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com/terms-and-conditions) on Wiley Online Library for rules of use; OA articles are governed by the applicable Creative Commons License
| |
Received: 27 July 2022    Revised: 22 January 2023    Accepted: 13 February 2023

DOI: 10.1002/ccr3.7053

CASE REPORT

Non-­contiguous multilevel spinal tuberculosis: A case

report of unusual spinal tuberculosis resembling spinal
metastasis

Salman Abbasi Fard1  | Pouria Pourzand2  | Farhad Tabasi3,4   |

Mohsen Mohammadi2  | Mohammad Nafeli2  | Zahra Jourahmad5

1
Department of Neurosurgery, Golestan
University of Medical Sciences, Abstract
Golestan, Iran Spinal tuberculosis (TB) is diagnostically challenging, particularly in atypical
2
School of Medicine, Zahedan forms. Non-­contiguous multilevel spinal TB (NMLST) is a rare presentation of
University of Medical Sciences,
Zahedan, Iran
spinal TB, mimicking spinal malignancies. We reported an unusual NMLST case
3
Department of Physiology, Faculty with a paraspinal and epidural abscess in a young patient with misleading clinical
of Medical Sciences, Tarbiat Modares and imaging presentations.
University, Tehran, Iran
4
Institute for Brain Sciences and KEYWORDS
Cognition, Tarbiat Modares University, non-­contiguous multilevel, Pott's disease, spinal metastasis, spinal tuberculosis, spinal
Tehran, Iran tuberculosis
5
Department of Neurology, School of
Medicine, Zanjan University of Medical
Sciences, Zanjan, Iran

Correspondence
Farhad Tabasi, Department of
Physiology, Faculty of Medical Sciences,
Tarbiat Modares University, Jalal Al-­
Ahmad Street, Nasr Bridge, Tehran,
Iran.
Email: f.tabasi@modares.ac.ir and
tabasif@gmail.com

1  |  I N T RO DU CT ION Approximately 16% of TB cases are extra-­pulmonary, of

which 10% have musculoskeletal involvement2,3; spinal
Tuberculosis (TB) is a chronic granulomatous infec- TB (tuberculosis spondylitis or Pott's disease) accounted
tion caused by aerosolized inhalation of Mycobacterium for nearly 1% of all TB cases.4
Tuberculosis.1 The World Health Organization estimates Spinal TB usually presents insidiously due to hematog-
that in 2021, almost 4.1 million people will suffer from enous dissemination from the primary source.5 The tho-
TB.2 TB is the second leading cause of death due to an racolumbar region is more common, followed by cervical
infectious disease in the COVID-­ 19 era, claiming 1.5 and sacral involvement.5 Typically, spinal TB involves two
million lives per year.2,3 Although pulmonary TB is the or more contiguous vertebrae and intervertebral discs,
most common presentation, extra-­ pulmonary involve- which could be associated with paraspinal extensions, ab-
ment may occur, with or without pulmonary infection. scess formation, vertebral destruction, cord compression,
This is an open access article under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs License, which permits use and distribution in any
medium, provided the original work is properly cited, the use is non-commercial and no modifications or adaptations are made.
© 2023 The Authors. Clinical Case Reports published by John Wiley & Sons Ltd.

Clin Case Rep. 2023;11:e7053.  wileyonlinelibrary.com/journal/ccr3   |  1 of 7

https://doi.org/10.1002/ccr3.7053
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2 of 7       FARD et al.
and spinal deformity.6 On imaging, typical findings are
easily recognizable, but atypical forms may resemble
other possible diagnoses and usually mandate more thor-
ough and invasive investigations.7,8
One atypical form is vertebral lesions separated by in-
tact vertebrae (i.e., skip lesions) with spared intervertebral
disc, which mislead toward other potential diagnoses, in-
cluding pyogenic spondylitis and malignancy.9 This non-­
contiguous multilevel spinal TB (NMLST) is quite rare,
with inconsistent incidence reports, ranging from 1.1%
to 71.4%, highlighting the importance of whole-­spine im-
aging and more wide-­ranging investigations.10,11 Herein,
we reported a quite unusual NMLST from southeast Iran
with preserved intervertebral discs accompanied by large
paravertebral soft-­tissue masses extending to the neural
foramen, epidural space, and compressing spinal cord in
a middle-­aged male patient, which led to a challenging
diagnostic dilemma. Also, we reviewed the relevant litera-
ture and discussed the educational notes.
2  |  C A S E PR E SE N TAT ION
A 37-­year-­old male patient presented with chronic, pro-
gressive back pain, paresthesia, and weakness of lower
limbs in the last 2 months, which had led to abnormal gait.
Moreover, he had constitutional symptoms, including
low-­grade fever, loss of appetite, and a significant 10  kg
weight loss in the last 2 months. He denied any history of
a specific disease, including sexually transmitted diseases
or malignancy, except for an infected left shoulder wound
after a falling 2 years ago that had been partially managed
with frequent courses of intravenous antibiotics, wound
debridement, and drainage. Also, there was no clear his-
tory of TB exposure, including in his family, and unclear
BCG vaccination.
At the admission, he was afebrile and hemodynami-
cally stable but with a cachectic appearance and unable
to walk without help. A crusted ulcer was noted at the left
upper thorax, below the lateral half of the clavicle. A phys-
ical examination revealed a local tenderness around the
paraspinal region at the upper thoracic level. Neurologic
examination revealed sensory deficit below T10 as a bi-
lateral symmetric paresthesia and proprioception sensory
deficit of lower limbs. A significant symmetric paraparesis
accompanied by ataxic spastic gait was also noted at the
motor examination, along with increased deep tendon re-
flexes of the lower extremities.
Initial laboratory investigation revealed a white blood
cell count of 9.2 * 1000/mm3 (upper limits of normal range)
with 66% of polymorphonuclear cells and hypo-­chromic/
microcytic anemia with hemoglobin of 9.7  mg/dL. The
erythrocyte sedimentation rate was 83 mm/h (normal
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F I G U R E 1   Chest radiograph of the patient. The left lobulated
paraspinal opacities, LUL atelectasis, and multiple left pleural
oval-­shaped opacities are noted (arrows). There is no evidence of
cavitary lesions. LUL, left upper lobe.
range for men under 50 years <15), and C-­reactive protein
was 12 (normal range <10 mg/L). Viral markers for hepa-
titis and HIV were reported as negative.
On routine imaging, chest radiography showed mul-
tiple lobulated opacities at the left paraspinal region,
atelectasis of the left upper lobe (LUL), and left hilar en-
largement, which was neither indicative of hilum overlay
sign nor suggestive of a mediastinal mass. Additionally,
several opacities were evident in the left pleura's lateral
margin, which, together with previous findings, raised
our suspicion of malignancy (Figure  1). These findings
required more radiological evaluation with computed to-
mography (CT) or magnetic resonance imaging (MRI),
while spinal imaging was the top priority due to progres-
sive neurological deficits.
An emergent spinal MRI was requested to explore
the cause of cord compression. The MRI revealed large
paravertebral lobulated soft-­tissue masses adjacent to the
C7 and T6 vertebral bodies measuring 49  *  27 mm and
71  *  27 m, respectively, extending into the right neural
foramen and posterolateral epidural space and causing
a significant compressive effect on the cord as well as
the abnormal high signal intensity in the adjacent cord.
Furthermore, abnormal bone signals in C7, T3, and T6 ver-
tebral bodies, posterior elements, and spinous processes
were demonstrated (low-­ intensity on T1-­ weighted and
high-­intensity on T2-­weighted) and (short-­TI inversion re-
covery [STIR] images). In adjacent structures, the involve-
ment of paraspinal muscles, the left pleura with multiple

FARD et al.
oval-­shaped masses, and the sixth rib and enlarged para-­
aortic lymph nodes were detected (Figures 2–­4).
Based on clinical, laboratory, and imaging findings, the
presumptive diagnosis was primary spinal malignancy or
metastasis, requiring a malignant disease workup. The
other possible diagnosis was pyogenic spondylitis, given
the non-­healing ulcer, a collection at the left shoulder re-
gion, and evidence of paravertebral abscess. The whole-­
spine MRI was required to examine whether other levels
were involved. However, due to the instrumental access
limitations and progressive neurologic deficits caused by
cord compression, the patient was a candidate for emer-
gent decompressive surgery after significant compression
on the upper thoracic spinal cord was demonstrated. Thus,
paravertebral pockets were evacuated using the posterior
midline approach, laminectomy and further pus drainage
through the spinal canal were performed, samples from
pus, laminae bone, and soft tissue were taken, and thor-
ough irrigation, debridement, and drain placement were
done.
Lastly, biopsies from paravertebral and epidural le-
sions were obtained. Microscopic examination showed
fibro-­connective tissue with marked infiltrative of chronic
inflammatory cells accompanied by a collection of epi-
thelioid and Langhans giant cells with caseous necrosis.
Additionally, fragments of destructed bone trabecula were
seen in the examination. Altogether, these findings were
compatible with chronic granulomatosis inflammation
with caseous necrosis, suggestive of TB (Figure 5).
Postoperatively, anti-­TB treatment, including rifam-
pin 10 mg/kg/day, isoniazid 5 mg/kg/day, pyrazinamide
25 mg/kg/day, and ethambutol 15 mg/kg/day, was initi-
ated consequently. After that, neurologic deficits resolved
gradually, and the patient responded satisfactorily to an-
ti-­TB treatment, reporting further neurological improve-
ment at discharge. Although identifying susceptibility to
F I G U R E 2   Spinal MRI (sagittal
view); large paravertebral lobulated soft
tissue (arrows) and changes in signal
intensity at the C7 level. The T1W image
shows a low signal intensity (A); the
T2W image (B); the STIR image shows a
high signal intensity (C). STIR, short-­TI
inversion recovery.
|
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    3 of 7
drugs is advisable at the baseline and during treatment,
we could not perform such tests due to our limitations.
Afterward, the patient showed further improvement in
his clinical condition in a short-­term follow-­up a few
months after starting anti-­ TB therapy; nevertheless,
despite our effort to follow his condition, we could not
reach the patient for a long-­term follow-­up (i.e., 1 year
after the initial management) to reassess his clinical and
radiological findings.
3  |  DISC USSION
In extra-­pulmonary TB, the infection spreads through
the paravertebral venous plexus and initially settles at
the anterior corners of the vertebrae.5 The most common
sites are thoracic and lumbar regions, followed by the
cervical and sacral spine,6 and typically involve adjacent
structures, like vertebral bodies and intervertebral discs.5
Atypical cases may present with posterior elements in-
volvement (i.e., pedicle, transverse and spinous processes,
and lamina), spared vertebral body and disc, solitary ver-
tebral bone destruction without intervertebral disc in-
volvement, non-­contiguous skip lesions (i.e., affecting two
or more separated spinal regions), and extradural lesions
with preserved vertebral bone.9
Non-­contiguous multilevel spinal TB is a rare, atypi-
cal form of spinal TB and may have a higher incidence
than reported.10,11 Novel imaging approaches (e.g., rou-
tine whole-­spine MRI) increased reports of atypical pre-
sentations in recent years. The NMLST incidence ranges
from 1.1%12 to 71.4%.10 However, the latter study might
be biased because it was conducted at a regional referral
center for spinal deformity and a small number of total
cases. Additionally, this incidence rate discrepancy may be
caused by different imaging modalities.
 |

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4 of 7       FARD et al.

F I G U R E 3   Spinal MRI (sagittal view); the compressive effect of the lesion, large paravertebral lobulated soft tissue, and changes in
signal intensity at multiple separated levels of thoracic vertebral bodies without collapse as well as uninvolved intervertebral discs. The
myelogram shows CSF blockade (arrowhead) at the thoracolumbar junction level (A); the T1-­weighted image shows a low signal intensity
for both involved vertebral bodies and abscess (B); the T2-­weighted image (C) and STIR image show high signal intensity in involved
vertebral bodies and also paravertebral abscess (D). CSF, cerebrospinal fluid; STIR, short-­TI inversion recovery.

F I G U R E 4   Spinal MRI (axial view). Bony destruction in the right side of the posterior arc and body of the inferior thoracic vertebra
is evident (arrowhead), suggesting osteomyelitis. Also, the right paraspinal abscess that extended to the right neural foramen and spinal
canal caused thecal sac compression (arrow), canal stenosis, and cord compression. Evidence of left empyema can be seen (red arrows). T2-­
weighted FSE image (A); the STIR image (B). FSE, fast spin-­echo; STIR, short-­TI inversion recovery.

Herein, we presented an NMLST case with two segre-
gated levels of involvement of vertebral body and poste-
rior elements, which occurred in the cervical and thoracic
regions, paravertebral and epidural abscesses, and intact
intervertebral discs. Mostly, NMLST cases have two non-­
contiguous levels of involvement, while there are few
cases with more than two sites and only eight reports with
extensive whole-­spine involvement.13–­18
The diagnosis is mainly based on imaging in conjuga-
tion with clinical suspicion, which should be established
by serology and histopathology. MRI is the modality of
choice due to higher sensitivity and specificity for soft tis-
sues than other options.19 Typically, MRI demonstrates de-
creased signal intensity on T1-­weighted (T1W) but higher
intensity on T2-­weighted (T2W) and short-­TI inversion
recovery (STIR) in the vertebrae and their adjacent discs
and soft-­ tissue collections.7,19 However, differentiating
spinal TB on imaging alone could be challenging in some
cases, like the case presented here.
The unique aspect of our case is the combination
of NMLST with posterior lesions, preserved interver-
tebral discs, and soft-­tissue components in paraspinal

FARD et al.
F I G U R E 5   Histopathological
findings of the paravertebral and epidural
lesion revealed chronic granulomatous
inflammation with caseous necrosis. (A)
hematoxylin and eosin (H&E) ×100, and
(B) H&E ×400.
and cord compression, which resemble pyogenic spon-
dylitis and spinal malignancy.20 Pyogenic vertebral os-
teomyelitis and spondylodiscitis are commonly caused
by Staphylococcus aureus.21 Potential risk factors are
intravenous drug abuse, diabetes, recent systemic infec-
tion, immunosuppression, or malignancy, while TB as a
causative agent is often seen in immunocompromised
patients or endemic areas with low socioeconomic sta-
tus.21 Several features in MRI are suggested to differenti-
ate spinal TB from pyogenic spondylodiscitis, including
heterogeneous T1W signal, epidural abscess/phlegmon,
and severe vertebral destruction in cases from a highly
TB prevalent region.22
Paravertebral TB abscesses tend to extend beyond
osseous involvement with intervertebral discs sparing,
demonstrated as T1W hypointensity and T2W hyperin-
tensity with a thin contrast-­enhancing wall on MRI; in
contrast, pyogenic spondylitis usually forms abscesses
with thick and irregularly enhancing walls, early her-
niation and disc destruction.23 In a review of 26 spinal
TB cases, only one had non-­contiguous thoracic and
lumbar involvement.24 Notably, abscesses were present
in 15 patients, 12 of which had an abscess at the initial
presentation, and in the other 3, abscesses developed
during treatment, highlighting the importance of fol-
lowing patients to detect abscess.24
Although paravertebral masses are highly sugges-
tive of spinal TB, non-­ contiguous multilevel lesions
with intact intervertebral discs are usually seen in ma-
lignancies,15,23,25 which could misdiagnose spinal TB
as malignancy.26,27 Bone metastases are the most com-
mon spinal neoplastic diseases, usually involving lower
thoracic and upper lumbar and spinal TB.25 Spinal me-
tastases show T1W hypointensity and T2W and STIR
hyperintensity, involving multiple non-­contiguous ver-
tebral bodies, particularly posterior elements with pre-
served intervertebral discs.25 Notably, posterior element
involvement in spinal TB is atypical and could predis-
pose to neurological deficits.8
To differentiate metastatic disease from spinal TB,
Du et al. introduced a scoring system based on five fea-
tures: history of malignancy, subligamentous spreading,
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    5 of 7
posterior element lesions, sequestrate formation, and
preserved disc.28 In this scoring system, spinal metas-
tasis gets a higher score than spinal TB, with a cutoff
value of 5, demonstrating 97.85% sensitivity and 88.33%
specificity for metastatic disease. Intriguingly, according
to this scoring system, our case gets a score of 8 in favor
of metastasis. Therefore, such a scoring system may not
be applicable in all conditions, particularly in patients
from endemic regions for TB like our case (estimated
10–­49 per 100,000 for the country29); TB should always
be kept in mind. In our case, histopathological examina-
tion ruled out metastatic disease despite highly sugges-
tive features.
Another alternative in our case, though less likely,
was pyogenic spondylitis, considering a non-­ healing
soft-­tissue infection that formed an abscess in his shoul-
der as a significant risk factor, partially managed by sev-
eral oral and intravenous antibiotic courses. However,
the patient was a candidate for emergent decompres-
sive laminectomy and abscess drainage due to progres-
sive neurological deficits. A biopsy was performed, and
pathology suggests TB infection as a highly possible
diagnosis. Therefore, our case highlights the histopatho-
logical assessment in uncertain diagnosis, which may
lead to treatment delay and error.
The cornerstone of spinal TB treatment is anti-­ TB
agents. The recommended regimen for spinal TB treat-
ment empirically is the 4-­ drug regimen, for usually
9–­12  months, though the recommendation on multi-
ple spine involvement (especially NMLST) is deficient.
Further, the susceptibility pattern of M. tuberculosis to the
anti-­TB agents should be (if feasible) identified to guide
the treatment regimen accordingly.30 On the contrary, sur-
gical decompression is indicated for cases with spinal cord
compression, neurological deficits, vertebral destruction
leading to spinal instability and deformity, and large para-
vertebral abscesses.13 However, thorough evidence-­based
guidance for treatment protocol, particularly surgical pro-
cedures for NMLST, is lacking. Since the diagnosis was
not certain in our case until pathology results, the anti-
biotic therapy was not started until the TB infection was
revealed.
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6 of 7       FARD et al.
4  |  CO N C LUSION
In conclusion, we reported an uncommon manifestation
of TB as an NMLST from southeast Iran, an endemic re-
gion for TB. The diagnosis of TB should always be kept
in mind in endemic regions, even when its manifesta-
tions are against the usual TB presentation. Increasing
awareness of atypical TB spondylitis is crucial, given the
emerging prevalence of TB in both endemic and non-­
endemic regions. Prompt diagnosis is vital to determine
the appropriate treatment and prevent severe sequels and
morbidities. Additionally, the only way to reach a definite
diagnosis in most circumstances is through biopsy and
histopathological examination.
AUTHOR CONTRIBUTIONS
SA was responsible for the patient's operation, involved
in patient care, and reviewed the literature. PP and FT
were involved in patient documents and data collection,
reviewed the literature, and drafted and wrote the manu-
script. MM, MN, and ZJ assisted in the literature review.
SA and FT critically reviewed and edited the final version.
All authors read and approved the final manuscript.
ACKNOWLEDGMENTS
None.
FUNDING INFORMATION
The authors did not receive any fund for this work.
CONFLICT OF INTEREST STATEMENT
The authors declare that they have no competing interests.
DATA AVAILABILITY STATEMENT
Data sharing is not applicable to this article as no datasets
were generated or analysed during the current study.
ETHICAL APPROVAL
The Institutional Review Board and Ethics Committee of
Zahedan University of Medical Sciences waived the re-
quirement for ethical approval.
CONSENT
Written informed consent was obtained from the patient
to publish this case report and any accompanying images.
ORCID
Farhad Tabasi  https://orcid.org/0000-0003-4877-0701
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How to cite this article: Fard SA, Pourzand P,
Tabasi F, Mohammadi M, Nafeli M, Jourahmad Z.
Non-­contiguous multilevel spinal tuberculosis: A
case report of unusual spinal tuberculosis
resembling spinal metastasis. Clin Case Rep.
2023;11:e7053. doi:10.1002/ccr3.7053