- Lana-Peixoto MA,
Callegaro D, Talim N, Talim LE, Pereira SA,
Campos GB, et al. Pathologic yawning in
neuromyelitis optica spectrum disorders. Mult
Scler Relat Disord 2014;3:527-32.
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- Spahlinger
V, Niessen A, Rauer S, Krämer S, Reinhard
M. The big yawning: Pathological yawning as a
symptom of neuromyelitis optica spectrum
disorders. Case Rep Neurol Med
2019;2019:9691863.
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- Gutierrez
C, Rodrigues S, Trillo M, Vasquez A,
Aguirre-Quispe W. Neuromyelitis optica
spectrum disorder after BIBP COVID-19 vaccine: A
case report. Neuroimmunol Rep
2023;3:100174.
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- Saroja
AO, Naik KR. Persistent Pathological Yawning
due to Neuromyelitis Optica Spectrum Disorder.
Ann Indian Acad Neurol. 2026 Jan 8.
-
- Abstract
- With the COVID-19 vaccine now available,
there have been occasional reports of
postvaccination neurological complications.
- In this report, we present a case of
neuromyelitis optica spectrum disorder (NMOSD)
that developed one month after the patient
received the second dose of BIBP COVID-19
vaccine (SARS-CoV-2-Vaccine [Vero Cell]
Inactivated). The patient presented with
itching, numbness in the hand and right side of
the face, as well as nausea, vomiting, and
hiccups. Brain MRI revelead lesions in the area
postrema, medulla, and bilateral hypothalamus.
which are typical of NMOSD. Serum antibodies to
anti-AQP4 and anti-MOG were negative.
-
- The pathogenesis of NMOSD development after
vaccination is still unknown. NMOSD is generally
aggressive and disabling, it is important for
the neurologist to be attentive to the highly
variable clinical presentation after COVID-19
vaccination for early diagnosis and effective
treatment.
-
- Avec la mise à disposition du vaccin
contre la COVID-19, des cas occasionnels de
complications neurologiques post-vaccinales ont
été signalés.
- Les auteurs présentent un cas de
trouble du spectre de la neuromyélite
optique (NMOSD) qui s'est
développé un mois après que
le patient ait reçu la deuxième
dose du vaccin BIBP contre la COVID-19 (vaccin
inactivé contre le SARS-CoV-2
[cellules Vero]). Le patient
présentait des démangeaisons, un
engourdissement de la main et du
côté droit du visage, ainsi que des
nausées, des vomissements, des
bâillements et des hoquets. L'IRM
cérébrale a
révélé des lésions
dans l'aréa postrema, le bulbe rachidien
et l'hypothalamus bilatéral, qui sont
typiques du NMOSD. Les anticorps sériques
anti-AQP4 et anti-MOG étaient
négatifs.
-
- La pathogenèse du
développement de la NMOSD après la
vaccination est encore inconnue. La NMOSD est
généralement agressive et
invalidante. Il est important que le neurologue
soit attentif à la présentation
clinique très variable après la
vaccination contre la COVID-19 afin de poser un
diagnostic précoce et de mettre en place
un traitement efficace.
-
- Introduction
- Neuromyelitis optica (NMO) is an
inflammatory, autoimmune, and demyelinating
disease that primarily affects the optic nerve
and spinal cord. The recognition of incomplete
forms of the disease and the inclusion of new
syndromes associated with AQP4 antibodies have
broadened the term to neuromyelitis optica
spectrum disorder (NMOSD) (Wingerchuketal.,
2007).
-
- NMOSD has recently been reported in certain
vaccines. Most of these cases were positive for
Aquaporin-4 (AQP4) antibodies (Choetal., 2019).
COVID-19 can present various neuroimmunological
complications, including Guillain-Barré
syndrome, NMOSD, Miller Fisher syndrome, cranial
polyneuritis, myasthenia gravis, and myelitis. A
few cases of NMOSD have been reported to have
developed during the recovery period from
COVID19 (Ghoshetal., 2021; de Ruijter etal.,
2020; Batumetal., 2022).
-
- With the recent availability of the COVID-19
vaccine, reported cases of post-vaccination
transverse myelitis have raised concerns about
vaccine safety (Gossetal., 2021). We present a
case of a previously healthy woman who developed
NMOSD after receiving the second dose of
inactivated virus vaccine against COVID-19 (BIBP
COVID-19 vaccine).
-
- Case report
- A 51-year-old woman who was previously
healthy presented with itching and numbness in
her right hand and face, along with excoriative
lesions in the right nasolabial fold, one month
after 1 month after receiving the second dose of
BIBP COVID-19 vaccine (inactivated
SARS-CoV-2-Vaccine - Vero Cell), a Sinopharm
COVID-19 vaccine. Her symptoms worsened
gradually and insidiously, and she subsequently
experienced nausea, uncontrollable vomiting and
hiccups, leading to her admission to our
hospital's emergency room.
-
- During her hospitalization stay, the patient
developed diplopia, excessive yawning,
hypersomnia, constipation, bladder retention,
left-predominantly suspended paraparesis, and
right palpebral ptosis. A clinical examination
upon admission revealed excoriative lesions in
the right nasolabial fold. Ten days after
hospitalization, she had paraparesis with a
sensory level at T10, left internuclear
ophthalmoplegia, predominantly right bilateral
palpebral ptosis, right Babinski sign, patellar
areflexia and Achilles hyporeflexia. Her
Ishihara test was normal. Several days later,
she presented with venous thrombosis in the
lower left limb, most likely related to
prostration.
-
- Brain MRI showed a lesion in the area
postrema and bilateral hypothalamus without
enhancement with gadolinium (Fig. 1). After two
weeks, an MRI of the cervical and dorsal spine
was performed with sagittal T2-weighted images
of the total spine, wich revealed focal lesions
of high signal intensity in the cervical segment
of the medullary cord at the level of C3-C6 and
the medullary-bulb junction. Additionally, the
dorsal segment of the spinal cord showed several
focal high signal intensity lesions on 12 and T2
STIR up to 40mm in length at the level of T10
and T11, most of which compromised the central
region of the medullary cord and without
evidence of contrast enhancement. These findings
were compatible with transverse mvelitis (Fig.
2).
-
- The cerebrospinal fluid was transparent,
with 19 cells/uL and a mononuclear predominance
of 100%. The glucose level was 64mg/dL, and the
protein level was 83.8 mg/dl. The
electromyography was normal, and the optic
nerves were normal on MRI. However, the
visual-evoked potentials showed decreased
amplitudes symmetrically with a normal latency
for the P100 wave of both optic nerves. .
- Serum antibodies against aquaporin-4
(IgG-AQP4) and myelin oligodendrocyte
glycoprotein (IgG-MOG) were negative. Routine
blood, liver and kidney function, vitamin B12
levels, tumor markers, erythrocyte sedimentation
rate, antinuclear antibody profiles, C-reactive
protein, rheumatoid factor, antiphospholipid
antibodies, ANA, SSA, SSB, and pANCA were all
negative.
-
- Pulmonary tomography revealed a
non-obstructive hypodensity of 13_5mm in the
right branch of the pulmonary artery, which
extended to the right inferior, middle and
superior lobar arteries, reported as a probable
tomographic sign of thrombosis, however the
patient did not develop respiratory
symptoms.
-
- The patient fulfilled the diagnostic
criteria for neuromyelitis optica spectrum
disorder without AQP4-IgG, as she presented with
brainstem syndrome, area postrema syndrome,
longitudinally extensive acute myelitis with
cervical involvement of C3-C6, dissemination in
space as evidenced by MRI. Other pathologies
were ruled out. Following treatment with
methylprednisolone (500mg for 5 days), the
patient showed improvement in symptom.
-
- Discussion
- NMOSD is an demyelinating, autoimmune,
inflammatory disease of the central nervous
system, distinguished from multiple sclerosis.
NMOSD involves the optic nerve as recurrent
optic neuritis and the spinal cord as acute
myelitis. The relationship between vaccination
and demyelination, particularly NMOSD, is rare
and not entirely understood. It is often
attributed to the body's autoimmune overreaction
against the vaccine's antigen. Previous case
reports have indicated that acute demyelinating
encephalitis is the most frequent demyelinating
disease, occurring more frequently in children
(Karussisand Petrou,2014).
-
- A recent literature review from the last
decade found that the most common
vaccineassociated demyelinating diseases were
diagnosed with acute disseminated
encephalomyelitis (ADEM) and NMOSD, at 44% and
9%, respectively (Kumaretal., 2020). The most
common reported side effects of COVID-19
vaccines have been localized pain at the
injection site, low-grade fever, fatigue,
myalgia, chills, and joint pain, and rarely,
anaphylactic shock (Kauretal., 2021).
-
- The reported case is the first known case in
Peru of NMOSD that developed after the second
dose of the attenuated COVID-19 vaccine. NMOSD
cases have already been reported worldwide, with
one case developing NMOSD two months after the
attenuated virus application against COVID-19
(Chenetal., 2021) and another case only ten days
after (Fujikawaetal., 2021). Our patient
developed NMOSD after one month. It has been
reported not only with attenuated viruses but
also with other types of vaccines
(Fujikawaetal., 2021; Badrawietal., 2021).
-
- The clinical presentation of acute brainstem
syndrome, with pruritus as the initial symptom,
was striking in our patient, a presentation that
has been described in NMOSD before (Pruritusas
an initial symptom of neuromyelitis optica
spectrum disorder,2022). The following week,
transverse myelitis was added, which delayed
early diagnosis on admission to the hospital. In
Peru, cases of multiple sclerosis with initial
symptoms of the brainstem have been reported,
which could have been the reason for initially
suspecting this pathology
(Caparó-Zamalloaetal., 2021). However,
the evolution and type of spinal cord lesions
observed in magnetic resonance imaging after two
weeks of the initial MRI clarified the
diagnostic doubt.
-
- The seropositive form of NMOSD, defined by
the presence of antibodies against AQP4,
accounts for approximately 80% of cases. In a
proportion of seronegative cases, other
pathogenic antibodies are found, against myelin
oligodendrocyte glycoprotein (MOG). Although our
case was negative for both antibodies, the
patient met the diagnostic criteria, and it was
decided to start treatment, as recommended in
seronegative cases (Jariusetal., 2016).
-
- De novo cases following vaccination against
different diseases appear more frequently
IgG-AQP4 negative, while 75% of relapsers are
IgG-AQP4 antibody seropositive, and primarily
occur in white women (Vanoodand Wingerchuk,2019;
Mealyetal., 2018).
-
- NMOSD can coexist with other autoimmune
disorders, such as Sjögren's syndrome (Alva
Díaz etal., 2016), but we did not find
any markers of an associated autoimmune disease.
Additionally, our patient presented with venous
thrombosis of the left lower limb, and NMOSD is
known to be a risk factor for venous
thromboembolism (Farberetal., 2017). She also
presented an alteration of the pulmonary
tomography without clinical translation, and an
interstitial pulmonary lesion has been described
since the Aquaporins4 are found in large
quantities at the pulmonary level (Asato etal.,
2018).
- Further studies may be necessary to clarify
the possible link between COVID-19 vaccination
and the development of NMOSD. However, both at
the individual and population levels, the
benefits of the COVID-19 vaccine far outweigh
the risks of a neurological complication. Still,
it would be important to consider the
benefit-risk balance in patients with a
diagnosis of NMOSD before vaccination.
-
- Conclusion
- The presented case of NMOSD developed
following administration of the inactivated Vero
Cell vaccine against SARS-CoV-2, is significant.
It contributes to expanding our understanding of
the specific COVID-19 vaccines that are
associated with NMOSD and highlights the
importance of early diagnosis and timely
intervention for a disease that can have
catastrophic consequences.
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