral sclerosis, changes in cortical contour of the rib, medium in synovial inflammatory arthritis. The slower and calcification of costal cartilage. However, the CT is less wash-out rate has been attributed to higher cellularity, and sensitive because it is difficult to detect early inflammatory thickening of small arteries and capillaries in the inflamed changes of bone and joint (5). synovium. Although a direct comparison is difficult, the Second, according to Martino et al. (6), the US reveals peak contrast enhancement might be lower and the wash- an increase in the echogenicity of the affected hyaline out rate might be steeper in the observed cartilage because of hypervascularization and calcification in Tietze's syndrome compared with synovial inflammatory of the cartilage (6). However, the change in the echogenicity arthritis, because the costochondral joints are poorly www.i-mri.org 59 DCE MRI of Tietze's Syndrome | Dong Chan Kim, et al. vascularized with a lower cellularity of the inflamed M, Fioravanti A. Magnetic resonance imaging in Tietze's perivascular tissue. syndrome. Clin Exp Rheumatol 2008;26:848-853 There is no broadly accepted treatment for Tietze’s 3. Cameron HU, Fornasier VL. Tietze's disease. J Clin Pathol syndrome; the condition is usually self-limited and 1974;27:960-962 typically resolves within a few months. If required, local 4. Aeschlimann A, Kahn MF. Tietze's syndrome: a critical steroid injections, NSAIDs (nonsteroidal anti-inflammatory review. Clin Exp Rheumatol 1990;8:407-412 drugs), minimization of physical activity, and application 5. Edelstein G, Levitt RG, Slaker DP, Murphy WA. Computed of local heat are adequate treatments. However, symptom tomography of Tietze syndrome. J Comput Assist Tomogr recurrence is common in many patients. 1984;8:20-23 In conclusion, Tietze’s syndrome is a rare and usually 6. Martino F, D'Amore M, Angelelli G, Macarini L, Cantatore FP. Echographic study of Tietze's syndrome. Clin Rheumatol underestimated disease, and the diagnosis is usually based 1991;10:2-4 on clinical examination. However, various imaging studies 7. Hoffer PB, Genant HK. Radionuclide joint imaging. Semin provide insight into the diagnosis, together with a detailed Nucl Med 1976;6:121-137 patient history. The DCE MRI study in conjunction with CT, 8. Oh JH, Park SB, Oh HC. 18F-FDG PET/CT and bone US, scintigraphy, and PET-CT facilitates the clinical diagnosis scintigraphy findings in Tietze syndrome. Clin Nucl Med of Tietze’s syndrome. 2018;43:832-834 9. Schwenzer NF, Kotter I, Henes JC, et al. The role of dynamic contrast-enhanced MRI in the differential diagnosis of REFERENCES psoriatic and rheumatoid arthritis. AJR Am J Roentgenol 2010;194:715-720 1. 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