Temporal Bone Endometriosis — A Multidisciplinary Approach. A Clinical Case

Introduction
Even though the ethiology of various
types of temporomandibular joint dysfunction is
still debatable, certain generally accepted factors
are identified in the dental community non the
less. Chief among these factors has always been
malocclusion (incorrect interdental contacts
between teeth in static or dynamic), which led to
a CO-CR dyscrepancy (that is, the mismatch of
the jaw’s position in the usual bite and in the
musculoskeletal stable ratio, in which the
articular condyles are in the correct position in
the articular fossae, and the maxillofacial
muscles are in the correct uniform tone), causing
the lower jaw to move into a forced position and
producing the corresponding symptoms. Many
other causes are also identified, such as
neurological disorders, postural disorders,
traumatic lesions, etc. However, only rare
sources mention that dysfunctions often arise
from dyshormonal conditions1
.
Only a few studies have mentioned that
fibrous cartilage, covering articular surfaces, has
estrogen receptors, responsible for the normal
conduction of synovial fluid through cartilage and,
consequently, its normal trophism2,3
.
Therefore, considering the joints from a
non-dental side is uncommon and is thought
unprovable in the dental world. However, as
practice shows, approximately 85% of
dysfunctions are of a polyetiological nature, quite
often not related to dentistry at all.
The presented case illustrates a rare joint
pathology mainly caused (despite real dental
etiological factors) by endometriosis of the
temporal bone (namely, the articular tubercle).
The case is truly unique since no such condition
has yet been described.
*Corresponding author:
Nazarii Brotskyi,
Private practice, Kyiv, Ukraine.
E-mail: brotskyi.n@gmail.com
Journal of International Dental and Medical Research ISSN 1309-100X Temporal Bone Endometriosis
http://www.jidmr.com Nazarii Brotskyi and et al
Volume ∙ 15 ∙ Number ∙ 3 ∙ 2022

Page 1306
Materials and methods
Patient Information
Patient E. sought medical assistance at a
dental clinic with complaints of constant acute
pain in the left TMJ (temporomandibular joint),
which intensified with any movement of the
mandible, the inability to open her mouth wide,
and constant headaches. Complaints began four
years ago when the patient first felt a crunch and
discomfort in the joint, which increased over time.
The patient could not identify a possible cause.
As a variant of the reason, she named wearing a
bracket system in 2010, but an asymptomatic
period of 5 years did not allow her to hold that.
The patient underwent several courses of splint
therapy with different doctors who tried to
rehabilitate her in the concept of Rudolf Slavicek
by anterization (shifting the jaw to the anterior
therapeutic position), which, however, did not
bring even temporary relief.
Figure 1. a) Frontal facial photos; b). Side facial
photos; с) Intraoral photos.
According to the patient, she was
somatically healthy, with negative family
anamnesis.
Clinical Findings
Objective examination by a dentist
revealed:
Horizontal type of the skull bone growth
2nd class, 1st subclass according to
Engle
VDO reduction
Chin shift to the left
Micrognathia of the lower jaw
Counterclockwise transversal rotation of
the upper jaw
Extrusion of 37 and 47 teeth
Front teeth protrusion of both jaws
No interdental contact in the frontal area
Enlargement of the nasolabial angle (see
Figure 1, Figure 2)
Opening the mouth by 28 mm with
deflection to the left
Palpation: pain in the temporomandibular
joints in 8 zones according to Mariano Rocabado;
bilateral pain in masticatory, temporal, medial
pterygoid, lateral pterygoid, mandibulohyoid,
digastric muscles, more significant on the left;
pain in the left trapezius and sternocleidomastoid
muscles.
Figure 2. Orthopantomography, teleradiography,
cephalometry.
Figure 3. Computed tomography and magnetic
resonance imaging of the TMJ. a. Right TMJ. b.
Left TMJ.
Diagnostic Assessment
CBCT of the temporomandibular joint with
a closed mouth revealed a posteroinferior
position of the right condyle, degenerative joint
disease of the right joint in the form of regressive
remodelling, posteroinferior position of the left
Journal of International Dental and Medical Research ISSN 1309-100X Temporal Bone Endometriosis
http://www.jidmr.com Nazarii Brotskyi and et al
Volume ∙ 15 ∙ Number ∙ 3 ∙ 2022

Page 1307
condyle, degenerative joint disease of the left
joint in the form of progressive remodelling
(formation of exostosis on the articulating surface
of the condyle in the form of a spike).
MRI of the TMJ without contrast showed
an incomplete anteromedial dislocation of the
articular disc of the right TMJ with reduction,
synovitis of the right TMJ; complete anteromedial
dislocation of the left TMJ articular disc without
reduction and block, synovitis of the left TMJ (see
Figure 3).
Therapeutic Intervention
The patient was prescribed a combined
treatment:
1. Registration the “bite do jour” position
using the front Okeson deprogrammer (see
Figure 4).
Figure 4. Centric do jour.
2. Fabrication of a three-section
myorelaxant splint for the upper jaw to be worn
according to the standard scheme (posterior
segment — around the clock, anterior segment
— during sleep). The period of wearing the splint
was not determined since it was necessary not
only to remove the symptoms but also to remodel
the articular heads to a stable form.
3. Drug therapy: Arcoxia (etoricoxib) 60
mg, 2 times a day for 14 days; Omez
(omeprazole) 20 mg, 2 times a day for 14 days;
Myorix (cyclobenzaprine hydrochloride) 15 mg, 2
times a day for 7 days.
Significant relief was observed on the 3rd
day after the splint therapy started, and on the
14th day, the symptoms disappeared completely.
By grinding the splint’s occlusal contacts, total
muscle relaxation and asymptomaticity of the
stomatognathic system were achieved.
Figure 5. The situation on the 5th month of splint
therapy. a. Right TMJ. b. Left TMJ. c. Gypsum
coating.
However, on the fifth month of treatment
(see Figure 5), the patient complained of pain
along the back of the neck, so she was sent to a
course of therapeutic massage and exercise
therapy to eliminate it. The patient felt significant
relief, but a small part of the symptoms remained,
so she arbitrarily decided to go to a chiropractor.
According to the patient, her cervical and lumbar
vertebrae were manually reset, which led to
acute pain and the inability to maintain postural
balance. Pain in the TMJ and muscles returned
to the initial level.
It was decided to send the patient for a
consultation with a neurosurgeon, who
prescribed her drug therapy (dynostat
(parecoxib), mydocalm (Tolperisone),
veroshpiron (spironolactone)). The therapy
brought little relief but caused acute allergic
dermatitis, so it was prematurely terminated.
All this time, the patient was morally
depressed and reacted aggressively to any
treatment.
Given the recurrence of pain and the
ineffectiveness of this type of splint therapy, it
was decided to transfer the patient to a full
myorelaxant splint on the lower jaw with incisor
and canine guidances on the 8th month of
treatment (see Figure 6).
Figure 6. The situation on the 8th month of splint
therapy. a. Right TMJ. b. Left TMJ. c. Gypsum
coating.
Journal of International Dental and Medical Research ISSN 1309-100X Temporal Bone Endometriosis
http://www.jidmr.com Nazarii Brotskyi and et al
Volume ∙ 15 ∙ Number ∙ 3 ∙ 2022

Page 1308
Wearing a new splint relieved the
patient’s symptoms on the seventh day, but mild
pain persisted and periodically increased
substantively for several days.
After 2.5 months of wearing the new splint,
the patient began to complain of sudden sharp
pain in the lower abdomen and was taken to the
admission department with a preliminary
diagnosis of acute appendicitis. In the admission
department, this diagnosis was ruled out and the
patient was hospitalized in the gynaecological
department. It turned out that the patient was
suffering from painful menstruation since
menarche, which was corrected by NSAIDs.
According to the anamnesis, the patient was last
examined by a gynaecologist 1.5 years ago,
when she was diagnosed with algodysmenorrhea
and prescribed Yaryna (a combined estrogenprogestogen contraceptive — Ethinyl estradiol
0.03 and drospirenone 3 mg).
In the gynaecological hospital, the pelvic
ultrasound detected endometrioid cysts of both
ovaries, approximately 3.0 cm in diameter, for the
first time. After correction of the pain syndrome,
the patient was discharged and prescribed
Regulon (a combined estrogen-progestogen
contraceptive — desogestrel 0.15 mg and Ethinyl
estradiol 0.03 mg) for up to 6 months.
The patient took Regulon for two months,
but the clinical picture had not changed. It was
decided to send the patient to the second
consultation with a gynaecologist, who stopped
taking Regulon and transferred her to Dienogest
2 mg.
Despite the expected worsening of
articular symptoms, which is a common side
effect of taking progestogens, the pain syndrome
completely disappeared. Normalization of TMJ
function was obtained on the 14th month of
treatment (see Figure 7).
Figure 7. The situation on the 14th month of
splint therapy. a. Right TMJ. b. Left TMJ. c.
Gypsum coating.
Due to this phenomenon, a retrospective
study of the time of the patient’s pain symptoms
intensification was conducted, which revealed
that periods of exacerbation during the treatment
most often occurred on the 3rd-6th days of the
menstrual cycle. Also, control TMJ CT scans
performed during splint therapy revealed a cavity
in the articular tubercle, which was empty at the
beginning and had a blackout inside upon the
CBCT scan made in the first days of the
menstrual cycle (see Figure 8).
Figure 8. The TMJ CT dynamics. The arrow
indicates the area of interest. а. Right TMJ. b.
Left TMJ.
For clinical verification of the diagnosis, it
was decided to make a control MRI of the TMJ
without contrast on the 3rd day of the menstrual
cycle, which revealed a cavity in the articular
tubercle of the left TMJ filled with a large amount
of fluid (see Figure 9).
Clinical data, the cyclicity of
exacerbations associated with the menstrual
cycle, data from additional research methods and,
most importantly, the response to Dienogest
therapy let us conclude that extragenital
endometriosis was the cause of articular
dysfunction.
Journal of International Dental and Medical Research ISSN 1309-100X Temporal Bone Endometriosis
http://www.jidmr.com Nazarii Brotskyi and et al
Volume ∙ 15 ∙ Number ∙ 3 ∙ 2022

Page 1309
Figure 9. The MRI of the TMJ at the end of splint
therapy. The arrow indicates the area of interest.
а. Right TMJ. b. Left TMJ.
Results
Figure 10. Ultrasound imaging of ovaries with
endometrioid cysts. Typical appearance: rounded
hypoechoic unilocular masses with hypoechoic
‘ground glass’ content.
After 6 months of taking Dienogest,
menstruation became less painful, but there was
no amenorrhea induced by this drug. Despite the
expected worsening of articular symptoms, which
is a common side effect of taking progestogens,
complete disappearance of pain and involution of
the formation on the left articular head were
achieved within three months.
According to the pelvic ultrasound, there
is a decrease in the size of ovarian cysts from the
initial level (approximately 30.0 mm in diameter)
at the time of the follow-up examination: 19.0 x
14.0 x 19.0 mm in the right ovary and 8.0 x 8.0
mm. in the left one (see Figure 10).
Considering the insufficient effectiveness
of Dienogest in the management of pelvic pain
and the lack of reproductive plans of the patient
in the nearest future (she planned pregnancy in
1-2 years), it was decided to prolong the use of
Dienogest until the start of pregnancy planning,
and also to administer Goserelin 10.8 mg s/c.
Discussion
Endometriosis is a condition in which the
endometrium (inner layer of the uterus) is present
in organs other than the uterus itself. According
to the latest systematic review, the incidence of
endometriosis in the population ranges from 0.8
to 28.6% with an overall estimate of 4.4%. When
analyzing distinct populations, endometriosis was
found in 33.5% of women undergoing surgery for
benign gynaecological conditions, in 23.8% of
infertile women, and in 49.7% of women with
chronic pelvic pain4,5.
Endometriosis is an estrogen-dependent
inflammatory disease that causes pelvic pain and
uterine bleeding and often leads to infertility6
.
Endometriosis usually occurs in the
myometrium (also called adenomyosis), ovaries
(endometrioid cysts), ligamentous apparatus of
the uterus and on the surface of the peritoneum,
less often in the intestines, bladder, ureter,
abdominal wall, chest cavity, and other organs7
.
We should note that statistical data on the
frequency of this disease relates to pelvic
endometriosis, and there are no statistical data
on extragenital endometriosis, given the rarity of
this pathology, the diversity and unpredictability
of localizations, and the frequent mimicry of other
pathological conditions8
.
Very often, the only symptom that can
help in the formation of the diagnosis is the
cyclicity of clinical manifestations (associated
with both the pathophysiology of endometriosis
and the affected organ)9
. So, for example, with
endometriosis of the chest, catamenial
hemothorax or hemoptysis occurs. Recurrent
pneumonia is also possible. With endometriosis
Journal of International Dental and Medical Research ISSN 1309-100X Temporal Bone Endometriosis
http://www.jidmr.com Nazarii Brotskyi and et al
Volume ∙ 15 ∙ Number ∙ 3 ∙ 2022

Page 1310
of the navel, the formation begins to increase and
hurt before and during menstruation10.
That is why multidisciplinary collaboration
is so substantial in the diagnosis and
management of this condition.
The only guideline regarding extragenital
endometriosis published in Japan in 2020
concludes that medical therapy is effective in the
treatment of rectosigmoid endometriosis, bladder
endometriosis, catamenial haemoptysis (when
symptoms are mild or moderate); surgical
treatment: endometriosis of the intestine or ureter
with hydronephrosis, endometriosis of the navel
(however, medication can also be considered)11.
At the moment, the treatment of
endometriosis lies in the suppression of ovulation
to create a hypoestrogenic state and
decidualization of endometrioid heterotopias,
which can be achieved by continuous use of
progestogens and, in severe cases, GnRH
agonists12.
In this case, as in most cases of
extragenital endometriosis that we observed, it
was the cyclical nature of the clinical
manifestations of TMJ dysfunction that was
detected retrospectively, the simultaneous
presence of genital endometriosis, and the
effectiveness of Dienogest, intended for its
treatment, in reducing TMJ pain, prompted
thought about endometriosis of the temporal
bone13.
This clinical case represents a unique
variety of extragenital endometriosis —
endometriosis of the temporal bone. This once
again reminds us of the "omnipresence" of this
condition and the need to include a range of
gynaecological diseases in the differential
diagnosis of maxillofacial pathology.
Conclusions
Temporomandibular joint dysfunction is a
complex polyetiological disease, often associated
with non-dental causes. Therefore, a systematic
assessment of the patient’s body is a critical part
of the examination, which allows establishing the
correct diagnosis and choosing the right
treatment tactics.
At the same time, extragenital
endometriosis can theoretically affect any organ
system, causing characteristic, often non-specific
symptoms. Therefore, understanding the
specifics of the pathology of all body systems is
important for a gynaecologist in the diagnosis
and treatment of extragenital endometriosis.
Declaration of Interest
The authors report no conflict of interest.