Case report
BMJ Case Rep: first published as 10.1136/bcr-2021-246359 on 29 November 2021. Downloaded from http://casereports.bmj.com/ on November 30, 2021 at Universiti Malaysia Sabah.
1
Anaesthesia and Intensive Care
Unit, Hospital Queen Elizabeth,
Kota Kinabalu, Sabah, Malaysia
2
Department of Paediatrics,
Melaka General Hospital,
Melaka, Malaysia
3
Department of Orthopaedics,
Hospital Queen Elizabeth, Kota
Kinabalu, Sabah, Malaysia
4
Department of Anaesthesiology
and Intensive Care, Faculty of
Medicine and Health Sciences,
Universiti Malaysia Sabah, Kota
Kinabalu, Sabah, Malaysia
Correspondence to
Dr Tat Boon Yeap;
​boontat@​ums.e​ du.​my
Accepted 11 November 2021
© BMJ Publishing Group
Limited 2021. No commercial
re-­use. See rights and
permissions. Published by BMJ.
To cite: Jamil J, Tay CY,
Bong CP, et al. BMJ Case
Rep 2021;14:e246359.
doi:10.1136/bcr-2021-
246359
Anaesthetic challenges in the spine surgery of a
young Asian man with lumbar amyloidosis
Jabraan Jamil,1 Chong Yan Tay,2 Chin Pei Bong,3 Tat Boon Yeap ‍ ‍4
SUMMARY
Primary amyloidosis is a rare systemic disorder often
associated with multiple organ dysfunction. The most
common form, light chain amyloidosis, has an estimated
age-­adjusted incidence of 5.1–12.8 cases per million
person-­years. Spine involvement is extremely uncommon.
We present the case of a young Asian man with newly
diagnosed amyloidosis involving the lumbar spine among
multiple organs with a pathological vertebral fracture
that required urgent spine surgery. We believe this is
the first reported case to discuss the perianaesthetic
challenges in the management of lumbar spine
amyloidosis.
BACKGROUND
A young healthy Asian man presented to us with
low-­grade fever and chronic back pain. Initially
suspected to be due to diseases (such as spinal
tuberculosis (TB)) common in our locality in the
Malaysian state of Sabah, further investigations,
however, later diagnosed the patient as suffering
from systemic amyloidosis with lumbar spine
involvement, complicated with a pathological L4
vertebral fracture that required emergency surgery.
Meticulous work-­ up of various systems was
conducted before an operative plan that included
specific considerations in multiple domains could
be drawn. The patient subsequently underwent a
successful elective posterior lumbar spine instru-
mentation surgery. We here share our experience in
the perioperative assessment, planning and execu-
tion of this case.
CASE PRESENTATION
A 47-­year-­old healthy man (weight: 72 kg; height:
1.65 m) presented to our institution with complaints
of non-­ productive cough and low-­ grade fever
which had persisted for 2 days. He did not report
any runny nose, hoarseness of voice, sore throat,
diarrhoea or recent close contact with COVID-­19
cases. According to the patient, he had also been
experiencing intermittent lower back pain for about
6 months which was associated with non-­specific
body aches. However, he denied having any history
of trauma.
Our examination found him pale, alert and
comfortable. His blood pressure, heart rate,
and temperature and arterial oxygen saturation
were, respectively, at 118/72 mm Hg, 75 beats
per minute, and 37.5°C and 98% on air with a
respiratory rate of 16 breaths per minute. A huge
hepatosplenomegaly was present and crepitations
were detected over the bilateral lower zones of his
Jamil J, et al. BMJ Case Rep 2021;14:e246359. doi:10.1136/bcr-2021-246359
lungs. Examination of his spine and back revealed
tenderness over the fourth and fifth lumbar verte-
brae (L4/L5) with the patient unable to either sit
up or turn from side to side. There were neither
motor nor sensory neurological deficits in his
lower limbs.
INVESTIGATIONS
►► The patient’s full blood count showed a
haemoglobin value of 77 g/L (normal value:
135–155 g/L), total white blood cell count of
15.77×109 /L (normal value: 7–11×109 /L)
and platelet count of 422×109 /L (normal
value: 150–450×109 /L).
►► Serum electrolytes were within normal range
except for the elevation of urea and creatinine
at 14.7 µmol/L (normal value: 3–11 µmol/L)
and 147 µmol (normal value: 60–100 µmol/L).
Protected by copyright.
Calculated creatinine clearance was 51 mL/
min/1.73 m2.
►► Serum albumin was markedly reduced at 20
g/L (normal value: 40–50 g/L). Liver func-
tion tests indicated mild elevation of alanine
aminotransferase of 56 µmol/L (normal value:
<50 umol/L), aspartate aminotransferase of 55
µmol/L (normal value:<50 µmol/L) and alka-
line phosphatase of 311 µmol/L (normal value:
50–300 µmol/L). Serum globulin and total bili-
rubin were within normal range.
►► Chest X-­ray showed right lower zone opacity.
►► Twenty-­ four-­
hour urine protein was grossly
elevated at 4667 mg/day (normal value: <150
mg/day).
►► Urine protein to creatinine ratio was raised at
343 mg/mmol (normal value: <300 mg/mmol),
suggestive of nephrotic syndrome.
►► Echocardiogram showed ejection fraction of
48% with mild mitral regurgitation and trivial
tricuspid regurgitation. No valvular stenosis
was observed, with good cardiac contractility.
►► Ultrasonography of the abdomen and pelvis
showed an enlarged liver with normal paren-
chymal echogenicity and texture spanning 20.3
cm with smooth margins. No focal lesion was
observed. The spleen was enlarged at 13.4 cm,
while both kidneys were of normal size and
echotexture.
►► MRI of the spine showed L4 pathological frac-
ture with adjacent enhancing paravertebral
soft tissue thickening and anterior epidural
space collection causing severe canal stenosis
(figures 1 and 2).
►► Bone marrow aspiration and trephine biopsy
(BMAT), transpedicular biopsy of L4 and
1
 Case report
Figure 1 Sagittal view of MRI of the spine showing L4 pathological
fractures with an anterior epidural space collection causing severe canal
stenosis.
renal biopsy under local anaesthesia indicated amyloid
depositions.
►► Urine free light chain (FLC) assay was massively elevated
with Kappa of 74.4 mg/L (normal value: 0.0–25.8 mg/L) and
Lambda of 59.6 mg/L (normal value: 0.0–11.3 mg/L). Urine
FLC Kappa to Lambda ratio was reduced at 1.248 (normal
range: 1.4–6.2).
►► The patient tested negative for both TB and COVID-­19.
DIFFERENTIAL DIAGNOSIS
The provisional diagnosis was pathological fracture of the L4
vertebra secondary to amyloidosis.
Differential diagnoses included spine TB due to this being
endemic in the state of Sabah, spinal malignancy and autoim-
mune spondyloarthropathies.
Figure 2 Axial view of MRI of the spine showing adjacent enhancing
paravertebral soft tissue thickening.
2 Jamil J, et al. BMJ Case Rep 2021;14:e246359. doi:10.1136/bcr-2021-246359
BMJ Case Rep: first published as 10.1136/bcr-2021-246359 on 29 November 2021. Downloaded from http://casereports.bmj.com/ on November 30, 2021 at Universiti Malaysia Sabah.
Figure 3 Frontal view of the patient with macroglossia.
TREATMENT
Protected by copyright.
In the ward, the patient developed multiple temperature spikes
of up to 39°C despite being commenced on a course of intra-
venous amoxicillin-­ clavulanic acid (Augmentin) for 1 week
for community-­acquired pneumonia. His lower back pain was
controlled with oral analgesics. Prednisolone was subsequently
commenced for 3 weeks on the diagnosis of nephrotic syndrome.
Systemic amyloidosis was confirmed by renal and bone biopsy
results 2 weeks later.
In view of his instability pain, an elective and minimally inva-
sive posterior instrumentation surgery was performed on the
patient. A detailed preoperative anaesthetic assessment revealed
that he had macroglossia with thick lips (figure 3). His Mallam-
pati score was recorded at 3 and thyromental distance at 6 cm.
There was no limitation in neck flexion and extension.
In the operating theatre (OT), non-­invasive blood pressure
monitoring, ECG, pulse oximetry and temperature probe were
placed on the patient. He was intubated with size 8.0 mm flex-
ometallic endotracheal tube (ETT) without any difficulty. An
arterial line and a central venous catheter were inserted into the
right radial and internal jugular veins, respectively, for perioper-
ative monitoring and resuscitation. The patient was then put in
prone position with adequate support over his head, neck, limbs
and ETT.
Intraoperatively, his vital signs were within normal range.
Anaesthesia was maintained with sevoflurane and analgesia with
controlled infusion of remifentanil. Tidal volume and
target-­
airway pressure were maintained at 400–450 mL and 18–22
cmH20, respectively. Pedicle screws were inserted from L2 to S1
(lumbar 2 to sacral 1) via minimally invasive technique. Each
screw was augmented with bone cement due to osteoporotic
bone at all instrumented levels to increase screw strength and
prevent loss of reduction. Intraoperative blood loss was 800 mL
and the patient was transfused with two pints of packed cells.
Mean arterial pressure (MAP) was kept adequate throughout the
OT procedure. He was later extubated and returned to his ward
for recuperation.

OUTCOME AND FOLLOW-UP
Postoperatively, the patient was able to move both his lower
limbs. He subsequently underwent regular physiotherapy and
rehabilitation. On review under a combined orthopaedic and
haematology outpatient clinic 2 weeks later, he was observed to
be responding well.
DISCUSSION
Primary amyloidosis is a rare systemic disorder character-
ised by the extracellular deposition of abnormal precursor
proteins.1 It is more common in men aged 40–55 years.2 3
Amyloid disease presents in various subtypes, depending on
the proteins derived, such as light chain (AL), serum amyloid
A (AA), and transthyretin (ATTR), with the most common
form of systemic amyloidosis being AL which is responsible
for the death of 1:1500 people in the UK.1 4 Although there is
a lack of recent epidemiological data on amyloidosis, a study
by de Asúa et al at the Mayo Clinic reported the estimated age-­
adjusted incidence of AL as 5.1–12.8 cases per million person-­
years.1 5 In developing countries, AA amyloid is more common
possibly due to the higher prevalence of chronic infectious
diseases such as leprosy, TB and osteomyelitis.6
In cases where AL amyloidosis is suspected, serum protein
electrophoresis with immunofixation electrophoresis is usually
conducted both as a screening test and to rule out multiple
myeloma.7 8 The definitive diagnostic method for amyloidosis
is tissue biopsy. Histological examination would demonstrate
the presence of amyloid deposits in affected tissues, as observed
in our patient’s BMAT, renal and transpedicular biopsies. The
main histological diagnostic feature is the presence of green
birefringence due to the positive reaction to Congo red stain
under cross-­polarising microscopy.9 10
Systemic amyloidosis usually affects the kidney, liver, heart,
nervous system and lungs, leading to organ damage. Patients
Table 1 Perioperative anaesthetic concerns and management for patient with systemic amyloidosis
Patient’s clinical issue
Anaemia
Macroglossia and thick lips
Bilateral lung crepitations with right lower zone haziness on
chest X-­ray
Massive hepatosplenomegaly
Impaired liver function with hypoalbuminaemia
Impaired renal function
Pathological L4 fracture
Cardiomegaly, mitral regurgitation, tricuspid regurgitation and
mildly reduced ejection fraction of 48%
Glucocorticoid dependence
Immunosuppression due to urinary loss of immunoglobulins and Increased perioperative risk of infection
long-­term steroid therapy
Jamil J, et al. BMJ Case Rep 2021;14:e246359. doi:10.1136/bcr-2021-246359
Case report
BMJ Case Rep: first published as 10.1136/bcr-2021-246359 on 29 November 2021. Downloaded from http://casereports.bmj.com/ on November 30, 2021 at Universiti Malaysia Sabah.
may present with signs and symptoms of nephrotic syndrome
(28%), congestive heart failure (17%) and malabsorption
(5%).11 However, amyloid deposition involving the vertebra
is extremely uncommon and difficult to diagnose radiologi-
cally.2 3 12 Patients would usually complain of non-­ specific
back pain which might be associated with loss of motor and
sensory functions. These symptoms could be improved or alle-
viated with surgical treatment. Our patient reported chronic
back pain and low-­grade fever, with these complaints initially
confused with other more common local diseases such as spinal
TB, abscess and bone malignancy. Features of a collapsed
vertebral segment, mimicking spinal metastasis, are commonly
seen via a spine MRI.12
Anaesthesia for patients with amyloidosis requires metic-
ulous considerations. The various sites of abnormal protein
deposits could challenge the anaesthetist, with the key
concerns being issues or risks of heart failure, arrhythmias,
airway difficulty, bleeding and altered responses to pharmaco-
logical measures.13
Amyloid deposition frequently occurs in the head, neck,
thorax and oropharynx region.14 Abnormal protein deposits
in the tongue cause macroglossia, which in turn increases the
risk of difficult intubation. This is a characteristic feature of
AL amyloidosis and occurs in 20% of patients.14 Amyloid
deposition in the neck and larynx could lead to subglottic
stenosis and hinder the process of intubation.15 16 Involvement
of the lung is fairly common although rarely symptomatic.15
Protected by copyright.
However, fibrosis of lung parenchyma with concomitant
pleural effusion can lead to difficult ventilation and extuba-
tion. A preoperative detailed airway assessment is therefore
necessary with the use of video laryngoscope to facilitate intu-
bation and lung protective ventilation strategies to prevent
atelectasis and hypoxaemia.
Anaesthetic concerns Anaesthetic management
Impaired tissue oxygenation Preoperative cross-­matching and transfusion of blood
Difficult intubation Meticulous airway assessment in anticipation of difficult
Risk of oral injury during laryngoscopy and intubation intubation
Intubation with videolaryngoscopy (CMAC) or awake
fibreoptic
Poor lung compliance Adequate preoxygenation and proper patient
Difficult ventilation positioning
Perioperative hypoxaemia Intraoperative protective lung ventilation strategy
Judicious fluid administration to prevent acute
pulmonary oedema
Risk of organ rupture and increased intra-­abdominal Proper cushioning of thorax and pelvis in prone position
pressure causing ventilation and oxygenation
difficulty
Risk of aspiration pneumonitis
Impaired drug metabolism and distribution Optimisation of drug dosing and interval
Impaired drug excretion Optimisation of drug dosing and interval
Fibrotic spine and risk of massive bleeding Proper patient positioning and cushioning
Gentle tissue dissection and handling of spine tissues by
experienced surgeons.
Low threshold for massive transfusion
Risks of hypotension, bradycardia and impaired Judicious fluid administration, invasive blood pressure
sympathetic responses monitoring and central venous catheter placement for
resuscitation
Risk of adrenal suppression Perioperative steroid replacement
Strict aseptic technique in all invasive procedures
Prophylactic antibiotics
3
Case report
Infiltration of amyloid into the myocardium could lead to
cardiomyopathy, valvulopathy, arrhythmia and pericardial
effusion. Cardiac performance is compromised when myocar-
dial contractility, cardiac output and systemic vascular resis-
tance are impaired. Cases of perioperative fatality or heart
failure due to cardiac amyloidosis have been documented.17
Preoperative estimation of cardiac function is mandatory
when patients with cardiac amyloidosis undergo anaesthesia,
in which echocardiographic examination would be useful.17
A judicious approach to intraoperative fluid replacement and
blood transfusion should be employed as hypervolemia could
lead to an excessive increase in myocardial preload, while
dehydration and tachycardia result in an increase in myocar-
dial oxygen demand.
AL amyloidosis is also associated with microvascular fragility,
platelet dysfunction and factor X deficiency which could lead to
increased bleeding tendency.13 18 19 Liver amyloid deposits are
more prevalent in AL compared with other forms of amyloi-
dosis; the incidence of hepatomegaly out of proportion to liver
enzyme derangement is a diagnostic clue, as was the case of our
patient.20 Abnormal liver function due to amyloid deposition
promotes risk of bleeding, hypoalbuminaemia and suboptimum
drug metabolism. Extreme caution therefore should be exercised
intraoperatively for blood transfusion and drug administration
in patients undergoing high-­risk surgery.
Both AL and AA amyloidosis, commonly associated with
renal dysfunction, occur in up to 50% of patients with systemic
immunoglobulin-­related amyloidosis.21 Patients develop renal
insufficiency and protein-­ losing nephropathy which may
require long-­term steroid therapy.22 Our patient demonstrated
laboratory features of nephrotic syndrome and was commenced
on oral prednisolone for 3 weeks, which increased his risk of
adrenal insufficiency.12 Perioperative hydrocortisone therapy
was thus administered to prevent cardiovascular collapse.
Fluids should also be judiciously administered to avoid acute
pulmonary oedema and worsening of renal function. Urinary
loss of immunoglobulins blunts immune responses and long-­
term steroid therapy further puts patients in an immunocom-
promised state.23–25
Our patient had a rare systemic amyloidosis which affected
his myocardium, lungs, tongue, spleen, liver, bone marrow
and spine, and there was a prolapse of the L4 disc. Despite
all the anticipated perioperative anaesthetic issues in prone
position, we were fortunate that he was saved. Our over-
riding concern was his macroglossia which we managed to
delicately navigate using conventional videolaryngoscopy and
intubation. His lung compliance was good despite being put
in prone position. We were nevertheless cautious not to intro-
duce any iatrogenic barotrauma by employing lung protective
strategies. His huge hepatosplenomegaly in prone position
posed risks of rupture, raised intrabdominal pressure and
difficult ventilation. These risks were addressed by placing
cushion rolls below the thorax and pelvis while ensuring a
free uncompressed abdomen. We exercised judicious fluid
resuscitation and packed cell transfusion to maintain an
adequate MAP perioperatively. The spine surgeons were
cautious during tissue dissection and the placement of spine
implants to minimise bleeding.
We believe this is the first reported case on the perianaes-
thetic challenges and management of lumbar spine surgery in
patients with systemic amyloidosis. We therefore feel privi-
leged with this opportunity to share this invaluable experience.
A summary of the perianaesthetic concerns surrounding our
patient is illustrated in table 1.
4 Jamil J, et al. BMJ Case Rep 2021;14:e246359. doi:10.1136/bcr-2021-246359
BMJ Case Rep: first published as 10.1136/bcr-2021-246359 on 29 November 2021. Downloaded from http://casereports.bmj.com/ on November 30, 2021 at Universiti Malaysia Sabah.
Patient’s perspective
I am thankful that my back pain problem was successfully
resolved after the surgery. However, developing a rare
multiorgan involvement of amyloidosis is a condition which I
would have to manage for the rest of my life, with the help of
steroids. I am lucky that my wife and children are very supportive
of me. I hope to pull through this battle of amyloidosis safely.
Learning points
►► Systemic amyloidosis is a rare disorder characterised by
abnormal extracellular protein deposition.
►► Amyloidosis of the spine is very rare and may cause severe
disability.
►► Diagnosis of amyloidosis and its implications to various
organs can be exhaustive and requires a high index of
suspicion.
►► Thorough preoperative work-­up and assessment are vital
to ensure the multitude of potential risks in an amyloidosis
patient are accounted for, and for a holistic plan to be put
into effect.
►► Anaesthesia for patients with severe systemic amyloidosis
requires meticulous considerations especially if they are put
in prone position.
Protected by copyright.
Acknowledgements We would like to thank the Director General of the
Malaysian Ministry of Health for his permission for us to publish this manuscript.
Contributors TBY, CPB and JJ are the clinicians managing the patient. They co-­
authored this manuscript as well as were involved in data collection. CYT is the co
author of this manuscript together with other clinicians mentioned.
Funding The authors have not declared a specific grant for this research from any
funding agency in the public, commercial or not-­for-­profit sectors.
Competing interests None declared.
Patient consent for publication Consent obtained directly from patient(s)
Provenance and peer review Not commissioned; externally peer reviewed.
Case reports provide a valuable learning resource for the scientific community and
can indicate areas of interest for future research. They should not be used in isolation
to guide treatment choices or public health policy.
ORCID iD
Tat Boon Yeap http://​orcid.​org/​0000-​0002-​2517-​597X
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