Headache Medicine 2021, 12(1) p-ISSN 2178-7468, e-ISSN 2763-6178

ASAA

DOI: 10.48208/HeadacheMed.2021.12

Headache Medicine

Case Report

Persistent idiopathic facial pain associated with borderline personality

disorder: a case report

Hugo André de Lima Martins

Bruna Bastos Mazullo Martins

Camilla Cordeiro dos Santos

Djanilson Jose Pontes

Daniella Araújo de Oliveira

Marcelo Moraes Valenca

4,5

Unidade do Cérebro, Surubim, Pernambuco, Brazil.

Esuda Faculty, Recife, Pernambuco, Brazil.

Departmente of Physiotherapy, Universidade Federal e Pernambuco, Recife, PE, Brazil.

Neurosurgery Unit, Universidade Federal de Pernambuco, Recife, Pernambuco, Brazil.

Unimed Recife, Recife, Pernambuco, Brazil.

Abstract

Introduction

Borderline personality disorder may be associated with persistent facial pain since its relationship with

different pain syndromes has been reported. Persistent idiopathic facial pain is commonly unilateral, pulsa-

ting, burning, or profound and challenging for clinicians. Therefore, excluding underlying organic causes

by appropriate clinical investigation and complementary tests is essential to diagnose this disease.

Objective

This case report aimed to provide evidence of the relationship between idiopathic persistent facial pain

and borderline personality disorder.

Case report

A 24-year-old woman reported severe pain in the left hemiface for ten months, three to six hours per

day, ve days per week. No abnormalities were found in dental and neurological assessments. A psy-

chiatric evaluation was performed, and the patient met the criteria for borderline personality disorder.

Pharmacological treatment consisted of daily lithium carbonate (900 mg) and venlafaxine (150 mg).

Weekly sessions of cognitive-behavioral therapy with emotional regulation and tolerance to stress were

performed. The patient was evaluated every 30 days and showed improved pain intensity and frequency

over six months.

Conclusion

Proper management of borderline personality disorder can modify the evolution of persistent idiopathic

facial pain when both pathologies are comorbidities.

Hugo André de Lima Martins

Rua Antônio Medeiros Sobrinho,

275, Cabaceira, Surubim-PE,

Brasil

hugomt2001@yahoo.com.br

Edited by:

Marcelo Moraes Valença

Keywords:

Cognitive Behavioral Therapy

Facial Pain

Borderline Personality Disorder

Lithium Carbonate

Venlafaxine Hydrochloride

Received: July 4, 2021

Accepted: July 30, 2021

ASAA

Martins HAL, Martins BBM, Santos CC, Pontes J, Oliveira DA, Valença MM

Persistent idiopathic facial pain associated with borderline personality disorder: a case report

Introduction

ersistent idiopathic facial pain (PIFP) is a chronic disease of

unknown etiology. Patients usually complain of unilateral pain

affecting the maxillary region, usually described as burning, pul-

sating, profound, or painful. However, as described in Figure 1,

PIFP neither meet criteria for neuralgia nor present objective signs of

neurological changes on imaging or clinical examination or dental

causes after a detailed investigation.

Furthermore, few controlled

studies demonstrated these patients might present a poor clinical

response to antidepressant use (e.g., amitriptyline and nortriptyline)

and improved quality of life when associated with psychotherapy.

Association of PIFP and depression, anxiety symptoms, and poor

quality of life are well documented in previous studies, although only

a few studies about personality traits were performed.

2,3

A. Facial and/or oral pain fullling criteria B and C.

B. Recurring daily for >2 hours/day >3 months.

C. Pain has both of the following characteristics:

1. Poorly localized and not following distribution of peripheral nerve.

2. Dull, aching, or nagging quality.

D. Clinical neurological examination is normal.

E. Dental cause has been excluded by appropriate investigations.

F. Not better accounted for by another ICHD -3 diagnosis.

Figure 1. Persistent idiopathic facial pain diagnostic criteria (ICHD-3)

A set of stable individual characteristics (i.e., specic functioning

patterns) is formed by a set of stable individual characteristics

with a personal manner of perceiving oneself, other people, and

life events.

However, a personality disorder might be present

when this pattern is rigid, pervasive, inexible, and maladaptive.

Personality disorders affect approximately 10% of the population,

with borderline personality disorder (BPD) corresponding to half

of this content.

Among all personality disorders, BPD is the most related to painful

syndromes.

Table 1 shows the

Diagnostic and Statistical Manual

of Mental Disorders

(DSM-5) diagnostic criteria for BPD (ve out of

nine criteria are required for diagnosis). BPD is characterized by

severe changes in cognition, emotion, behavior, and interpersonal

relationships. Self-injurious behaviors are frequent, such as hair

pulling, slapping, throwing on the oor, hitting head on the wall,

cutting, or scratching. Non-lethal attempt methods sometimes

follow suicidal ideation (e.g., excessive medication-taking at once)

or violent methods, such as hanging, jumping from high places,

throwing themselves in front of automobiles at high speed, and

self-harm with knives or rearms.

6-8

Studies observed a 30% prevalence rate of BPD among patients

with chronic pain. Pain scores are higher in patients with chronic

pain and BPD than those patients without BPD.

One study

evaluated 777 patients undergoing pain rehabilitation and

observed more pain complaints in those with BPD.

In a study

evaluating patients with BPD found more painful syndromes (e.g.,

lumbar pain, temporomandibular joint [TMJ] disorders, and

bromyalgia) in patients with active BPD symptoms than patients

with remission of BPD symptoms.

Table 1 Diagnostic and Statistical Manual of Mental Disorders (DSM-5) diagnostic

criteria for borderline personality disorder

1. Frantic efforts to avoid real or imagined abandonment (Note: Do not

include suicidal or self-mutilating behavior covered in Criterion 5);

2. Pattern of unstable and intense interpersonal relationships characterized by

alternating between extremes of idealization and devaluation;

3. Identity disturbance: markedly and persistently unstable self-image or sense

of self;

4. Impulsivity in at least two areas that are potentially self-damaging (e.g.,

spending, sex, substance abuse, reckless driving, binge eating) (Note: Do not

include suicidal or self-mutilating behavior covered in Criterion 5);

5. Recurrent suicidal behavior, gestures, threats, or self-mutilating behavior;

6. Affective instability due to a marked reactivity of mood (e.g., intense episodic

dysphoria, irritability, or anxiety usually lasting a few hours and only rarely

more than a few days);

7. Chronic feelings of emptiness;

8. Inappropriate, intense anger or difculty controlling anger (e.g., frequent

displays of temper, constant anger, recurrent physical ghts);

9. Transient, stress-related paranoid ideation or severe dissociative symptoms.

Clinical observations showed that patients with BPD are more

intolerant to chronic pain.

Brain imaging studies in BPD

demonstrated top-down dysregulation in emotional control

inuencing chronic pain signaling.

7,12

Some neural structures in

the fronto-limbic region are also altered in BPD. Studies using

functional magnetic resonance imaging demonstrated increased

amygdala reactivity and reduced activity in the prefrontal cortex

and anterior area of the cingulate gyrus, regions involved in

abnormal pain processing.

Therefore, this study aims to report a patient with PIFP and BPD

who did not respond to treatments before BPD diagnosis and

showed good clinical evolution after proper psychotherapeutic

BPD management associated with psychopharmacological

treatment for mood stabilization.

Case report

This study was conducted at the Brain Unity Clinic in Surubim-PE

(Brazil) and approved by the human research ethics committee

(protocol number 464456921.4.000.5640). This case report

concerns a 24-year-old woman with a history of severe pain on

the left side of the face for ten months. She reported diffuse pain

without specic exacerbation points lasting three to six hours, ve

days per week. She denied dental procedures before pain onset

and attended several dental evaluations because she believed it

could be a dental problem. TMJ imaging by magnetic resonance

(MRI) and arthroscopy showed nonspecic changes and did not

elucidate the cause of pain. She was also evaluated and treated

by a neurologist, who did not encounter any abnormalities after

a detailed neurological examination of facial sensitivity. Brain

MRI was also normal. She used daily 50 mg of amitriptyline,

carbamazepine (600 mg), and gabapentin (900 mg) during

previous treatments.

ASAA

Martins HAL, Martins BBM, Santos CC, Pontes J, Oliveira DA, Valença MM

Persistent idiopathic facial pain associated with borderline personality disorder: a case report

The patient mentioned two events that negatively impacted her

life. During childhood (ve years old), the rst happened when

she was run over and hospitalized for several days for medical

observation, which psychically affected her. The other event was

her mother's absence during baptism, triggering an intense belief

of rejection, despite becoming aware of this fact several years

later after other people telling her.

In the rst evaluation, the patient complained about severe

mood instability, intense feelings of emptiness, frequent crisis

of unmotivated anger, suicidal ideation, frantic efforts to avoid

abandonment, and severe binge eating as emotional regulation

(i.e., six out of nine criteria used for BPD diagnosis). During a

mental evaluation, the patient was conscious and auto- and

allopsychically oriented, anxious, with mood lability, reduced

attention and concentration, presented hypomnesia for recent

events, organized thinking, and no sensoperceptual alterations.

Pharmacological treatment with lithium carbonate initiated with

300 mg at night, until reaching 900 mg per day, was associated

with venlafaxine (initial dose of 37.5 mg per day and nal dose of

150 mg after 30 days). Serum lithium dosage was 0.7 millimole

per liter during treatment. Weekly sessions of psychotherapy were

also indicated with training elements for emotional regulation and

tolerance to stress.

Furthermore, four inventories were used to quantify mental changes

in a psychiatric evaluation. For Borderline Symptom List 23 (BSL-

23), an inventory with a list of borderline symptoms, the patient

achieved a total score of 44/92 of possible symptoms at initial

evaluation and 29/92 after six months of treatment. For Beck

Depression Inventory (BDI), the score was 32 at initial evaluation

and 13 after six months of treatment. For Beck Anxiety Inventory

(BAI), the score was 22 at initial evaluation and nine after six

months of treatment. Finally, she achieved an initial score of 6/20

in the Subjective Memory Complaints scale (SMCs), decreasing to

2/6 after six months of treatment. The patient was evaluated every

30 days and improved pain intensity and frequency gradually

over six months. Pain intensity was assessed using the eleven-point

(0-10) visual analog scale and improved during this period (from

eight at initial evaluation to two after six months of treatment)

(Figure 2).

Figure 2. Evolution of pain symptoms on visual pain scale.

Discussion

This study reports a patient diagnosed with persistent and refractory

facial pain investigated for personality disorder. Such comorbidity

directly inuences PIFP treatment since it changes the therapeutic

approach by focusing on pharmacological and psychotherapeutic

treatments, most indicated in personality disorders.

Pain is a sensory and emotional experience that may become

chronic after six months, leading to loss of function and severe

disability.

PIFP is probably the most challenging type of facial

pain for clinical management. A multidisciplinary team approach

helps identify social, environmental, and intrapsychic factors

inuencing patient evolution.

Herein, the patient presented a distorted belief of affective bond

with her mother because her mother did not attend the baptism.

For this reason, the patient developed a trait of insecurity and

frequent fear of being abandoned, which is characteristic of BPD

patients.

Existential emptiness reported was probably because

she thought other people were invalidating her suffering, which is

reinforced by lack of ndings in complementary exams.

In the past, PIFP was called psychogenic facial pain. A study

from the United Kingdom evaluated 150 patients with PIFP and

observed that three out of 55 patients excluded were referred

to psychiatric hospital admission, and four patients decided to

search for psychiatric care at another service.

As 20% to 25% of

patients admitted to psychiatric hospitals have BPD, it would not be

surprising if some of these patients with PIFP also presented severe

emotional dysregulation. Another study assessing the personality

prole of three patients with PIFP using Cloninger's Temperament

and Character Inventory found common characteristics among

patients (egocentrism, high levels of insecurity, shame, and

harm avoidance), suggesting exaggerated attention to pain and

oneself, which may play a fundamental role in chronicity and

resistance to treatment.

Depression is present in more than 80% of patients with BPD.

The clinician must identify the correlation between depression

and BPD since treatment with antidepressants alone may not be

effective. Anxiety is also commonly found in patients with BPD.

BPD and anxiety may not only co-occur but also inuence the

therapeutic response of each other.

Our patient presented many

depressive and anxiety symptoms at the beginning of treatment

and improved signicantly after six months of follow-up, as shown

by the marked decrease in BDI and BAI scores.

Patients with BPD frequently complain about memory problems

during daily activities, although this nding is not present in

memory tests.

The patient of the present study partially improved

subjective memory complaints throughout treatment, as evidenced

by SMC. Memory complaints in patients with BPD are probably

from the negative self-perception of these patients; therefore,

improving affective dysregulation may also improve memory.

ASAA

Martins HAL, Martins BBM, Santos CC, Pontes J, Oliveira DA, Valença MM

Persistent idiopathic facial pain associated with borderline personality disorder: a case report

Know about emotional dysregulation is crucial to understand

BPD.

The patient described in this study showed severe emotional

instability, crises of unmotivated anger, and binge eating. Many

patients with BPD manifest emotional dysregulation by alcohol

and drug abuse, leading to substance dependence. Other

patients manifest this dysregulation in the sexual area (promiscuity

conditions), food area (binge eating), or pain area (chronic pain).

In this study, the patient manifested characteristics of binge eating

and chronic pain.

The interplay between BPD and pain is complex. For example,

patients with this disorder are insensitive to acute pain, making

supercial skin cuts during self-mutilation episodes. On the other

hand, clinical studies show that these patients are more sensitive

to chronic pain than individuals without personality disorders.

Therefore, evaluating borderline traits in patients with PIFP is

essential because patients with BPD symptoms remission present

pain relief and less analgesic use, which are essential for pain

chronication.

9,10

The most important symptom of the studied patient was persistent

suicidal ideation, one of the most notable characteristics of BPD.

This symptom guided lithium (mood stabilizer with high anti-suicide

action) as pharmacological choice

18,19

associated with venlafaxine

(dual-action antidepressant). Moreover, these drugs act in the

serotoninergic pathway by decreasing impulsivity, aggressiveness,

and the risk of suicide.

However, lithium must be within the

therapeutic window due to its high lethality rate.

Psychotherapy was indicated for cognitive restructuring and

behavioral change since the patient was diagnosed with a

personality disorder. The patient changed her main beliefs

regarding her and the outside world using adequate cognitive and

behavioral techniques as muscle relaxation and diaphragmatic

breathing. Emotion regulation and stress tolerance skills were

also provided, contributing to managing inappropriate anger and

compulsive eating.

The association between pharmacological treatment for main

psychiatric symptoms and psychotherapeutic follow-up probably

improved the patient's facial pain. Thus, this study highlights the

association between BPD and PIFP. Many PIFP cases may remain

unsolved due to the lack of a detailed assessment regarding the

meaning of pain of each patient. Ideally, all patients diagnosed

with PIFP presenting some degree of refractoriness to usual

treatment should be evaluated by a mental health team, increasing

treatment efcacy.

Hugo Andre de Lima Martins

https://orcid.org/0000-0002-5216-2074

Bruna Bastos Mazullo Martins

https://orcid.org/0000-0002-6251-992X

Camilla Cordeiro dos Santos

https://orcid.org/0000-0002-5094-7337

Djanilson Jose Pontes

https://orcid.org/0000-0001-9578-5805

Daniella Araújo de Oliveira

https://orcid.org/0000-0002-6013-978X

Marcelo Moraes Valenca

https://orcid.org/0000-0003-0678-3782

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