**1. Trichotillomania**

#### **1.1. Introduction**

Trichotillomania is the disorder of repetitively pulling out one's hair from different areas of the bodythatresults innoticeablehairloss [1].Thename trichotillomaniawasgiventothisbehavior byaFrenchdermatologist,FrancoisHenriHallopeau,in1889;however,thedisorderalsoappears

© 2017 The Author(s). Licensee InTech. This chapter is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. © 2017 The Author(s). Licensee InTech. This chapter is distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

in the literature in the works of Hippocrates. It is even found in plays by William Shakespeare such as Romeo and Juliet and The Life and Death of King John. The name is derived from the combination of Greek *thrix* for hair; *tillein*, plucking; and *mania*, madness.

Trichotillomania is classified under the obsessive‐compulsive and related disorders along with hoarding disorder, skin‐picking disorder (excoriation) and body dysmorphic disorder in the *Diagnostic and Statistical Manual of Mental Disorders* Fifth Edition (DSM‐5; American Psychiatric Association, May 2013) [2] (**Table 1**). It is also grouped under obsessive‐compulsive disorders in the eleventh revision of the World Health Organization's International Classification of Diseases and Related Health Problems (ICD‐11) [3].


• The hair pulling is not better explained by the symptoms of another mental disorder

**Table 1.** Diagnostic criteria (DSM‐5) [2].

#### **1.2. Epidemiology**

Trichotillomania occurs more frequently in females. The lifetime prevalence is 3.4% for women and 1.5% for men [4]. The typical age of onset is between 5 and 12 years or early childhood to adolescence, but it may occur in any age.

#### **1.3. Pathogenesis**

The onset of trichotillomania often occurs after a stressful event such as the divorce of parents, loss of a loved one or unemployment [5]. Occasionally, trichotillomania is only seen while sleeping, a condition known as sleep‐isolated trichotillomania [6].

#### **1.4. Diagnosis**

#### *1.4.1. Clinical features*

Patients may have other problems with self‐mutilation such as nail‐biting or dermatitis artefacta. Approximately one‐third of the patients chew or swallow the hair they pull out, which is called trichophagia and some of them develop trichobezoars, which is the accumu‐ lation of the patients' own hair in the intestines. These trichobezoars may result in a "tail" that lies along the duodenum, a phenomenon which is called *Rapunzel Syndrome*. Around 1% of trichobezoar patients may need surgical intervention [4].

Symptom severity can be measured by using different validated instruments including Massachusetts General Hospital Hair Pulling Scale that has seven parameters, rating symptom severity from 0 to 4 and assessing various aspects of plucking during the past seven days: actual pulling, urge to pull, associated distress and perceived control, The Yale‐Brown Obsessive‐Compulsive Scale, The Psychiatric Institute Trichotillomania Scale, The Trichotillo‐ mania Scale for Children, The Milwaukee Inventory for Styles of Trichotillomania‐Child Version [7]. The MGHHS includes seven parameters, rating symptom severity from 0 to 4 and assesses several aspects of hair pulling during the previous 7 days: urge to pull, actual pulling, perceived control and associated distress. The MGHHS and its Dutch adaptation have been reported to provide good psychometric properties [7].

Three subtypes of hair pulling have been described (**Table 2**).

• Early onset: occurring in young children, mostly under the age of 8; usually does not need any treatment.

• Automatic: occurring when the individual is busy with other activities, such as reading. This type affects 75% of patients.

• Focused: occurs with the patient's attention and is associated with strong impulses to pull hair.

**Table 2.** Subtypes of trichotillomania.

in the literature in the works of Hippocrates. It is even found in plays by William Shakespeare such as Romeo and Juliet and The Life and Death of King John. The name is derived from the

Trichotillomania is classified under the obsessive‐compulsive and related disorders along with hoarding disorder, skin‐picking disorder (excoriation) and body dysmorphic disorder in the *Diagnostic and Statistical Manual of Mental Disorders* Fifth Edition (DSM‐5; American Psychiatric Association, May 2013) [2] (**Table 1**). It is also grouped under obsessive‐compulsive disorders in the eleventh revision of the World Health Organization's International Classification of

• The hair pulling causes clinically significant distress or impairment in social, occupational, or other important areas

Trichotillomania occurs more frequently in females. The lifetime prevalence is 3.4% for women and 1.5% for men [4]. The typical age of onset is between 5 and 12 years or early childhood to

The onset of trichotillomania often occurs after a stressful event such as the divorce of parents, loss of a loved one or unemployment [5]. Occasionally, trichotillomania is only seen while

Patients may have other problems with self‐mutilation such as nail‐biting or dermatitis artefacta. Approximately one‐third of the patients chew or swallow the hair they pull out, which is called trichophagia and some of them develop trichobezoars, which is the accumu‐ lation of the patients' own hair in the intestines. These trichobezoars may result in a "tail" that lies along the duodenum, a phenomenon which is called *Rapunzel Syndrome*. Around 1% of

Symptom severity can be measured by using different validated instruments including Massachusetts General Hospital Hair Pulling Scale that has seven parameters, rating symptom

combination of Greek *thrix* for hair; *tillein*, plucking; and *mania*, madness.

Diseases and Related Health Problems (ICD‐11) [3].

• The hair pulling or hair loss is not attributable to another medical condition

• The hair pulling is not better explained by the symptoms of another mental disorder

sleeping, a condition known as sleep‐isolated trichotillomania [6].

trichobezoar patients may need surgical intervention [4].

• Recurrent pulling out of one's hair, resulting in hair loss • Repeated attempts to decrease or stop the pulling out of hair

**Table 1.** Diagnostic criteria (DSM‐5) [2].

adolescence, but it may occur in any age.

**1.2. Epidemiology**

**1.3. Pathogenesis**

**1.4. Diagnosis**

*1.4.1. Clinical features*

of functioning

224 Hair and Scalp Disorders

Any part of the hair may be affected, but the targeted hair is mostly on the scalp (75%). The eyelashes (53%), eyebrows (42%), pubic region (17%) and beard (10%) may also be involved, but sometimes there is more than one location (17%) [8, 9]. The most affected scalp areas are the frontoparietal region and vertex, while the least affected region is the occiput. The lower eyelid is usually not involved; this is helpful to distinguish trichotillomania from alopecia areata [10].

Alopecic plaques are usually located on the contralateral side of the dominant hand. There may be more than one plaque. These plaques of hair loss most often have irregular shapes and contain many broken hairs of varying lengths. The margins have normal and long hair.

The plucked hairs have fiber fractures and feel rough on examination. There are usually no signs of inflammation in the plaques, but there may also be signs of excoriation, lichenification and post‐inflammatory hyperpigmentation in some cases.

#### *1.4.2. Dermoscopy*

Trichoscopy may also be utilized for differential diagnosis. The trichotillomania plaque includes broken and irregular coiled hair and hair density is decreased. Black dots, follicular hemorrhages and V‐sign may be seen [11]. Trichoscopy is very useful in the differential diagnosis of trichotillomania from alopecia areata where exclamation mark hairs, yellow dots and proximal tapering hairs could be seen, in contrast with trichotillomania where trichopti‐ losis, pointed hairs, flame hairs, V‐sign, hook hairs, hair powder, follicular microhemorrhage and tulip hairs are more characteristic. Follicular microhemorrhage meaning a red dot that corresponds to a follicular ostium stuffed with blood clot, may support local trauma, a clue for trichotillomania [12].

#### *1.4.3. Histopathology*

The diagnosis of trichotillomania is usually made by clinical examination and patient history. However, occasionally especially in pediatric patient group both child and parents may deny the possibility of pulling or plucking as a cause of hair loss and especially in localized involved patients the diagnosis may be difficult. Histopathological examination may be necessary in these patients. Follicules of normal size, increased catagen and telogen hairs (up to 75%) which is a result of mechanical trauma to the hair frequently propelling anagen follicules into the catagen phase, pigmentary defects and casts, evidence of traumatized hair bulbs and tricho‐ malacia (a complete but distorted, fully developed terminal hair in its bulb) are the most common findings in histological examination of trichotillomania [13, 14]. Catagen hairs may be present in areas that have recently been injured and telogen hairs may present after a few weeks from pulling. Some hair follicles in anagen phase may be present, but they are usually seen empty because of hair shaft avulsion. If the hair matrix and suprabulbar epithelium are injured, but not severely disrupted, the follicle may remain in the anagen phase which may produce a hair shaft. Follicles can show distortion of the bulbar epithelium and sometimes conspicuous hemorrhage.

Hair shaft avulsion may deposit melanin pigment in the hair papilla and peribulbar connective tissue [13, 14]. Frequently, chunks of pigmented hair matrix or cortex cells are torn from their moorings during the plucking process and come to rest in superficial portions of the follicles. These cells then shrink to form a dark black homogeneous clump called a pigment cast. Pigment casts which are very characteristic for trichotillomania simply occur as the by‐product of fragmented, ectopic matrix or cortical epithelium and usually seen in the isthmus or infundibulum. Trichomalacia, which defines shaft abnormalities such as distorted in shape, smaller than normal and incompletely cornified, is very characteristic for trichotillomania.

These injuries of pulling or plucking to the bulbar portions of follicles do not induce inflam‐ mation but may cause follicular microhemorrhage within the lower portion of the follicle. A few eosinophils may be rarely seen around the lower portion of the traumatized follicule. Also miniaturization of follicules is usually not seen in trichotillomania and absence of inflamma‐ tory infiltrate and loss of miniaturization are usually serve to differentiate it from alopecia areata [14].

#### **1.5. Differential diagnosis**

Tinea capitis, alopecia areata, loose anagen hair, monilethrix, lichen planopilaris and secon‐ dary syphilis need to be considered in the differential diagnosis of trichotillomania. Tricho‐ scopy, medical history and scalp biopsy can be used to distinguish trichotillomania from other diseases. Catagen and telogen hair numbers are found to be increased and usually there are no signs of inflammation unless there is an infection in histologic examination. The number of catagen hairs exceeds telogen hairs in chronic lesions. Perifollicular hemorrhage may be found at the circumference of the hair bumbs [1].

Potassium hydroxide examination, fungal culture and Wood's lamp examination may be performed to exclude tinea capitis. The hair is weak and may easily be pulled out in tinea capitis.

Alopecia areata plaques are oval and well‐demarked. A hair pull test may be helpful as a diagnostic test for alopecia areata. Telogenic hairs may be pulled out easily in alopecia areata, which indicates the activity of the disease in contrast with trichotillomania. Shaving the involved area and waiting for the regrowth may also be useful for the diagnosis of trichotil‐ lomania. Alternatively, a small part of the hair is clipped near the scalp with scissors and the hairs in trichotillomania display uniform hair regrowth.

Trichoscopy may also be utilized for differential diagnosis. Exclamation mark hairs and yellow dots may be seen and white hairs are usually not involved in alopecia areata [11]. The tricho‐ tillomania plaque has broken and irregular coiled hair and the hair density is decreased. Black dots, follicular hemorrhages, v‐sign may be seen [12].

#### **1.6. Treatment**

*1.4.3. Histopathology*

226 Hair and Scalp Disorders

conspicuous hemorrhage.

areata [14].

**1.5. Differential diagnosis**

at the circumference of the hair bumbs [1].

The diagnosis of trichotillomania is usually made by clinical examination and patient history. However, occasionally especially in pediatric patient group both child and parents may deny the possibility of pulling or plucking as a cause of hair loss and especially in localized involved patients the diagnosis may be difficult. Histopathological examination may be necessary in these patients. Follicules of normal size, increased catagen and telogen hairs (up to 75%) which is a result of mechanical trauma to the hair frequently propelling anagen follicules into the catagen phase, pigmentary defects and casts, evidence of traumatized hair bulbs and tricho‐ malacia (a complete but distorted, fully developed terminal hair in its bulb) are the most common findings in histological examination of trichotillomania [13, 14]. Catagen hairs may be present in areas that have recently been injured and telogen hairs may present after a few weeks from pulling. Some hair follicles in anagen phase may be present, but they are usually seen empty because of hair shaft avulsion. If the hair matrix and suprabulbar epithelium are injured, but not severely disrupted, the follicle may remain in the anagen phase which may produce a hair shaft. Follicles can show distortion of the bulbar epithelium and sometimes

Hair shaft avulsion may deposit melanin pigment in the hair papilla and peribulbar connective tissue [13, 14]. Frequently, chunks of pigmented hair matrix or cortex cells are torn from their moorings during the plucking process and come to rest in superficial portions of the follicles. These cells then shrink to form a dark black homogeneous clump called a pigment cast. Pigment casts which are very characteristic for trichotillomania simply occur as the by‐product of fragmented, ectopic matrix or cortical epithelium and usually seen in the isthmus or infundibulum. Trichomalacia, which defines shaft abnormalities such as distorted in shape, smaller than normal and incompletely cornified, is very characteristic for trichotillomania.

These injuries of pulling or plucking to the bulbar portions of follicles do not induce inflam‐ mation but may cause follicular microhemorrhage within the lower portion of the follicle. A few eosinophils may be rarely seen around the lower portion of the traumatized follicule. Also miniaturization of follicules is usually not seen in trichotillomania and absence of inflamma‐ tory infiltrate and loss of miniaturization are usually serve to differentiate it from alopecia

Tinea capitis, alopecia areata, loose anagen hair, monilethrix, lichen planopilaris and secon‐ dary syphilis need to be considered in the differential diagnosis of trichotillomania. Tricho‐ scopy, medical history and scalp biopsy can be used to distinguish trichotillomania from other diseases. Catagen and telogen hair numbers are found to be increased and usually there are no signs of inflammation unless there is an infection in histologic examination. The number of catagen hairs exceeds telogen hairs in chronic lesions. Perifollicular hemorrhage may be found As mentioned above, trichotillomania is a psychiatric disorder with dermatological findings which is characterized by compulsive avulsion of hair shafts leading to thin, ragged, broken hairs on the affected region clinically [15]. The inability to control self‐pulling of hair resulting in hair loss may progress into alopecia in long time.

Treatment procedure must be multidisciplinary including both dermatological but mainly psychiatric approach to increase the effectiveness of the therapy and prevent relapse.

Cognitive and behavioral therapies (CBT), antipsychotic agents, selective serotonin reuptake inhibitors (SSRI), tricyclic antidepressants are the main options of treatment [7, 16–18]. The most appropiate therapeutic approach must be chosen according to the patient's age, medical status and mental status.

Cognitive and behavioral therapies are the first steps of treatment and must be considered together with pharmacotherapeutics in treatment [16, 19]. In a randomized controlled trial with 7–8‐year‐olds, cognitive‐behavioral therapies alone were found to decrease the symptoms in 75% of the participants [16].

The results about efficacy of SSRIs are conflicting. They were reported to be the safest and well‐ established medication choice. However, the clinical results show that medication, which is usually an SSRI, in addition to CBT, is more successful, if CBT alone fails.

In a meta‐analysis which was reported in 2007, in which the efficacies of pharmacologic and behavioral treatments were evaluated in treatment of trichotillomania, it was found that SSRIs were not more effective than placebo. In two trials clomipramine was found to be more effective compared with placebo. In three trials, it was shown that there is a beneficial effect of habit reversal therapy compared with no intervention [20].

In another systematic review which was published in 2013, similar results were found. In two included trials, fluoxetine was not more effective than placebo in reducing the mean severity rating of hair pulling. Clomipramine was found to be more effective than placebo, although 3 of 10 participants receiving clomipramine dropped out because of drug‐related adverse effects [21].

At last, in a recent meta‐analysis of 11 randomized trials, the efficacy of behavioral therapy and SSRI for the treatment of trichotillomania; the outcome measure was the standardized mean difference of change in hair pulling [22]. This publication demonstrated a large effect for behavioral therapy and only a moderate effect for SSRI. A greater treatment effect was reported for clomipramine in two included trials [22].

The side effects and limited efficacy of pharmacological treatment, especially in pediatric population and difficulty in long‐term maintenance of behavioral therapies require alternative options of treatment. The glutamatergic system dysregulation is involved in obsessive‐ compulsive disorders etiology and it has been reported that N‐acetyl‐cysteine (NAC) might have a therapeutic effect on these entities by acting on the glutamatergic system and reducing oxidative stress [23]. It was reported as a safe and effective treatment option given 1200 mg/d per os. The efficacy of the glutamate modulator NAC was evaluated in a small randomized trial including 50 adults with trichotillomania [24]. N‐acetylcysteine was more effective than placebo in reducing hair‐pulling symptoms as measured by the Massachusetts General Hospital Hair Pulling Scale. A subsequent trial in children and adolescents did not find any beneficial effect of NAC compared with placebo [24, 25].
