Psychosomatics 46:362-366, August 2005
© 2005 The Academy of Psychosomatic Medicine
Untreated Trichotillomania and Trichophagia: Surgical Emergency in a Teenage Girl
Karriem Salaam, M.D.,
Joel Carr, D.O.,
Harsh Grewal, M.D.,
Ellen Sholevar, M.D., and
David Baron, D.O.
Received May 27, 2004; revision received Oct. 8, 2004; accepted Oct. 28, 2004. From the Department of Psychiatry and the Department of Surgery, Temple University School of Medicine. Address correspondence and reprint requests to Dr. Baron, Temple University Health Sciences Center, Department of Psychiatry, Jones Hall, 8th Floor, 1316 West Ontario St., Philadelphia, PA 19140.
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ABSTRACT
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Trichophagia secondary to trichotillomania can be a potentially life-threatening condition if left untreated. This report documents the unique case of a 16-year-old girl whose untreated trichotillomania led to a trichobezoar requiring surgical intervention and follow-up psychiatric treatment. Challenges in the identification and treatment of the disorder and its complications are discussed as well as the importance of early and accurate diagnosis.
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INTRODUCTION
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The word "trichotillomania" is of Greek origin, resulting from the combination of the words "trich" (hair), "tillo" (to pull), and "mania" (an abnormal love for a specific object, place, or action) and was first noted in a scientific report of hair-pulling behavior in 1885, although it was not coined as trichotillomania until 1889 by the French physician Hallopeau.1 In DSM-IV-TR, trichotillomania is classified as an impulse control disorder not otherwise specified.2 However, some believe it should be more appropriately classified as an obsessive-compulsive spectrum disorder because trichotillomania and obsessive-compulsive disorder (OCD) share similar patterns of comorbidity and appear to share a familial and/or genetic relationship.3–6 The essential features of trichotillomania outlined in DSM-IV-TR include a recurrent pulling out of one’s hair resulting in noticeable hair loss, an increasing sense of tension immediately preceding or when resisting hair pulling, and pleasure or relief when pulling out the hair. In addition, the symptoms cannot be better accounted for by another psychiatric or medical disorder (e.g., schizophrenia or dermatitis) and must also cause significant distress or impaired social or occupational functioning.2 Estimates of clinically relevant hair pulling in the general population range from 1% to 4%.6
Trichobezoars (hairballs) are complications of trichotillomania (recurrent hair pulling) and subsequent trichophagia or mouthing of the hair.2 Bezoars are foreign bodies in the lumen of the digestive tract that increase in size over time by the accretion of ingested, nonabsorbable food or fibers.7,8 Five types of bezoars have been described: phytobezoar (vegetable fibers), trichobezoar (hair), disopyrobezoar (persimmons), lactobezoar (inspissated milk or formula), and miscellaneous.7,8 Other ingested materials can include blanket or carpet fibers and animal or doll hair.1,9 Trichobezoars have been described as typically extending from the stomach to the small intestine and are noted to resemble a tail. This phenomenon has been named Rapunzel’s syndrome.10
Diagnosing trichobezoars relies on objective radiological and clinical evidence, especially when patients present for medical intervention.11 Signs and symptoms of trichobezoars include abdominal pain, nausea, bilious vomiting, hematemesis, anorexia, early satiety, weakness, weight loss, and abdominal mass.7,10–14 Cases can be complicated by gastrointestinal obstruction, perforation, intussusception, bleeding ulcer, acute pancreatic necrosis, gastric polyposis with malignant degeneration, obstructive jaundice, protein-losing enteropathy, hypochromic anemia, and vitamin B12 deficiency.7,10,12,13 Trichobezoars can lead to gastrointestinal obstruction requiring surgical intervention.12
Trichobezoars are most common in children and adolescents.7,9 Five to 18% of patients with trichotillomania ingest hair; however, the incidence of trichobezoars in trichotillomania is unclear.6 Of those who engage in trichophagy, approximately 37.5% are at risk of forming a trichobezoar.14 The most common presenting symptoms of trichobezoar include a palpable abdominal mass and noticeable hair loss.13 Surgeons, emergency room physicians, and radiologists must consider a trichobezoar in the differential diagnosis of gastrointestinal obstruction, even in patients who show no signs of hair loss.13
Early diagnosis and intervention is of paramount importance and may avoid fatal complications.12 Both pharmacotherapeutic and behavioral interventions have been demonstrated to be effective in treating patients with this disorder.6,14 Selective serotonin reuptake inhibitors, tricyclic antidepressants, antipsychotics, mood stabilizers, and stimulants have been used.15–17 Although most of the published clinical trials are open label and some support the effectiveness of clomimpramine or fluoxetine, randomized controlled studies comparing behavior therapy and pharmacotherapy are lacking.17 One recent randomized controlled study of patients older than 16 years found behavior therapy superior to fluoxetine in reducing the symptoms of trichotillomania and the severity of hair loss.17 The few controlled studies of medication treatment of trichotillomania have been reported only with adult patients.16 Behavioral interventions of various types, including cognitive behavior therapy and habit-reversal therapy, have been reported to be effective. Habit-reversal therapy includes techniques in which the patient monitors the undesirable behaviors and substitutes a desired behavior for the problem behavior. Social approval is given for efforts to eliminate the unwanted behavior.18 For children, supportive family counseling has also been reported to be effective.16
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Case Report
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"Anna," a healthy 16-year-old Hispanic girl, came to the emergency room in mild distress complaining of a several-day history of intractable nausea, vomiting, and diarrhea. She was afebrile, with stable vital signs and did not appear toxic. Her abdomen was soft and diffusely tender, without rebound or peritoneal signs. An epigastric mass was discovered upon abdominal examination. A rectal examination was hemoccult positive with loose stools in the vault and no masses. Laboratory data revealed a WBC count of 9.9 with no left shift, electrolyte derangement consistent with metabolic alkalosis, and normal amylase and lipase. An ultrasound of the right upper quadrant was negative for cholecystitis. Abdominal flat plates showed no free air, no air fluid levels, and what appeared to be an unusual gas pattern in the right upper quadrant with a rim of air inside. Upon placement of a nasogastric tube, a few strands of hair were found in the effluent. A large malleable mass was palpated in the epigastric area. Computerized tomography revealed a grossly distended stomach that contained a large quantity of undigested material and some edematous loops of bowel with a small amount of free fluid in the pelvis. The diagnosis of a trichobezoar was made, and Anna was brought to the operating suite where she underwent an exploratory laparotomy. A grossly distended pale stomach was encountered. The mass within the stomach was found to extend from the antrum to the gastroesophageal junction. A gastrostomy incision was made in the anterior wall of the stomach, and a trichobezoar weighing 1350 g was extracted (Figure 1).
The child/adolescent psychiatric service evaluated Anna in consultation. She was aware that swallowing hair could lead to medical problems and had unsuccessfully attempted to stop on her own. "I was scared when I knew it could get worse if nothing was done." She had seen a television documentary "a long time ago" on the topic of trichotillomania and immediately worried that her hair pulling was the cause of her current complaints. She stated that she pulled the hair only when alone, especially at bedtime. She did not pull her hair in the presence of others or when in school. She ate approximately half of the hair that she pulled.
Anna’s Spanish-speaking mother was interviewed with the use of a translator and reported an uneventful developmental history, with the exception of gastrointestinal reflux at 3 months of age, which cleared spontaneously. At 3 years of age, with no known psychosocial stressors, Anna began twisting two to three strands of hair around her finger and swallowing them. Bald spots on her head were reported shortly thereafter. She also had a habit of sucking her finger, a habit that continued until the present. Anna’s mother reported that she took her to a psychologist in Puerto Rico when she was 11 years old. The child was referred to her pediatrician after one visit, without any recommendation for treatment. The pediatrician recommended that Anna be gloved and that a swimming cap be placed on her head at bedtime. After only a few failed attempts, this intervention was abandoned, and there was no further therapy. Anna’s and her family’s concerns were heightened when a relative, a registered nurse, warned both Anna and her mother that continued hair pulling and eating could result in serious medical and surgical consequences. This information increased familial conflict between Anna and her mother and concern over the hair pulling but did not decrease the behavior. Both Anna and her mother reported her to be "neat and orderly" in her habits; Anna would become upset if her room was not "in order." She was an honor student in a public high school at the time of admission to the hospital and had no history of any type of learning disorder. Her academic performance had always been excellent.
Anna’s family history revealed that her 39-year-old mother and 38-year-old father had separated when she was 11 years old. Her family psychiatric history included attention deficit hyperactivity disorder (ADHD) in a brother 2 years her junior, alcohol dependence in her paternal grandfather, and trichotillomania with trichophagy in a 3-year-old female cousin living in Puerto Rico. This child had no contact with Anna. Her paternal grandmother was reported to have a history of "nervous breakdowns that required many medications," including alprazolam. Furthermore, Anna’s mother reported that her ex-husband’s family had a strong psychiatric history of anxiety disorders.
Anna was an attractive Hispanic teen of the stated age who was in mild discomfort because of the nasogastric tube. She reported that she had recently cut her hair very short in an attempt to decrease her hair pulling. There were no areas of thinned hair or hair loss noted. Her mood was depressed and her affect blunted. She reported no suicidal ideation or auditory or visual hallucinations and delusions. She was oriented and appeared to be of above-average intelligence.
Anna’s postoperative recovery was uneventful. She was discharged with recommendations to contact a trichotillomania research program. Upon follow-up, it was discovered that she had told the research program that she no longer pulled or ate her hair, and thus she did not meet entry criteria for the study. No other follow-up treatment had been arranged. Anna was offered outpatient psychiatric treatment by one of the author’s (J.C.), which she and her family accepted.
Outpatient therapy (with J.C.) began on a weekly basis. Anna reported having satisfactory social and romantic relationships as well as maintaining part-time employment. She elaborated on current family stressors, including a feeling of responsibility for managing two cousins who had recently moved in with her family. She reported anxiety about school achievement and concern about helping with her younger brother, who was engaged in outpatient treatment for ADHD as well as distress about facial acne. She also reported that her mother had difficulty coping with family pressures. She was initially very positive toward the therapist and the therapy. After three initial sessions, Anna reported a gradual return to thumb sucking and hair pulling (without trichophagy). She was distressed about this and reported that she had attempted to interrupt the hair pulling on her own with competitive responses such as drawing, manipulating a braided wire, and learning to crotchet. These activities failed to abate the urge to pull.
Supportive and behavioral interventions, including elements of habit reversal training and patient education were provided by the therapist. The therapist encouraged Anna to set realistic goals and was given support for her efforts. Her mother was given information on how to work with her to support the therapy. Conflict between mother and the teen decreased, and they began to work together to decrease the hair pulling. Anna was given sertraline, 25 mg/day, for 6 weeks. The hair pulling was minimized but not eliminated. The sertraline was increased to 50 mg/day for 8 more weeks, and the therapy continued. Anna reported that she was not pulling her hair for some time but eventually began to pull it again. Skin hygiene and tretinoin cream were added to reduce her anxiety about her facial acne. At a dose of 100 mg/day of sertraline, with the improvement of the facial acne and ongoing therapy, there was a dramatic decrease in the frequency of hair pulling and total cessation of thumb sucking. The hair pulling was reported at a frequency of once or twice a week precipitated by acute family or academic stressors. This response was seen after 8 weeks of treatment and continued for an additional 12 weeks. Anna continues in treatment and will be monitored for improvement with the Massachusetts Hairpulling Scale and clinical observations.19
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Discussion
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Patients with hair loss should be questioned about trichotillomania and trichophagia. Additional clues to considering this diagnosis include a personal or family history of anxiety disorders, especially OCD. This intelligent teenager was aware of the potential consequences of her actions but was unable to control the behavior. Although claiming she would never pull or eat her hair again after undergoing abdominal surgery, only time will tell if psychiatric outpatient treatment will be successful in helping her deal with this behavior. The fact that it had been diagnosed at a young age did not alter the course of the illness leading up to her surgery. It is not clear whether the outpatient psychotherapy or the medication were most important in the improvement in symptoms noted in this case because both were provided.
This fascinating case has implications for a variety of health care providers in addition to consultation/liaison psychiatrists, including psychiatrists, psychologists, primary care physicians, emergency room physicians, pediatricians, surgeons, physician assistants, school nurses, and social workers. The patient had an abdominal obstruction, highlighting the need to include a trichobezoar in the differential diagnosis of patients with symptoms of gastrointestinal obstruction. Despite a longstanding knowledge of her hair-eating behavior by her parents and psychological treatment being previously recommended for the problem, she ultimately required an emergency surgical procedure. It is important to note that an accurate diagnosis is necessary but not always sufficient. The need to liaison with a primary care provider and a mental health professional to ensure necessary follow-up treatment is vital. Further research, especially with children and adolescents, is needed to guide clinicians in the choice of treatment modalities for this serious problem.
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ACKNOWLEDGMENTS
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The authors thank Dilip Ramchandani, M.D., for his editorial assistance.
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REFERENCES
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- Hallopeau F: Alopecia par grottage (trichomania ou trichotillomania). Ann Dermatol Syphil 1889; 10:440
- American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, 4th Ed., Text Revision. Washington, American Psychiatric Association, 2000
- Tynes LL, Winstead DK: Behavioral aspects of trichotillomania. J Louisiana State Med Society 1992; 144:459–463
- Stein DJ, Muller L, Islam MN, Cohen L, DeCaria CM, Hollander E: Compulsive and impulsive symptomatology in trichotillomania. Psychopathology 1995; 28:208–213[Medline]
- King RA, Scahill L, Vitulano LA, Schwab-Stone M, Tercyak KP Jr, Riddle MA: Childhood trichotillomania: clinical phenomenology, comorbidity, and family genetics. J Am Acad Child Adolesc Psychiatry 1995; 34:1451–1459[CrossRef][Medline]
- Bouwer C, Stein DJ: Trichobezoars in trichotillomania: case report and literature overview. Psychosom Med 1998; 60:658–660[Abstract/Free Full Text]
- Phillips MR, Zaheer S, Drugas GT: Gastric trichobezoar: case report and literature review. Mayo Clin Proc 1998; 73:653–656[Medline]
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- Deslypere JP, Praet M, Verdonk G: An unusual case of the trichobezoar: the Rapunzel syndrome. Am J Gastroenterol 1982; 77:467–470[Medline]
- Stone KD, Rush BM, Westphal JR: Trichotillomania resulting in a trichobezoar: a case report. J Louisiana State Med Society 1998; 150:478–481
- Pul N, Pul M: The Rapunzel syndrome (trichobezoar) causing gastric perforation in a child: a case report. Eur J Pediatr 1996; 155:18–19[Medline]
- Konen O, Rathaus V, Shapiro M: Unsuspected trichobezoar in a child with short hair. Am J Roentgenology 2001; 176:258–259[Free Full Text]
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- Lynch K, Feola P, Guenther E: Gastric trichobezoar: an important cause of abdominal pain presenting to the pediatric emergency department. Pediatr Emerg Care 2003; 19:343–347[CrossRef][Medline]
- Tay Y, Levy M, Metry D: Trichotillomania in childhood: case series and review. Pediatrics 2004; 113:e494-e498
- van Minnen A, Hoogduin KA, Keijsers GP, Hellenbrand I, Hendriks GJ: Treatment of trichotillomania with behavioral therapy or fluoxetine: a randomized, waiting-list controlled study. Arch Gen Psychiatry 2003; 60:517–522[Abstract/Free Full Text]
- Azrin N, Nunn R: Habit-reversal: A method of eliminating nervous habits and tics. Behav Res Ther 1973; 11:619–628[CrossRef][Medline]
- Keuthen NJ, O’Sullivan RL, Ricciardi JN, Shera D, Savage CR, Borgmann AS, Jenike MA, Baer L: The Massachusetts General Hospital (MGH) Hairpulling Scale: 1. development and factor analyses. Psychother Psychosom 1995; 64:141–145[Medline]
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ABSTRACT
INTRODUCTION
