CLINICAL PRACTICE

Trichotillomania and Trichobezoar: A Clinical Practice Insight With Report of Illustrative Case

Joel R. Carr, DO, MPH Ellen H. Sholevar, MD David A. Baron, DO, MSEd

Untreated trichophagia secondary to trichotillomania is a for TTM, even though she had a long history of both disfiguring potentially life-threatening condition. Taking a thorough hair pulling and trichophagia (ingesting the pulled out hair). family and social history, most notably with the aid of a In one study of adults with TTM, 17% did not describe tension genogram or family tree, can aid in including this disorder before or relief after pulling out their hair2 and therefore did in the differential diagnosis. This case presentation describes not meet DSM-IV-TR criteria for the diagnosis. Thus, the cur- a unique occurrence of untreated trichotillomania in a female rent diagnostic criteria may exclude patients with clinically adolescent that led to formation of a trichobezoar requiring significant symptoms of TTM. emergent surgical intervention and follow-up psychiatric treatment. This case highlights osteopathic medicine’s fun- Epidemiology and Presentation damental concept of treating the whole person rather than Although TTM is a relatively rare condition with estimates just symptoms by considering factors such as genetic influ- ranging from less than 1% to as high as 4% of the US popula- ences in understanding disease. tion, the number of patients with TTM seen in physicians’ J Am Osteopath Assoc. 2006;106:647–652 offices is considerable. If even 1% of the US population has TTM, there would be 25 million people with TTM in the United he American Psychiatric Association’s Diagnostic and States.3 TStatistical Manual of Mental Disorders, Fourth Edition, Text The clinical presentation of TTM may be confusing. Revision (DSM-IV-TR)1 includes trichotillomania (TTM) in Patients with TTM are markedly embarrassed and ashamed, the category of a diverse group of impulse-control disorders so they may deny their hair-pulling behavior. The patient that are not classified elsewhere. The essential features of described in this report was rejected from participation in a this disorder include recurrent pulling out of one’s hair research study of TTM because she denied such behavior resulting in noticeable hair loss, increasing sense of tension when she was screened in a telephone interview. Before diag- immediately preceding or when resisting hair pulling, and nosing TTM, physicians must first rule out hair loss that is pleasure or relief when pulling out the hair. In addition, the related to dermatologic conditions or other systemic disor- symptoms of TTM cannot be better accounted for by another ders. They must also exclude it as an act of another person’s psychiatric or medical disorder (eg, schizophrenia, dermatitis) abuse of a youth.4 Hair pulling also has been reported in asso- and must also cause clinically significant distress or impaired ciation with cocaine use.5 social or occupational functioning.1 The site from which hair Trichotillomania may be an unconscious or intentional is most frequently pulled is the scalp, but hair may be pulled action.6 In 5% to 18% of patients, TTM may include trichopha- from eyelashes, eyebrows, the pubic region, or other parts of gia, which in turn leads to the potentially serious complica- the body.2 tion of a trichobezoar (hair ball).7 It is estimated that more than Many patients meet all the criteria for TTM except tension a third of those who ingest hair may form a trichobezoar.7 before or relief after pulling out hair. The patient described here Trichobezoars are more common in children and adoles- had neither criterion and did not meet DSM-IV-TR criteria cents.8,9 When first seen by a physician, a patient with a trich- obezoar may have gastrointestinal obstruction with nausea and vomiting, gut perforation, acute pancreatic necrosis, obstructive jaundice, hypochromic anemia, vitamin B12 defi- From the Department of Psychiatry at Drexel University School of Medicine ciency, weight loss, an abdominal mass, or other serious prob- (Dr Carr), and the Department of Psychiatry at Temple University School of lems. Emergent surgical intervention may be required.8 Medicine (Drs Sholevar and Baron) in Philadelphia, Pa. The diagnosis of a trichobezoar is made through careful Address correspondence to David A. Baron, DO, MSEd, Chairman, Depart- ment of Psychiatry, Temple University, Temple Episcopal Campus, 3rd Floor, history taking, a thorough physical examination, and radiologic 100 E Lehigh Ave, Philadelphia, PA 19125-1012. evidence. Physicians should be aware that a trichobezoar may E-mail: [email protected] be present even in patients who show no signs of hair loss.10 Submitted January 8, 2005; revision received October 20, 2005; accepted It is essential that primary care physicians, emergency November 7, 2005. department physicians, and surgeons consider a trichobezoar

Carr et al • Clinical Practice JAOA • Vol 106 • No 11 • November 2006 • 647 CLINICAL PRACTICE when examining patients with symptoms of gastrointestinal Figure. Genogram of family of patient with trichotillomania. Squares obstruction or any of the possible emergent presentations of this indicate male family members; circles, female family members. Cen- disorder. Early intervention may prevent potentially fatal out- tered number designations in color indicate age of family member in comes.8 years. Smaller numbers in black refer to relevant aspects of family members’ social and psychiatric history not otherwise noted in the key. ᮣ Etiology NA indicates date unknown. Explanations of the cause of TTM parallel some of the major theoretic orientations in the study of human behavior. Psy- choanalytic explanations for hair pulling center on the behavior In habit-reversal therapy, patients learn to be aware of as being related to unconscious conflicts. The teenager the times, cues, and situations in which they pull their hair. described in this report pulled out her hair in the evening They practice movements such as those in knitting, crochet, and when she was alone in her bedroom and when she was tense needlepoint that redirect their urges to pull their hair. Thus they and worried. She had been through a difficult separation of her learn to “substitute a different and more adaptive behavior”14 parents when she entered adolescence, though her TTM symp- and receive social approval for efforts to interrupt the hair toms began when she was 3 years old. She was anxious, fearful, pulling.13 Other approaches such as cognitive-behavioral treat- and ashamed about her hair pulling, feelings that intensified ment, negative practice, and variations of these interventions the urges to pull her hair. have been tried.3 Biologic explanations emphasize TTM as a familial dis- One of the authors (J.R.C.) used elements of habit-reversal order or part of the spectrum of an obsessive-compulsive dis- therapy to help the patient become more aware of the internal order. The evidence is not robust, however, for TTM as an and external cues that triggered her hair pulling. He instructed obsessive-compulsive–related disorder, and convincing evi- her to wear a rubber band on her wrist and to pull it when she dence for a hereditary basis is lacking. felt the impulse to pull her hair. He also worked with the Positron emission tomographic scans of patients with patient and her mother to counteract the criticism and nega- TTM show increased metabolic activity in the cerebellar and tivity that had developed around the hair pulling. parietal cortex,11 while magnetic resonance imaging studies The SSRIs are the pharmacologic agents used most fre- have found decreased volume in the left side of the putamen.12 quently in the treatment of patients with TTM. Other psy- These studies, though fascinating in that they document bio- chotropic agents that have been used for treating patients with logic differences between patients with TTM and healthy con- TTM include the tricyclic antidepressants, the antipsychotic trol subjects, are not useful for diagnosing TTM, nor do they medications, and mood-stabilizing (anticonvulsant) medica- assist in developing an effective treatment strategy. tions. Clomipramine hydrochloride and fluoxetine hydrochlo- Imaging studies are not clinically indicated for TTM, and ride have been used successfully.15 they were not done in our patient. In addition to cognitive-behavioral therapy, the patient The patient described here neither met the diagnostic received SSRI therapy because of her long history of hair DSM-IV-TR criteria for obsessive-compulsive disorder or tics, pulling with its serious consequences and her recurring hair- nor did she have a reported family history of these disorders. pulling impulses after surgical removal of a trichobezoar. This The family history and genogram (Figure) revealed that she had combination treatment was effective initially. a cousin with TTM and other family members who had an anx- The use of hypnosis for the treatment of patients with iety disorder, alcohol dependence, or attention deficit hyper- TTM has been reported,16 but hypnosis was not used in our activity disorder (ADHD) and multiple family members who patient. Books, information, and support groups for patients had had a “nervous breakdown.” with TTM and their families are available through the Tricho- Behavioral theories emphasize modeling and conditioning tillomania Learning Center, Inc, in Santa Cruz, Calif (see as factors that establish and maintain hair pulling as a habit.3 http//www.trich.org). Our patient was referred to the latter Some behavioral modes of therapy, including habit-reversal resource, and she and her family found it helpful. therapy, are derived from this theoretic explanation.13 Ele- ments of habit-reversal therapy13 were used to treat our patient. Illustrative Case Presentation A 16-year-old Latino girl in good general health was seen in the Treatment emergency department because of a several-day history of No clear evidence-based practice guidelines for the treatment intractable nausea, vomiting, and diarrhea that she attributed of patients with TTM are available, and more research is to a Fourth of July picnic. She was in mild distress and afebrile needed. Behavioral techniques and selective serotonin reuptake with stable vital signs; she did not have a toxic appearance. inhibitors (SSRIs) are the most commonly used treatment After receiving intravenous hydration, she returned home. modalities, having the most evidence for efficacy. The behav- Two days later, the patient returned to the emergency ioral approaches, including habit-reversal therapy, have been department with the same complaints. At physical examina- shown to be effective.14 tion, the abdomen was soft and diffusely tender, without

648 • JAOA • Vol 106 • No 11 • November 2006 Carr et al • Clinical Practice CLINICAL PRACTICE 9 8 7 6 5 4 3 2 1 loves him greatly Maternal grandfather lives in Philadelphia, Pa, near identified patient, who ment for goiter; trichotillomania recently diagnosed in daughter Aunt has systemic lupus erythematosus, hyperthyroidism with history of treat- sion, coronary artery disease with bypass grafting, stroke History of “nervous breakdowns,” hypercholesterolemia, diabetes, hyperten- according to former spouse (patient’s mother) Father lives in state adjacent to Pennsylvania since 1998; has anxiety disorder, school graduation United States, but returned from Philadelphia, Pa, to Puerto Rico after high Since patient was 11 years old, she has lived with her mother in the continental hyperactivity disorder Brother 2 years younger than the patient, had diagnosed attention deficit Maternal aunt, close to the patient; now living with maternal grandfather Paternal grandfather whom patient especially loved and misses Identified patient NA

6 5 38 39 16 1 1998 4

5 14 5 2 3

37 35 16 9 Divorced (date shown when available) Unmarried Alcohol dependent, male Deceased, male Married, related Identified patient, 16-year-old 3 7 8 30 22 NA 21

Carr et al • Clinical Practice JAOA • Vol 106 • No 11 • November 2006 • 649 CLINICAL PRACTICE rebound or peritoneal signs but with a vague epigastric mass. a relative, a registered nurse, warned both the patient and her Rectal examination revealed a positive screening fecal occult mother that continued hair pulling and eating the hair could blood test and loose stool in the vault, but no masses. Labo- result in serious medical and surgical consequences. This infor- ratory tests revealed a white blood cell count of 9900/␮L with mation increased familial conflict and concern over hair pulling, no left shift, electrolyte derangement consistent with metabolic but it did not decrease the patient’s behavior. The increased alkalosis, and serum amylase and lipase levels within the ref- anxiety of the patient and her family escalated the impulses to erence range. pull hair. Findings of the ultrasound examination of the right upper Both the patient and her mother reported that the patient quadrant were negative for cholecystitis. Abdominal flat plates was “neat and orderly” in her habits and that she would showed no free air, no air-fluid levels, and an apparent unusual become upset if her room was not “in order.” At the time of gas pattern in the upper right quadrant with a rim of air inside. hospital admission, the patient was an honor student in a On placement of a nasogastric tube (NGT) and suction, a few public high school serving a disadvantaged population. strands of hair were found. After placement of the NGT, a Although her family was not affluent and she lived in a pre- large mass was palpated in the epigastric region. dominantly minority inner-city neighborhood, the patient Computed tomography scans revealed a grossly dis- hoped to attend college after high school graduation. tended stomach containing a large quantity of undigested Family history revealed that the 39-year-old mother and material and some edematous loops of bowel with a small the 38-year-old father separated when the patient was 11 years amount of free fluid in the pelvis. A trichobezoar was diag- old (Figure). Family psychiatric history includes ADHD in a nosed, and the patient was taken to the operating room, where brother who was 2 years younger than the patient. The paternal she underwent exploratory laparotomy. grandmother and the paternal great-grandmother were Her stomach was distended and pale, and the mass within reported to have a history of “nervous breakdowns that it extended from the antrum to the gastroesophageal junc- required many medications,” notably alprazolam. Further- tion. After incision, a trichobezoar weighing 1350 g (3 pounds) more, the patient’s mother reported that her ex-husband’s was removed. family had a clinically significant psychiatric history of anxiety Postoperatively, the child and adolescent psychiatric ser- disorders. The paternal grandfather, now deceased, had a vice evaluated the patient in consultation. The patient was strong alcohol dependence disorder. The patient especially anxious and ashamed. She indicated that she was aware that loved and missed him. swallowing hair could lead to medical problems and reported The patient’s mother was the oldest of six children. A that she had attempted to stop the hair pulling on her own, but maternal aunt of the patient had a 3-year-old daughter who had she was unable to do so. She admitted, “I was scared when I recently begun to pull her hair. This child had no contact with knew it could get worse if nothing was done.” She had seen the patient, as all the patient’s maternal family was reared in a television documentary film “a long time ago” on the topic Puerto Rico. The patient’s mother reported feeling stressed of TTM and immediately worried that her hair pulling was the as a single parent rearing two troubled teens. cause of her initial complaints. She spontaneously said, “I’ll When the patient was seen in the child and adolescent psy- never do that again.” She said that she pulled her scalp hair chiatry department, she appeared as an attractive Latino only when alone, especially at bedtime. She did not pull her teenager who looked her stated age. She had facial acne that hair at school or when she was in the presence of others. She was not severe and extremely short hair. She appeared to be ate approximately half of the hair that she pulled out. in mild discomfort from the NGT. The patient’s Spanish-speaking mother, interviewed via The patient reported that she had recently cut her hair an interpreter, reported an uncomplicated developmental his- short in an attempt to reduce her hair pulling behavior. No tory. At 3 years of age, with no known clinically significant psy- areas of thinned hair were noted. Her mood was depressed and chosocial stressors, the patient began twisting two or three her affect blunted. The patient denied having thoughts of sui- strands of hair around a finger and swallowing them. Bald cide or hallucinations and delusions. She was oriented and spots appeared on the patient’s head shortly thereafter. It was appeared to be of above-average intelligence. noted that the patient sucked a finger, a habit that continued The patient’s postoperative recovery was uncomplicated. to the present. The mother reported that when the patient She was discharged with recommendations to contact a TTM was 11 years old, she took her to a psychologist in Puerto research program. On follow-up, it was discovered that the Rico. The psychologist referred the child back to her pediatri- patient did not meet entry criteria for the study for the previ- cian after one visit and without any recommendation for treat- ously noted reasons and because she denied current hair ment. The pediatrician recommended that the patient try pulling. One of the authors (J.R.C.) offered the patient outpa- gloving with nylon stockings and putting on a swim cap at bed- tient psychiatric treatment, which she and her family accepted. time. After a few failed attempts to comply, the patient aban- Outpatient therapy (with J.R.C.) began on a weekly basis. doned this intervention. The patient reported having active social and romantic rela- The patient and her family had heightened concerns when tionships as well as maintaining part-time employment tutoring

650 • JAOA • Vol 106 • No 11 • November 2006 Carr et al • Clinical Practice CLINICAL PRACTICE youths. She reported more detail of current family stressors, Comments including a feeling of responsibility for managing two younger This interesting case highlights a number of important clinical cousins who had recently moved into her family’s home. She issues. reported having anxiety about school achievement and concern Our young patient had a long history of physical and about helping with her younger brother, who was under- emotional problems that though accurately diagnosed early in going outpatient treatment for ADHD. She also reported that the course of her illness, resulted in her needing emergent her mother had difficulty coping with the overwhelming surgery. Thus, making the correct diagnosis is important but household and that she and her mother had conflicts over the not sufficient to ensure a positive outcome. Accurate diag- hair pulling. nosis is only the first step in successfully treating patients. After three initial sessions, the patient reported a gradual Given this adolescent’s strong genetic loading for anxiety dis- return to thumb sucking and hair pulling. She was distressed orders and her well-documented hair-pulling behavior, trich- about this regression and reported that she had attempted to obezoar formation was predictable. interrupt the hair pulling on her own with competitive activ- Treatment needed to include more than the use of ities such as drawing, manipulating a braided wire, and mechanical constraints to the hair-pulling behavior. She needed learning to crochet. These activities failed to abate the urge to a comprehensive biopsychosocial approach to the treatment pull her hair. strategy. Therapy included educating the patient about TTM. She This case clearly demonstrates the importance of under- was directed to the Trichotillomania Learning Center. Ele- standing patients in the context of their life situation (and life ments of habit-reversal therapy were used, and the patient stressors). Not attending to our patient’s underlying stress was helped to identify in advance triggering external situations and anxiety early in the course of her illness directly resulted and internal feelings that led to the urges to pull her hair. She in her near-fatal surgical emergency. The patient showed learned alternate activities that could substitute for hair pulling. improved control of her urges to pull her hair in response to Her mother was included in the therapy and learned about the psychiatric intervention after her surgery, but she was lost TTM, and she began to participate in helping the patient to follow-up and ultimately regressed. develop strategies to handle the urges to pull hair, as well as It is necessary but not sufficient to merely diagnose a learning to praise her daughter for her efforts to handle the maladaptive behavior and instruct patients in how to overcome urges appropriately. Thus, the mother-daughter relationship the behavior. Whether it is overeating by obese patients or improved. hair pulling as in our patient, achieving a positive therapeutic The patient’s facial acne responded to the topical treatment outcome requires that physicians identify issues that con- provided by one of the authors (J.R.C.). This favorable response tribute to the maladaptive behaviors and establish effective led to improvement in the patient’s self-esteem. She shared with treatment interventions such as psychotherapy and medica- her therapist her feelings about her parents’ divorce and the tions. In addition, long-term follow-up is critical to reinforcing subsequent infrequent contacts she had with her father. long-term compliance with treatment. As was seen in our As the patient attended more therapy sessions, she devel- patient, without follow-up, the likelihood of exacerbation of oped a sense of understanding and mastery of TTM, and she symptoms is high. and her mother spoke on the telephone to the patient’s aunt in Puerto Rico, educating her about the TTM that the patient’s 3- Acknowledgment year-old cousin had developed. The patient and her mother The authors thank Karriem Salaam, MD, of the Division of Child/Adolescent became the “TTM experts” in the family. Psychiatry at Jefferson Medical College in Philadelphia, Pa, for supporting Sertraline hydrochloride, starting at 50 mg/d and titrating information. up to 100 mg/d, was given to the patient to provide addi- tional help in reducing her hair-pulling urges. After several References months of weekly psychotherapy and pharmacotherapy, the 1. American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders Fourth Edition Text Revision (DSM-IV-TR). Washington, DC: patient reported no hair-pulling events, even when faced with American Psychiatric Association; 2000. academic or family stressors. 2. Christenson GA, Mackenzie TB, Mitchell JE. Characteristics of 60 adult The sertraline therapy and weekly psychotherapy ses- chronic hair pullers. Am J Psychiatry. 1991;148:365–370. sions were continued until after the patient graduated from 3. Diefenbach GJ, Reitman D, Williamson DA. Trichotillomania: a challenge high school. Then, the dose of sertraline was tapered. The to research and practice. Clin Psychol Rev. 2000;20:289–309. patient was lost to office follow-up when she left the United 4. Saraswat A. Child abuse and trichotillomania. BMJ. 2005;330:83–84. Avail- States to attend college in Puerto Rico. A brief follow-up via able at: http://www.bmj.com/cgi/content/full/330/7482/83. Accessed August 21, 2006. telephone revealed that the patient could not find a therapist (continued on the next page) in Puerto Rico and she was not receiving sertraline. Thus, her urges to pull her hair recurred.

Carr et al • Clinical Practice JAOA • Vol 106 • No 11 • November 2006 • 651 OSTEOPATHIC CLINICAL PRACTICE MANIPULATIVE MEDICINE EDUCATION OPPORTUNITY 5. George S, Moselhy H. Cocaine-induced trichotillomania. Addiction (Abingdon, England). 2005;100:255–256. Mercy Health Partners is currently seeking a board-cer- 6. Christenson GA, Crow SJ. The characterization and treatment of trichotil- tified Osteopathic Manipulative Medicine Practitioner lomania. J Clin Psychiatry. 1996;57(suppl 8):42–47. to join the Mercy Health Partners osteopathic medical 7. Bouwer C, Stein DJ. Trichobezoars in trichotillomania: case report and education program. The program is dually accredited literature overview. Psychosom Med. 1998;60:658–660. by the AOA and the ACGME. The selected candidate 8. Phillips MR, Zaheer S, Drugas GT. Gastric trichobezoar: case report and lit- would serve as a faculty resource for all of the osteo- erature review. Mayo Clin Proc. 1998;73:653–656. pathic programs and support appropriate clinics. Fifty 9. Caprai S, Massimetti M, Ughi C, Quinti S, Maggiore G. Clinical quiz: trich- percent of the candidate’s time would be devoted to obezoar, a rare and unique form of gastrointestinal foreign body. J Pediatr OMM and fifty percent would be devoted to providing Gastroenterol Nutr. 1998;26:436,453. clinical care. Primary 10. Konen O, Rathaus V, Shapiro M. Unsuspected trichobezoar in a child with hospital affiliations include short hair. AJR Am J Roentgenology. 2001;176:258–259. Available at: http://www.ajronline.org/cgi/content/full/176/1/258. Accessed November 13, St. Vincent Mercy Med- 2006. ical Center, a 568-bed ter- tiary care center and St. 11. Swedo SE, Rapoport JL, Leonard HL, Schapiro MB, Rapoport SI, Grady CL. Regional cerebral glucose metabolism of women with trichotillomania. Arch Charles Mercy Hospital a Gen Psychiatry. 1991;48:828–833. 390-bed community hos- 12. O’Sullivan RL, Rauch SL, Breiter HC, Grachev ID, Baer L, Kennedy DN, et pital. We are seeking a al. Reduced basal ganglia volumes in trichotillomania measured via mor- physician with superior phometric magnetic resonance imaging. Biol Psychiatry. 1997;42:39–45. clinical, and interpersonal 13. Azrin NH, Nunn RG, Frantz SE. Treatment of hairpulling: a comparative skills to join our highly study of habit reversal and negative practice training. J Behav Ther Exp Psy- motivated team. Mercy chiatry. 1980;11:13–20. Health Partners is a rec- 14. Stewart RS, Nejtek VA. An open-label, flexible-dose study of olanza- ognized leader in pro- pine in the treatment of trichotillomania. J Clin Psychiatry. 2003;64:49–52. viding quality, cost-effec- 15. American Psychiatric Association. Consumer & family information. Obses- tive healthcare and is a regional leader in collaboration sive-compulsive disorder. Psychiatric Services. 2001;52:577. Available at: with other community organizations to meet community http://psychservices.psychiatryonline.org/cgi/reprint/52/5/577. Accessed needs. Mercy Health Partners encompasses seven November 28, 2006. acute care hospitals and offers a medical staff of over 16. Iglesias A. Hypnosis as a vehicle for choice and self-agency in the treat- 2,400 members to support the comprehensive range ment of children with trichotillomania. Am J Clin Hypn. 2003;46:129–137. of inpatient and outpatient services. Outstanding schools coupled with affordable housing give northwest Ohio families a great lifestyle. Toledo boasts of having 33 colleges and universities within a 60-mile radius, including a medical college within the metro area. A culturally prosperous city, Toledo is home to a nationally renowned art museum and zoo, the Toledo Symphony, as well as the newly renovated All Join Hands Valentine Theater. The community is home to the The week of October 1–8, 2006, marked the first obser- Toledo Mud Hens, the triple A affiliate of the Detroit vance of National Trichotillomania Awareness Week, Tigers, and recently built a new downtown stadium, an effort to bring attention and awareness to this little Fifth Third Field, which was named as one of the top known disorder of repetitive hair pulling. The Tri- minor league baseball stadiums in the country. Toledo chotillomania Learning Center sponsored this first also offers an ECHL hockey team, NCAA division 1A national US observance, using the “All Join Hands” sports, and an annual LPGA golf tournament. Nature slogan “because when [we] join hands, we’re working and outdoors lovers enjoy the close proximity to Lake together, we’re moving forward—and we’re not Erie. Additionally, Ann Arbor, Chicago and Detroit are pulling.” all within a short driving distance. For more information, see http://www.trich.org. Please send inquires or CV’s to: Jeffrey A. Dempsey, Mercy Health Partners Medical Staff Recruiter 2200 Jefferson Avenue, Toledo, Ohio 43604 phone: 800-837-4664, ext. 3664, email: [email protected], fax: 419-251-2102. 652 • JAOA • Vol 106 • No 11 • November 2006