An unusual case of extensive contiguous cervicothoracic spinal tuberculosis involving fourteen damaged segments: A case report

Highlights • Cervicothoracic spinal tuberculosis (CTSTB) is a rare and disabling disease involving the cervicothoracic junction.• Approximately half of these cases involves one or two segments of cervicothoracic vertebrae.• We reported a 28-year-old female with tuberculous involvement of fourteen contiguous vertebral segments.


Introduction
Cervicothoracic spinal tuberculosis (CTSTB), defined by tuberculosis infection involving the vertebrae between C7 to T3, is a rare disease that typically results in disabling complications such as kyphotic deformity, large paravertebral abscesses, and progressive spinal cord damage with severe neurological deficit [1][2][3]. This disease constitutes only 5% of all cases of spinal tuberculosis [4], and it often presents as a treatment challenge for spine surgeons. The cervicothoracic junction is anatomically located in the transitional zone between the more mobile, lordotic cervical and the more rigid, kyphotic thoracic spine [1]. Since the junction is weight-bearing structure, destruction of such structure by tuberculosis infection not uncommonly results in numerous complications aforementioned above. Moreover, the unique interrelationship of cervicothoracic vertebrae commands difficult exposure of the * Corresponding author.
E-mail address: totosuryoefar@gmail.com (I. Saleh). infection focus, both anteriorly and posteriorly, making operative treatment a considerable challenge [1]. Approximately half of cervical spinal tuberculosis involves at most two segments. In this report, we present a case of 28-year-old female diagnosed with CTSTB with involvement of contiguous fourteen vertebrae.

SCARE criteria compliance
This work has been reported in line with the SCARE criteria [5].

Case report
A 28-year-old female presented to Fatmawati Hospital, Jakarta, Indonesia due to progressive tetraparesis accounting for seven months. Initially there was mild neck pain, and then she experienced numbness along with weakness of all four extremities. The symptoms were gradually worsened such that at the time of admission, she could not walk.
A series of radiographic and CT scan depicted multiple vertebral body destruction anteriorly, along with facet joint dislocation   and mild retrolisthesis of C4-C5 segments (Figs. 1, 2). MR images of the cervical region was demonstrated pathologic contrast enhancement on C4 to T7 vertebrae, a total of fourteen contiguous segments, with spinal canal stenosis on the level of C4 to T4 and bilateral anterolateral paravertebral soft tissue abscess at the level of C4 to T9 (Fig. 3). Those findings were highly typical of spinal tuberculosis.
The patient was administered anti-tuberculous agents for two months before undergoing one-staged posterior-only debridement, decompression, and instrumentation. Postoperative radiographs demonstrated that the kyphosis was obviously improved (Fig. 4). After the surgery, the patient was put on a cervicothoracic brace. The patient was closely followed up, and after 12 months postoperatively, she regained neurological recovery with only mild residual neck pain. The patient demonstrated a satis-fying level of functional improvement as recorded by the Neck Disability Index (NDI) and SF-36 scores of 4/100 and 94%, respectively.
Bony bridge on CT images was discovered at 18 months of follow-up, along with the normal value of erythrocyte sedimentation rate and C-reactive protein; it was the time we discontinued the anti-tuberculous therapy.

Discussion
Tuberculosis, caused by Mycobacterium tuberculosis, remains one of the oldest diseases worldwide. Despite an ancient disease, tuberculosis remains an important problem, particularly in underdeveloped countries. Extra-pulmonary tuberculosis accounts for 15-20% of all tuberculosis cases [6], and the most common form of this spectrum is spinal tuberculosis, which constitutes of 50% of all skeletal tuberculosis cases [7]. Spinal tuberculosis remains a great challenge to physicians do its nonspecific symptoms that may result in delay of diagnosis, as well as significant high morbidity and mortality.
Of all spinal tuberculosis, CTSTB accounts for only 5% [4]. In addition to its rarity as a site for tuberculosis, the cervicothoracic junction has anatomical and clinical peculiarities, as a reversal of the mobile-lordotic cervical vertebrae to rigid-kyphotic thoracic vertebrae occurs at this location [8][9][10]. In fact, of all regions of the spine, the cervicothoracic junction is arguably the most challenging entity [11]. Moreover, such junction presumes specific biomechanics and stability different from other spinal regions [9,12]. Affecting mainly to the anterior column, contiguous tuberculous lesion of cervicothoracic junction leads to profound instability and altered biomechanics of the weight-bearing area. CTSTB was also associated with high degree of spinal cord compression that invariably leads to neurological deficit [8].
Most CTSTB involves only two segments; however, in this case, we found a very extensive case wherein there were fourteen damaged segments. In a series of 20 patients with CTSTB, Lan et al. [13] found that 11 (55%) of the patients had two damaged segments,  and the most extensive case only involved three segments, which only occurred in one (5%) subject. In a case series of 10 patients, 6 (60%) subjects had three damaged segments, and the most extensive case involved 8 segments. To date, there is no published report regarding CTSTB involving more than ten segments; this is the first report that reports CTSTB involving fourteen damaged segments.
At present, CTSTB is still rarely reported; thus, there has been no specific concensus, let alone for the extensive one. Indications for surgery in CTSTB include kyphosis of ≥ 20 • , instability, neurological compromise, and persistent pain [14]. CTSTB could be managed with anterior-only debridement with or without instrumentation, and combined anterior debridement followed by posterior instrumentation. In this case report, we performed one-stage posterior transpedicular debridement, decompression, and instrumentation. Several authors recommended the anterior debridement with or without instrumentation for the main treatment of CTSTB due to its access to tuberculous lesion and its paravertebral abscess, which occurs anteriorly [1,15,16]. However, it is often considered difficult due to the complex structure of the cervicothoracic junction. Many structures, including thoracic bones, clavicles, costal bone, large blood vessels, etc, cover the region [1,9]; thus, the operative field is narrow, which presents a significant challenge to the spine surgeon [17]. Recently, the posterior-only surgery has garnered interests as an alternative treatment for CTSTB, as it is simple, labelled with good clinical efficacy and few complications [9,18]. Feyza et al. [19] and Rath et al. [20] reported good neurological results after posterior debridement and internal fixation in those with neurological impairment due to CTSTB. The results were similar to those obtained via anterior approach. Zeng et al. [9] found that posterior instrumentation may provide better kyphotic correction and is beneficial to the stress dispersion, which can effectively prevent implant failure. Several published studies regarding the management of CTSTB are presented in Table 1.  In addition to posterior procedures, strut graft or mesh cage could be inserted anteriorly to provide additional stability, especially in our patient whose spinal biomechanics was heavily disrupted from destruction of multiple vertebral segments anteriorly. However, as the risk of graft or cage failure increased along with the increasing number of involved segments, we decided not to perform any procedure anteriorly. To provide additional stability, we put the patient on external brace for six months, afterwards the brace was removed gradually until the involved segments regained bony fusion.

Conclusions
Our report demonstrates one of the longest involvement of extensive contiguous CTSTB who was treated with one-stage posterior-only approach. However, as this is only a report of one case, further studies are required to investigate the safety and efficacy of such approach for treating extensive CTSTB.

Sources of funding
None declared.

Ethical approval
Ethical approval has been received from Fatmawati Hospital, Jakarta, Indonesia.

Consent
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Author contribution
Ifran Saleh: performing the procedure, study concept, providing revisions to scientific content of the manuscript.
Didik Librianto: performing the procedure, data collection, providing stylistic/grammatical revisions to the manuscript.
Phedy Phedy: performing the procedure, data analysis, writing the paper.

Registration of research studies
This study has been registered at researchregistry.com (UIN: researchregistry5169).

Provenance and peer review
Not commissioned, externally peer-reviewed.

Declaration of Competing Interest
None.