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CASE REPORT  
Year : 2019  |  Volume : 53  |  Issue : 4  |  Page : 574-577
Refractory tietze's syndrome occurring after lumbar spine surgery in prone position


Mallika Spine Centre, Guntur, Andhra Pradesh, India

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Date of Web Publication17-Jun-2019
 

   Abstract 

Positional complications in spine surgery are not uncommon. Commonly encountered complications include ocular and aural, other than musculoskeletal injuries. However, development of Tietze's syndrome due to malpositioning has not been reported till date. A 40-year-old male patient presented with postlaminectomy syndrome, for which posterior pedicle screw fixation and fusion was performed. Postoperatively, patient complained of new-onset pain associated with redness and swelling at parasternal region. After thorough radiological investigations, he was diagnosed with Tietze's syndrome at 6th and 7th costo-cartilaginous junction. Tietze's syndrome is itself a rare entity, and its association with malpositioning during prone positioning is uncommon. It is important for the surgeons to be aware of the condition as Tietze's syndrome may be encountered as an off-centered complication due to malpositioning.

Keywords: Chest wall insult, positional complication, prone position, Tietze's syndrome

How to cite this article:
Kumar VA, Babu J N. Refractory tietze's syndrome occurring after lumbar spine surgery in prone position. Indian J Orthop 2019;53:574-7

How to cite this URL:
Kumar VA, Babu J N. Refractory tietze's syndrome occurring after lumbar spine surgery in prone position. Indian J Orthop [serial online] 2019 [cited 2019 Jul 18];53:574-7. Available from: http://www.ijoonline.com/text.asp?2019/53/4/574/257495



   Introduction Top


Majority of spine cases, especially lumbar surgeries are done through posterior approach where the patient has to be in prone position for longer durations. The positional complications are higher if the patient is under general anesthesia. Ocular and aural complications are very commonly reported in the literature, one of the reasons being improper positioning of the patient during anesthesia.

Tietze's syndrome is a condition of unknown etiology characterized by painful nonsuppurative swelling over costal cartilages. It was first described by Alexander Tietze in 1921.[1],[2],[3] It is most commonly seen in young individuals and is equally prevalent among both sexes.[4],[5] This is one of the rarest diseases listed by National Institutes of Health,[6] indicating its limited occurrence in the region. It commonly involves 2nd and 3rd costochondral junctions leading to chronic pain, but is usually self-limited. There were reports to state it can easily be misdiagnosed with malignancies, pneumonia, acute coronary syndrome, etc.[7],[8] This condition is differentiated from costochondritis with the presence of tender swelling at the coastal cartilaginous area.

Here, we report a refractory Tietze's syndrome developed few months postoperatively after lumbar fixation with pedicle screw system due to malpositioning during the surgery.


   Case Report Top


Special history

A 40-year-old male patient (body mass index: 22.5) presented to our outpatient clinic with symptoms of postlaminectomy syndrome. After thorough clinical and radiological examination, he was diagnosed with L4–L5 instability with L4–S1 lumbar canal stenosis. After exhaustion of conservative management, he was advised posterior stabilization, fusion, and decompression. He successfully underwent the revision procedure where L4–S1 pedicle screw fixation with L5–S1 transforaminal lumbar interbody fusion was done.

Surgery

After intubation and prone positioning, all bony landmarks (elbows and knees) were padded. For positional support, two soft cushion bolsters are used, one below chest wall and one below pelvis. Repositioning of the head was done regularly to avoid pressures on ocular bulbs. Surgery procedure took 3 hours and was uneventful. The patient was placed back to supine position and extubated. The patient showed no signs of any neurological abnormality.

Postoperative sequel

On 2nd postoperative day, the patient complained of right-sided parasternal chest pain which was tender on examination. It was correlating to the area covered by proximal bolster placed during prone positioning. Pain was controlled with anti-inflammatory medications and the rest of the period was uneventful till suture removal. After 15 days, he started complaining of similar pain at the same point on right side of the chest. On examination, there was swelling, redness, tenderness, and local rise of temperature at the site [Figure 1]. His blood investigations showed increased erythrocyte sedimentation rate and C-reactive protein. Oral antibiotics and anti-inflammatory agents were given for a period of 5 days where pain started to come under control and he was discharged.
Figure 1: Clinical picture showing redness and inflammation on anterior aspect of the chest wall

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Further sequel

Later after 1 month, he returned back with the same complaint and with equal intensity. Then clinically, the diagnosis of Tietze's syndrome of lower ribs was made and has been advised for further investigations. Ultrasound of the area showed edematous coastal cartilage in the lower ribs with increased thickness of about 2.3 cm (normal 1.3 cm). Non-contrast computed tomography (NCCT) study of thorax was done to rule out other differential diagnoses [Figure 2]. Magnetic resonance imaging (MRI) showed thickening and enlargement of coastal cartilage at 6th and 7th costo-cartilaginous junction [Figure 3]. In delayed imaging phase of bone scintigraphy, there was avid uptake of radiotracer noted in the 6th and 7th costo-cartilaginous junctions on the right side. All investigations came with the positive correlation of the diagnosis made and was confirmed with Tietze's syndrome of the 6th and 7th ribs. Even after being on nonsteroidal anti-inflammatory drugs for 3 weeks, pain did not subside. Thereby, surgical excision of the 6th and 7th ribs' cartilage was performed followed by tumor necrosis factor inhibitors (adalimumab) for 6 months. He recovered completely with visual analog scale 0/10 over a period of 9 months.
Figure 2: Noncontrast computed tomography of the thorax showing the presence of edema and induration around the right costal cartilage at the 6th rib

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Figure 3: Magnetic resonance imaging dorsal spine showing enlargement and thickening of coastal cartilage with hyperintensity noted at the 6th and 7th costochondral junctions

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   Discussion Top


Positional complications in spine surgeries are not uncommon. The most common one is posterior ischemic optic neuropathy that may either result due to improper padding of the eye causing a direct pressure effect when the patient is under general anesthesia or due to blood loss, hypotension, and anemia which all three may conspire to produce the condition.[9],[10] In a study by Chang et al.,[11] the incidence of ocular complications was a mere 0.028%. This, however, is a total nil if the surgery is being performed under spinal anesthesia.[12] Other complications include upper and lower limb injuries due to inadequate padding of the pressure points, neck pain, and stiffness and brachial plexus injury. Oropharyngeal swelling and macroglossia are reported after cervical spine surgery in prone position.[13]

The exact cause of Tietze's syndrome is still unknown (idiopathic); however, few researchers have speculated that multiple microtrauma to the anterior chest wall may lead to the development of Tietze's syndrome.[14],[15],[16] Tietze's syndrome differentiates itself from costochondritis by the presence of reddish, tender swelling at the costochondral margin, making it the hallmark of the disease and also indicating its severity.[14] It is mostly seen in the upper ribs, but very rarely reported in lower ribs. The incidence of Tietze's syndrome is not well studied, and, hence, only few clinicians have a handful of experience with it. Many studies contradicted each other regarding the sex distribution of the disease which has a wide range from equal to twice common in females to males.[15],[16] The rarity of the disease and the location of the pain often lead to differential diagnosis. Wide variety of conditions can cause symptoms similar to Tietze's syndrome, which includes irritation of nerve root from upper thoracic spine, fracture of rib, seronegative arthritis, ankylosing spondylitis, fibromyalgia, pneumonia, and coronary heart disease. Hence, it is always important to clinically exclude cardiac, gastrointestinal, and pulmonary origins of pain before confirming the diagnosis.[17]

In the recent past, imaging has become an important diagnostic tool for the clinicians. However, in case of Tietze's syndrome, simple imaging of plain radiograph is often misleading as it is normal in almost all instances. Ultrasound of the area may be useful in visualizing the edema. Bone scintigraphy is an effective diagnostic tool for localizing the pathology in cases of costochondral pain.[18] CT is also useful in these cases where the findings are chondral enlargement and destruction with/without calcification of costal cartilage.[19] Although we find MRI not to be the diagnostic investigation of choice in this case, there are articles supporting MRI as a decisive investigation for Tietze's syndrome.[20]

Tietze's syndrome which is a self-limiting chronic pain condition is itself a rare entity. Few case reports about variants of Tietze's syndrome have been mentioned in the literature.[21],[22],[23],[24],[25] However, as per the author's knowledge, occurrence of Tietze's syndrome following surgery in prone position is a rarest complication which is not reported till date. Surgeons should be aware of this complication and try to prevent such incidents through proper cushioning at the dependent areas during prone positioning.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Hiramuro-Shoji F, Wirth MA, Rockwood CA Jr. Atraumatic conditions of the sternoclavicular joint. J Shoulder Elbow Surg 2003;12:79-88.  Back to cited text no. 1
    
2.
Wolf E, Stern S. Costosternal syndrome: Its frequency and importance in differential diagnosis of coronary heart disease. Arch Intern Med 1976;136:189-91.  Back to cited text no. 2
    
3.
Tietze A. Über eine eigenartige Häufung von Fällen mit Dystrophie der Rippenknorpel. [About a peculiar accumulation of cases with dystrophy of costal cartilage] Berliner klinische Wochenschrift. Volume v. 58:2 1921. pp 829-31.  Back to cited text no. 3
    
4.
Store SD. Tietze's syndrome. J Postgrad Med 1964;10:28-30.  Back to cited text no. 4
    
5.
Jelenko C 3rd. Tietze's syndrome at the xiphisternal joint. South Med J 1974;67:818-20.  Back to cited text no. 5
    
6.
National Institutes of Health. Rare Diseases and Related Terms. National Institutes of Health; 2013. Available from: https://rarediseases.info.nih.gov/diseases/10100/tietze-syndrome. [Last accessed on 2018 Nov 04].  Back to cited text no. 6
    
7.
Jelenko C 3rd, Cowan GS Jr. Perichondritis (Tietze's syndrome) at the xiphisternal joint: A mimic of severe disease. JACEP 1977;6:536-42.  Back to cited text no. 7
    
8.
Epstein SE, Gerber LH, Borer JS. Chest wall syndrome. A common cause of unexplained cardiac pain. JAMA 1979;241:2793-7.  Back to cited text no. 8
    
9.
Edgcombe H, Carter K, Yarrow S. Anaesthesia in the prone position. Br J Anaesth 2008;100:165-83.  Back to cited text no. 9
    
10.
Lee LA, Roth S, Posner KL, Cheney FW, Caplan RA, Newman NJ, et al. The American society of anesthesiologists postoperative visual loss registry: Analysis of 93 spine surgery cases with postoperative visual loss. Anesthesiology 2006;105:652-9.  Back to cited text no. 10
    
11.
Chang SH, Miller NR. The incidence of vision loss due to perioperative ischemic optic neuropathy associated with spine surgery: The Johns Hopkins hospital experience. Spine (Phila Pa 1976) 2005;30:1299-302.  Back to cited text no. 11
    
12.
Tetzlaff JE, Dilger JA, Kodsy M, al-Bataineh J, Yoon HJ, Bell GR, et al. Spinal anesthesia for elective lumbar spine surgery. J Clin Anesth 1998;10:666-9.  Back to cited text no. 12
    
13.
Sinha A, Agarwal A, Gaur A, Pandey CK. Oropharyngeal swelling and macroglossia after cervical spine surgery in the prone position. J Neurosurg Anesthesiol 2001;13:237-9.  Back to cited text no. 13
    
14.
Stochkendahl MJ, Christensen HW. Chest pain in focal musculoskeletal disorders. Med Clin North Am 2010;94:259-73.  Back to cited text no. 14
    
15.
Aeschlimann A, Kahn MF. Tietze's syndrome: A critical review. Clin Exp Rheumatol 1990;8:407-12.  Back to cited text no. 15
    
16.
Volterrani L, Mazzei MA, Giordano N, Nuti R, Galeazzi M, Fioravanti A, et al. Magnetic resonance imaging in Tietze's syndrome. Clin Exp Rheumatol 2008;26:848-53.  Back to cited text no. 16
    
17.
Gijsbers E, Knaap SF. Clinical presentation and chiropractic treatment of tietze syndrome: A 34-year-old female with left-sided chest pain. J Chiropr Med 2011;10:60-3.  Back to cited text no. 17
    
18.
Koç ZP, Balcı TA, Ozyurtkan MO. The role of the three phase bone scintigraphy in the management of the patients with costochondral pain. Mol Imaging Radionucl Ther 2013;22:90-3.  Back to cited text no. 18
    
19.
Edelstein G, Levitt RG, Slaker DP, Murphy WA. Computed tomography of Tietze syndrome. J Comput Assist Tomogr 1984;8:20-3.  Back to cited text no. 19
    
20.
De Filippo M, Albini A, Castaldi V, Monaco D, Sverzellati N, Carbognani P, Rusca M, Rindi G, Zompatori M. MRI findings of Tietze's syndrome mimicking mediastinal malignancy on MDCT. European Journal of Radiology Extra, 65, 33–35. https://doi.org/10.1016/J.EJREX.2007.10.006.  Back to cited text no. 20
    
21.
Carabasi RJ, Christian JJ, Brindley HH. Costosternal chondrodynia: A variant of Tietze's syndrome? Dis Chest 1962;41:559-62.  Back to cited text no. 21
    
22.
van Schalkwyk AJ, van Wingerden JJ. A variant of Tietze's syndrome occurring after reconstructive breast surgery. Aesthetic Plast Surg 1998;22:430-2.  Back to cited text no. 22
    
23.
Matthews JC. Chest pseudoangina due to costal chondrodynia. Tietze's syndrome. Prensa Med Argent 1963;50:3006-10.  Back to cited text no. 23
    
24.
Shambrom E, Feher A. Tietze's syndrome; report of a case with involvement of the cricoarytenoid joint. AMA Arch Intern Med 1955;96:697-9.  Back to cited text no. 24
    
25.
Valesová M, Hanus J. Tietze's syndrome as a manifestation of the metastasis of Grawitz's adenocarcinoma. Fysiatr Revmatol Vestn 1984;62:88-90.  Back to cited text no. 25
    

Top
Correspondence Address:
Dr. Viswanadha Arun Kumar
Mallika Spine Centre, 12-12-30, Old Club Road, Kothapet, Guntur - 522 001, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ortho.IJOrtho_276_18

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