Magnetic resonance imaging in Tietze’s syndrome L. Volterrani1, M.A. Mazzei2, N. Giordano3, R. Nuti3, M. Galeazzi4, A. Fioravanti4

1Department of Human Pathology and Oncology, 3Department of Internal Medicine, and 4Rheumatology Unit, Department of Clinical and Immunological Science, University of Siena, Siena, Italy; 2Radiologia Universitaria, Policlinico Santa Maria alle Scotte, Azienda Ospedaliera Universitaria Senese, Siena, Italy.

Abstract Objective To evaluate the usefulness of magnetic resonance imaging (MRI) in Tietze’s syndromesyndrome which,which, toto ourour knowledge,knowledge, hhasas nnotot previously been reported in the literature.

Methods Twelve consecutive outpatients with clinical features of Tietze’s syndromesyndrome underwentunderwent evaluation,evaluation, iincludingncluding tthehe aanamnesis,namnesis, clinical general examination, clinical evaluation of costosternal and sternoclavicular joints (SCJ) and biochemical and instrumental investigations. Twenty normal subjects age- and sex-matched to the patients’ groupgroup werewere examined in a similar manner. MRI of costosternal and SCJ was performed using a 1.5 Tesla unit (Gyroscan NT 1.5 Philips, The Netherlands and GE Signa Excite HD, GE Healthcare, Milwaukee, Wis., USA).

Results The MRI pattern of primary Tietze’s syndromesyndrome waswas characterizedcharacterized asas follows:follows: enlargementenlargement aandnd tthickeninghickening ooff ccartilageartilage aatt the site of complaint (12/12 patients); focal or widespread increased signal intensities of affected cartilage on both TSE T2-weighted and STIR or FAT SAT images (10/12 patients); bone marrow oedema in the subcondral bone (5/12 patients); vivid gadolinium uptake in the areas of thickened cartilage, in the subcondral bone marrow and/or in capsule and ligaments (10/12, 4/12 and 7/12 patients respectively).

Conclusion Magnetic resonance is an excellent technique to evidence both the cartilage and bone abnormalities, therefore it represents the elective method in the investigation of primary Tietze’s syndrome,syndrome, duedue toto itsits highhigh sensitivity,sensitivity, ddiagnosticiagnostic rreliabilityeliability aandnd biological advantages thanks to the lack of ionizing radiation.

Key words Tietze’s syndrome, magnetic resonance imaging, diagnosis, costosternal joints, sternoclavicular joints.

Clinical and Experimental 2008; 26: 848-853. MRI in Tietze’s sindrome / L. Volterrani et al.

Luca Volterrani, MD, Prof. of Radiology Introduction to evaluate the usefulness of MR in the Maria Antonietta Mazzei, MD Named in 1921 after Alexander Tietze, diagnosis of Tietze’s syndrome which, Nicola Giordano, MD, Prof. of Rheumatology a German surgeon (1), Tietze’s syn- to our knowledge, has not previously Ranuccio Nuti, MD, Prof. of Int. Medicine drome, also known as , been reported in the literature. Mauro Galeazzi, MD, Prof. of Rheumatology Antonella Fioravanti, MD is an infl ammatory process involv- ing one or more of the costochondral Patients and methods Please address correspondence and reprint requests to: cartilages. Tietze’s syndrome can be Between July 2004 and December Dr. Maria Antonietta Mazzei, defi ned as a benign, painful, non-sup- 2006, 12 consecutive outpatients (ten Radiologia Universitaria, purative, with localised swelling of women and two men, mean age 56.08 Policlinico Santa Maria alle Scotte, the costosternal, costochondral, and/or years, age range 29 to 70 years) with Azienda Ospedaliera Universitaria Senese, sternoclavicular joints (SCJ) (Tietze’s Tietze’s syndrome were observed at the Viale Bracci 2, Siena 53100, Italy. area), in the absence of other causes Rheumatology Unit (A. F. and M. G.) E-mail: [email protected] which could be responsible for this dis- and the Department of Internal Medi- Received on July 26, 2007; accepted in order (1, 2). In most cases (80%), only cine (N.G. and R.N.) of the University revised form on March 19, 2008. one is involved (most of Siena. The demographic data and © CopyrightCopyright CLINICAL AND commonly the second or the third ), clinical characteristics of the patients EXPERIMENTAL RHEUMATOLOGY 2008.2008. but other joints can be affected simul- are summarized in Table I. The same taneously (2, 3). The exact occurrence rheumatologist performed patients’ of this condition is not well known. It evaluation, including the medical his- predominantly strikes subjects between tory, general examination and clinical twenty and fi fty years of age, even if evaluation of costosternal joints and cases in children and the elderly have SCJ. All the subjects examined had no been documented (3, 4). Moreover, it history of thoracic trauma, aggressive has been reported that females are di- exercise able to cause strain, prior up- agnosed with the disease more often per respiratory tract infection, or either than males by a 2:1 ratio (2). The ae- signs and/or symptoms of systemic dis- tiopathogenesis of Tietze’s syndrome ease. Furthermore, a series of labora- is still being debated: the micro-trau- tory exams including blood cell count, matic theory currently seems to be the erythrocyte sedimentation rate, C-re- most cited one (3, 5, 6). Histological active protein, serum uric acid, lactate examinations of the swellings showed dehydrogenase, creatine phosphoki- non-specifi c fi ndings in the hyaline nase (CPK), CPK-MB, troponin, rheu- cartilage, consisting of an increased matoid factor, anti-cyclic citrullinated vascularity and degenerative changes peptide antibodies and urinalysis were with patchy loss of ground substance performed. Bacterial, viral, and my- leading to a fi brillar appearance (7). cotic cultures of blood, sputum, urine Different rheumatic and non-rheumatic and stools were also collected, in order diseases can account for with or to exclude other pathologies. Finally, without swelling around Tietze’s area chest x-rays and electrocardiograms (8-15). The diagnosis of this condition (ECG) were recorded for all patients. is primarily clinical. Many radiologi- During the same period, 20 normal sub- cal techniques have been suggested to jects, age and sex matched, were exam- confi rm the diagnosis, but there are few ined in a similar manner. The study was studies on the value of conventional ra- approved by the Ethics Committee of diography (16, 17), computed tomog- the School of Medicine of the Univer- raphy (CT) (18, 19), scintigraphy with sity of Siena. All patients provided in- 67Ga and 99mTc diphosphonate (20, formed oral consent. In all the individ- 21), and ultrasound (US) (22-25). Mag- uals examined, an MR imaging of the netic resonance (MR) (26) is an excel- anterior thoracic wall was performed lent technique to show cartilaginous, by using a 1.5 Tesla unit (Gyroscan joints and bone abnormalities: it has NT 1.5 Philips, The Netherlands and been employed in cases of chest wall GE Signa Excite HD, GE Healthcare, pain following thoracic trauma (27), Milwaukee, Wis.) with a superfi cial (28), septic ar- and phased array detection receiving thritis and malignant tumours (26, 29), coil. Subjects were placed prone on the which may mimic Tietze’s syndrome. MRI table to reduce respiratory mo- Competing interests: none declared. The purpose of the present study was tion artefacts, arms alongside the body.

849 MRI in Tietze’s sindrome / L. Volterrani et al.

Table I. Demographic data and clinical features of patients with Tietze’s syndrome. T2-weighted images) compared with the normal bone signal intensity, and Patient Age (year)/sex Duration of Clinical fi ndings Tietze’s syndrome, capsular and ligament involvement. when fi rst examined The criterion for diagnosis of cartilage (years) enlargement used was represented by the thickened and bulbous aspect of the 1 70/F 2 Painful tender swelling of the right SCJ 2 62/F 0.6 Painful swelling of the right SCJ affected tissue compared to the oppo- 3 56/F 1.6 Painful swelling of either SCJ site normal side (18). The enhancement 4 62/F 3 Painful swelling at left fi rst costochondral junction characteristics of the abnormal tissue 5 64/F 1.5 Painful swelling of either SCJ after gadolinium-based contrast mate- 6 56/F 2.3 Painful swelling of the right SCJ rial injections were also evaluated. The 7 58/F 0.6 Painful swelling of the right SCJ fi nal decision regarding MR morphol- 8 42/M 0.8 Painful of the left third costochondral junction ogy as well as MR signal characteristics 9 58/F 0.8 Painful of the left SCJ and enhancement of the cartilaginous, 10 29/F 1 Painful swelling at right third costochondral junc- bone and joint components was done by tion 11 56/F 0.6 Painful tender swelling of the right SCJ consensus. 12 60/M 1.8 Painful swelling of the right second costochondral junction Results Biochemical parameters, chest x-rays In the pre-contrast examination, fast were taken for all patients examined and electrocardiogram appeared within spin-echo T1-weighted (FSE T1) on on the Gyroscan MR unit. The princi- the normal limits for both patients and axial or coronal planes, fast spin-echo pal data of the MR sequences used are control subjects; cultures also gave neg- T2-weighted (FSE T2) and T2 STIR reported in Table II. The MR images ative results in patients and controls. (short time inversion recovery) or FAT were evaluated by two experienced SAT (saturation) sequences on coronal musculoskeletal radiologists (L.V. and Normal subjects planes were chosen. For all sequences, M.A.M.). The following MR imag- Among the 20 normal subjects exam- slice thickness was 3-4 mm and slices ing fi ndings were evaluated: abnormal ined, 14 presented signs that are known were separated by 0.3-0.4 mm gaps. thickness and abnormal signal inten- to be “degenerative”: osteophytes, me- In all patients 0,2 ml gadolinium/kg sity (increased signal intensity on T2 niscal calcifi cation, calcifi cation of the body weight (Magnevist, Schering, STIR or FAT SAT weighted images) costal cartilage and bone sclerosis. In Berlin, Germany) was administered of the hyaline cartilage compared with order to help the reader to differentiate and dynamic acquisition with gradient the thickness and signal intensity of the between normal and pathological pat- echo sequences on coronal planes were controlateral cartilage, presence or ab- terns, MRI fi ndings of normal SCJ and conducted with the same TR and TE sence of cortical bone erosion, abnormal condrosternal joints in a young patient as the pre-contrast scans on GE Signa signal intensity of subcondral bone (de- are shown in Figure 1 (A-E). MR unit. T1-weighted gadolinium-en- creased signal intensity on T1-weighted The SCJ is formed by the sternal end hanced sequences with fat saturation images, increased signal intensity on of the clavicle, the clavicular notch of the manubrium sterni, and the cartilage Table II. Technical parameters of principal utilized MR sequences. of the fi rst rib. It is completely divided into two articular compartments by a Sequences TR (ms) TE (ms) TI (ms) Matrix Section Acquisition MR unit fi brocartilaginous disk. SCJ and cos- (pixel) thickness/ time (min) tosternal cartilage size may vary with gap (mm) body habitus and costal cartilage level, FSE* T1 756 9 256X512 3.00/0.3 5:54 Gyroscan even if the costal cartilage is normally weighted NT 1.5 T symmetrical in size and orientation at FSE T2 3500-4000 100-120 196X256 3.00/0.3 3:33 Gyroscan any single level (18, 19). MR imaging weighted NT 1.5 T showed the normal cartilage as an in- STIR** 1400 20 155 176X256 3.00/0.3 6:45 Gyroscan termediate signal intensity on FSE T1- NT 1.5 T weighted images and hypo-intense on FSE T1 600 10.3 384X256 4.00/0.4 5:15 GE Signa FSE T2-weighted, STIR and FAT SAT 1.5 T images without enhancement in post- T2 FAT 3560 101 384X256 4.00/0.4 2:58 GE Signa SAT*** 1.5 T contrast images. FSPGR**** 100 2-8 320X288 3.00/0.3 0.58 GE Signa The enhancement pattern of the peri- 1.5 T articular and intra-articular structures and surrounding bone after administra- * ** *** **** FSE: fast spin echo; STIR: short time inversion recovery; SAT: saturation; FSPGR: fast tion of gadolinium was gradual, pro- spoiled gradient echo. gressive and homogeneous.

850 MRI in Tietze’s sindrome / L. Volterrani et al.

Patients with Tietze’s syndromesyndrome The peculiar MR pattern in Tietze’s syndrome is shown in Figures 2 (A-C) and 3 (A-D). In all the subjects exam- ined, no mass was identifi ed at the site of complaint. The results obtained with MR are reported in Table III. In par- ticular, enlargement and thickening of the hyaline cartilage at the site of com- plaint was found in 12/12 patients, fo- cal or widespread oedema of cartilage was found in 10/12 patients, marrow oedema in the subcondral bone in 5/12 patients, vivid gadolinium uptake of the cartilage, the subcondral bone and capsular components in 10/12, 4/12 and 7/12 patients respectively. Both TSE T2-weighted and STIR or FAT SAT images showed thickened carti- lage with focal or widespread increased signal intensity. Bone marrow oedema was often noticeable in the subcondral bone of the involved cartilage; in ad- dition, in post-contrast MR images, vivid gadolinium uptake was present in the areas of thickened cartilage, in the subcondral bone marrow and/or in the other peri-articular components.

Fig. 1. 35-year-old woman without involvement of anterior chest wall. Images show normal MR Discussion appearance of the SCJ and condrosternal joints on coronal T1-weighted MR image (A) coronal T2- In the present study, we examined 12 weighted MR image (B). Coronal T2 STIR image (C) and normal MR appearance of the SCJ and of the condrosternal joints on axial T1- weighted MR image respectively (D and E). consecutive outpatients with clinical characteristics of Tietze’s syndrome, 10 females and 2 males, aged between 29 and 70 years. The fi ndings confi rm the data already found in the literature (2, 3) on the high prevalence of the disease in adults and in the female gender. As previously reported (2, 3), also in our cases Tietze’s syndrome was prevalent- ly monolateral (10/12) and more often affected the right (2/12) or the right SCJ (7/12), probably due to its micro- traumatic aetiology (3). Analysing the literature on Tietze’s syndrome, we not- ed that costosternal, costochondral and SCJ involvement, often neglected in the past, has recently been the object of a great deal of interest, probably due to recent reports of the anterior chest wall involvement in many rheumatic dis- eases, in particular in spondyloarthri- tis, psoriatic arthritis, sternoclavicular Fig. 2. 62-year-old woman with painful swelling at left fi rst condrosternal junction. , and SAPHO syndrome A. axial T1-weighted MR image shows enlargement of the left fi rst cartilage (black arrow). B and C, (8, 10, 14, 28, 30). coronal FSE T2 and STIR images, respectively, show oedema in the subcondral bone of the (black arrows). Tietze’s syndrome is included in the dif- ferential diagnosis of visceral anterior

851 MRI in Tietze’s sindrome / L. Volterrani et al.

in echogenicity and thickness of patho- logical cartilage compared to the oppo- site normal side. Furthermore, it shows in real time the topographic correspond- ence between the cartilage alterations and the painful swelling, increased by digital pressure. This technique, howev- er, has some limits as it does not allow an immediate comparison between the two sides; this is due mainly to technical reasons. In particular, the 7-12 MHz lin- ear scanner usually utilized is not wide enough to scan simultaneously the two sides so that the comparison is only pos- sible by fl anking the images obtained separately. Moreover, this technique depends strongly on the operator and it is not suitable for getting a clear image of the complex anatomy of the SCJ and condrosternal joints; the above-men- tioned joints’ anatomy is well shown Fig. 3. 70-year-old woman with painful tender swelling of the right SCJ. by CT and MR imaging. Edelstein et A and B coronal T2-weighted and T2 FAT SAT MR images show enlarged right SCJ cartilage (white al. (18) proved the usefulness of CT in arrow) with abnormal signal intensity (increased signal intensity on T2-weighted images) of the hya- the evaluation of six patients with clini- line cartilage (black arrows) and focal area of oedema in the subcondral bone of the sternal end of the right clavicle (white thin arrow). C and D axial T1-weighted images before and after intravenous ad- cal features of Tietze’s syndrome: the ministration of gadolinium, respectively, show enlarged right SCJ cartilage and contrast enhancement spectrum of CT fi ndings includes focal of affected cartilage and capsular components (black thin arrows). cartilage enlargement, ventral angula- tions of the costal cartilage and normal chest wall pain (31, 32). Many diagnos- to exclude bone lesions, but they are not anatomic features. The author (18) con- tic techniques have been used to inves- able to show cartilage damage. Scinti- cluded affi rming that CT is able to show tigate Tietze’s syndrome, but only a few graphy (19, 20) shows an abnormal ac- the costal cartilage, bone and adjacent studies have analysed and compared the cumulation of 67Gallium and 99mTc di- structures and to defi ne the characteris- suitability of radiography (16, 17), CT phosphonate at the level of the involved tics of the mass noted during the clini- (18, 19), scintigraphy (20, 21), and US joint, but this technique is un-specifi c cal exam. Even if MR is an excellent (22-25). In many cases, conventional and is unable to represent different com- technique for highlighting cartilage and radiological methods (standard x-rays, ponents of the examined joint. US (22- bone abnormalities (26, 28), its value conventional tomography) (16,17) help 25) shows an inhomogeneous increase in the diagnosis of Tietze’s syndrome

Table III. Pathological MR fi ndings.

Patient 1 2 3 4 5 6 7 8 9 10 11 12 Site of complaint right right either left fi rst either right right left third left right third right right second SCJ SCJ SCJ costochondral SCJ SCJ SCJ costochondral SCJ costochondral SCJ costochon- junction junction junction dral junction

Enlargement and thickening Y Y Y Y Y Y Y Y Y Y Y Y of hyaline cartilage (particular left side) Focal or widespread oedema – Y – Y Y Y Y Y Y Y Y Y of cartilage Bone marrow oedema in the Y Y Y Y – Y – – – – – – subcondral bone (left) Gadolinium uptake of cartilage Y Y Y low Y Y Y Y Y – Y Y Gadolinium uptake of Y Y Y Y – low – – – – – – subcondral bone Gadolinium uptake of capsular Y Y Y low Y Y Y – Y – – – components

Y= yes.

852 MRI in Tietze’s sindrome / L. Volterrani et al. has not been amply investigated. In pathologies. In conclusion, we recom- 18. EDELSTEIN G, LEVITT RG, SLAKER DP et al.: the present study, we have reported mend MR as the elective method in the Computed tomography of . J Comput Assist Tomogr 1984; 8: 20-3. the spectrum of MR fi ndings in twelve investigation of Tietze’s syndrome, due 19. EDELSTEIN G, LEVITT RG, SLAKER DP et al.: patients with clinical characteristics of to its high sensitivity, diagnostic relia- CT observation of rib abnormalities: spec- Tietze’s syndrome; to our knowledge bility and biological advantages thanks trum of fi ndings. J Comput Assist Tomogr similar studies are not present in litera- to the lack of ionizing radiation. 1985; 9: 65-72. 20. MILLER JH: Accumulation of gallium-67 in ture until now. MR images revealed a costochondritis. 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