Clinical Case Conference

Hair Apparent: Syndrome

Ariel S. Frey, B.A., M.A.T. Initial Psychiatric Consultation The child team (A.S.F. and A.M.) met with Milissa McKee, M.D., M.P.H. Emily and her mother the morning before surgery. By her own and her mother’s account, she was happy and Robert A. King, M.D. well adjusted. She had many friends and was enrolled in a mainstream public school second-grade class that she Andrés Martin, M.D., M.P.H. enjoyed. Emily lived in the home of a family friend with her mother (Kim), her older half sister, and her younger brother. Up until 2 years before, Emily had lived with her mother, siblings, and father in their own home. Kim felt Rapunzel had magnificent long , fine as spun that family life had been generally good during the mar- gold. riage and that her husband had been a good father; —The Grimm Brothers, Rapunzel (1) Emily was especially close to him. Emily’s father, how- ever, had a problem that led to dissolu- Rapunzel’s hair fell to the ground like a rainbow. tion of the marriage 2 years before admission. After this, It was as strong as a dandelion the parents’ relationship became strained, and Emily and as strong as a dog leash. had not seen her father in a year. Emily was briefly in counseling to help her deal with the —Anne Sexton, Rapunzel (2) changes in her life surrounding her parents’ separation, but the counselor felt that she was cop- ing well and did not require further psy- History of the Problem chotherapy. Kim, however, expressed “[It] had a long tail private concern that Emily was not deal- Emily (all names and identifiers have ing with the loss of her father. Mean- been changed to protect confidentiality) that extended past the while, Emily had started at a new school was a 7-year-old girl seen by her pedia- duodenum, a rare and was doing well academically and trician for a routine annual examina- socially. tion. Although Emily had voiced no complication… With this background information, complaints, her mother had noted that further evaluation was deferred until she had appeared paler than usual. referred to as after the surgery, when the identity of Upon physical examination, her pedia- Rapunzel syndrome.” the mass would be elucidated. trician noted a nontender palpable mass in the child’s , leading Surgical Procedure him to order an abdominal ultrasound. The study showed no evidence of any abnormalities, al- Because the mass in Emily’s stomach appeared too though the technician did note that Emily must have just large to be removed laparoscopically, a gastrotomy was eaten because her stomach was full. However, her mother performed instead through a 7-cm median xiphoumbili- reported that Emily had not eaten since breakfast, 4 cal incision. The mass was found to be a trichobezoar, or hours earlier. hairball, 45 cm long and 8 cm in diameter (Figure 2). The On reevaluation the next day, Emily’s pediatrician still hair mass was cast in the shape of the stomach, which it palpated the same mass, leading him to order a com- filled, and extended down through the distal end of the puted tomography (CT) scan of the abdomen. The abdom- duodenum, past the ligament of Treitz. inal CT demonstrated a free-floating mass that filled the entire stomach and was surrounded by a thin rim of con- Trichobezoars trast material (Figure 1). Emily’s pediatrician did not think The smooth surface of hair does not allow for its propa- the mass was consistent with a neoplasm but was con- gation through peristalsis. Thus, when hair is ingested, it cerned that it could be a conglomeration of something she had ingested. Upon further questioning, Emily re- gets trapped in the mucosa of the stomach. As more hair is ported no stomach pain, nausea, vomiting, reflux, diar- added, the resulting mass causes the stomach to cease rhea, flatulence, recent illnesses, or fever. Her mother re- peristalsis completely. This mass is a trichobezoar (3), first ported no change in her bowel habits and stated that described in 1779 by Baudomant (4). Emily never wanted to eat much in one sitting. Trichobezoars are nearly impossible to diagnose by Given the large size and appearance of the mass on plain film alone. Ultrasound has been shown to be effec- tomography, a decision was made to remove it surgi- tive in diagnosing in up to 88% of cases if a “clean” cally. Just as the elective surgery was scheduled, Emily acoustic shadow, which represents a solid mass, can be vi- was referred for evaluation by the child psychiatry sualized (4, 5). However, a calcified mass, neuroblastoma, consultation-liaison team. aneurysm, abscess, or fecal material can appear similarly

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FIGURE 1. Abdominal Computed Tomography Showing a Mass in the Stomach of a 7-Year-Old Childa

Right Left

a Orally ingested contrast material has been displaced against the lining of the gastric wall. on an ultrasound (4). An abdominal CT with contrast has removal (10). In the case of Emily, the trichobezoar had a been shown to diagnose 97% of bezoars (5), which appear long tail that extended past the duodenum, a rare compli- as free-floating filling defects in the stomach (3). cation of referred to as Rapunzel syn- Although trichobezoars can be removed in several dif- drome in the surgical literature. ferent ways, Emily’s was too large to avoid open surgery. Trichotillomania has many different clinical presenta- Smaller trichobezoars can be removed by endoscopy after tions. Patients with trichotillomania usually pull hair from fragmentation by water pick, laser, or extracorporeal the scalp, but , , legs, armpits, and pu- shock-wave lithotripsy (6, 7) or by enzymatically dissolv- bic regions are also targets for pulling. Although by defini- ing the mass. Finally, small bezoars can be removed lap- tion, trichotillomania entails hair pulling to the point of aroscopically through a small incision (6). However, none alopecia, there is a much larger number of individuals of these techniques are successful for very large bezoars who pull their hair but not to the point of noticeable thin- (over 20 cm), which usually require removal through open ning (11, 12). Not all hair pullers experience the sense of surgery (3). tension before pulling followed by relief afterward that is described in the DSM-IV definition; one study showed Trichotillomania, , that among college students, although 1.5% of men and and Rapunzel Syndrome 3.4% of women had chronic hair pulling, only 0.6% met the full DSM criteria for trichotillomania (9, 13). Clinical Presentation Clinical studies of trichotillomania delineate two types Trichotillomania was first described in the literature in of the disorder, which may coexist in one individual (14). 1889 (3) and first recognized by APA as a distinct disorder The “focused” type is trichotillomania in which time and in 1987 (8). The prevalence of trichotillomania is 0.6%– attention are set aside specifically for the purpose of hair 1.6% if DSM-IV criteria are used (9). Among those who suf- pulling. This type of hair pulling is associated with ten- fer from trichotillomania, only 30% will engage in tricho- sion before and relief after pulling, and with negative af- phagia, or eating their hair, and of these, only some 1% fect. More common is the “automatic” or “sedentary” will go on to eat their hair to the extent requiring surgical type of hair pulling, in which subjects pull their hair while

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FIGURE 2. Trichobezoar From a 7-Year-Old Childa

a Specimen preparation and photograph courtesy of Dr. Jonathan Bennett, Department of Pathology, Yale-New Haven Hospital and Yale University School of Medicine. engaged in other activities, such as lying in bed, driving, dren who begin hair pulling before the age of 8, the disor- or watching television (15, 16). Patients with the latter der is usually benign and self-limited, although not all type are unaware that they are pulling their hair at all or cases disappear. Most of these cases are of the sedentary become aware only when a clump of hair “appears” in type, and the children, when verbal, often deny pulling their hands (13). Many patients with trichotillomania (15). In one case, a 28-month-old girl was found to be pull- have other associated habits; about 48%–70% have some ing her own hair, and upon review of her history, it was form of oral behavior after pulling hair, such as running found that she had pulled and eaten her dolls’ hair as early the hair across their lips, biting the root, or eating the hair as 6 months of age. Thus, in early-onset trichotillomania, (13, 16). pulling and eating dolls’ hair can be a first symptom of tri- chotillomania (17). Although habits such as thumb suck- Developmental Features ing have been noted in early-onset trichotillomania, other Trichotillomania is far more common in girls than boys. comorbid features have not been found (15). The age at onset of trichotillomania is bimodal; it begins Trichotillomania occurring in the later school years and either in early childhood or in adolescence (15). adolescence is more likely to be chronic and accompanied Of the early-onset cases, one-third develop it before the by comorbid psychiatric disorders, such as , age of 10, and 14% develop it before the age of 7. In chil- anxiety, obsessive-compulsive disorder (OCD), body dys-

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FIGURE 3. Scanning Electron Micrographs of Gold-Coated Fresh Hair Clippings From a 7-Year-Old Child and Her Mother and From a Random Sample of the Bezoara

Hair from mother, 1000x magnification Hair from child, 1000x magnification

Sample from trichobezoar, 500x magnification

a The distinctive diameter and surface scaling pattern of both hair strands are evident in a random sample taken from the trichobezoar. Images courtesy of Mr. Barry Piekos, Department of Molecular, Cellular, and Developmental Biology, Yale University. morphic disorder, eating disorders, and alcohol and sub- dren with trichobezoars or Rapunzel syndrome are sparse, stance abuse (13, 15, 18). As is generally true, clinical sam- and many link the trichophagia either to early childhood ples of patients seeking treatment for trichotillomania deprivation or abuse (7, 10, 21), psychiatric comorbid con- appear to have higher rates of than nonre- ditions (21, 22), or mental retardation (23, 24). While there ferred community groups (11). are case studies of individuals with trichobezoars who seemingly had none of these associated conditions (25– Complications 32), cases severe enough to require surgical removal are The physical complications of trichotillomania include certainly rare. rash or infection on the scalp, repetitive injury, and carpal tunnel syndrome (19). When trichophagia is present, A Parent’s Hair complications can also include gingivitis and, secondary to formation of a trichobezoar, gastrointestinal blockage The cutting of hair represents a restitution offered and perforation or secretory diarrhea and malabsorption the mother, whom the subject fancies to have harmed of nutrients, leading to vitamin deficiencies (10, 19, 20). in some fashion. While rare, the formation of trichobezoars is the most seri- ous complication of trichotillomania. Case reports of chil- —Jeffrey J. Anderson (33)

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Further Psychiatric Consultation social stressors in her life. Socially, there were factors to predispose her to psychopathology: an unstable family Upon discovering that Emily’s stomach mass was a constellation, exposure to marital arguments, the loss of trichobezoar, the psychiatric team reapproached her. her father, and her change of school may all have contrib- Emily clearly had several nonspecific vulnerabilities put- ting her at risk for a psychiatric disorder: she had re- uted (14). From a dynamic perspective, her hair eating cently lost her father and had changed schools. Earlier, could reflect a desire to hold on to and keep inside what her father had been periodically absent and had a his- she had lost—perhaps her father. Rather than outwardly tory of substance abuse. Furthermore, she had been wit- expressing her feelings of loss, Emily may be conceptual- ness to significant marital discord. However, these alone ized as physically holding on to her past life by literally in- did not shed light on Emily’s trichophagia. gesting it and holding it within. Another explanation, from Kim was completely taken aback by the finding in her a cognitive psychological perspective, might posit that daughter’s stomach, since she had never seen Emily eat Emily ate her hair because she had unopposed impulses or pull her own hair. She remembered that when Emily to do so. These internal feelings may have come to be was 3, she had had the habit of chewing on her hair and her dolls’ hair, and this had concerned her. Her pediatri- because she felt a need for control in a world that was cian, however, had not been concerned. Subsequently, changing around her while she was powerless. Or these Emily appeared to have stopped these behaviors. And compulsive urges could have been related to a biological yet, Emily’s hair had seemed to grow very slowly, and predisposition, maybe mediated through vulnerable sero- she had needed few haircuts during her 7 years. Emily, tonergic pathways. An argument can also be made from a when interviewed alone, agreed that she used to chew behavioral perspective that perhaps having once tried her hair when she was “little.” Upon further questioning, pulling and eating her hair, Emily found it soothing and she admitted that she had in the past sometimes twirled continued to maladaptively engage in it for the desired her hair around a finger and that hair would at times break off, and she would eat it. She denied a sense of positive effect. Emily’s reasons for starting to pull and eat tension or an urge before pulling or a sense of relief af- her hair probably have aspects of all of these explanations, terward. She said that she no longer pulled her hair but as well as other idiosyncratic reasons. could remember doing it in the first grade. When asked Another way to conceptualize Emily’s trichotillomania why she had hair in her stomach, Emily was unable to accompanied by trichophagia is to group it with compul- explain how the hair had gotten there. sive forms of disordered eating that may serve the func- Kim and Emily’s older sister both had very , tion of self-soothing, namely bulimia, , and rumina- reaching past their waists. Although Kim assumed that tion. Both trichophagia and pica involve an urge to eat she did shed hair around the house, she doubted that Emily was eating her hair and stated that she always kept nonnutritive substances. The literature on pica distin- her in her purse, where Emily would not have guishes between voluntary pica (eating in response to access to it. what is available) and involuntary pica (eating in response Determining with certainty the provenance of the hair to a compulsion or addiction) (34). The possibility that in the was a complicated process. The length of some cases of pica may respond to selective serotonin re- individual strands (over 25 inches for the longer ones) uptake inhibitors (SSRIs) has led some to view it as a form pointed to the mother’s and sister’s hair, as Emily’s own of compulsion and to theorize that pica belongs on the had never been over 12 inches long. The damage to the same compulsivity-impulsivity spectrum that includes tri- follicles and their cells did not permit DNA analysis that chotillomania, trichophagia, and OCD (35). would have been confirmatory, and light microscopy did not help in differentiation. Scanning electron micros- Thus, a parsimonious view is that Emily probably had copy did offer useful information to distinguish between trichotillomania and trichophagia compounded by pica— the daughter’s and mother’s hair. Specifically, the differ- eating her own hair, as well as her mother’s, just as she had ent widths and surface scaling patterns of the two al- earlier chewed on her dolls’ hair, similar to the case noted lowed for ready differentiation of fresh hair clippings, as in the literature (17). As her onset was clearly before the well as of hair samples taken from the bezoar itself (Fig- age of 8, her trichotillomania can be characterized as an ure 3). In summary, the combination of gross pathology early-onset form. As is characteristic of that form, she ap- and scanning electron microscopy techniques confirmed parently did not experience any urge or sense of relief in that hair in the bezoar had derived from at least two sources: Emily and her mother. pulling, as required by the DSM-IV criteria for the disor- der. Like many children with this form of trichotillomania, Psychiatric Formulation she appeared to have no other psychiatric comorbid con- Emily appeared to be an intelligent, cheerful child who ditions. Emily also experienced significant social stressors reported no negative affect and was either very secretive in her home, such as has been reported to often accom- about her trichophagia or entirely unaware of it. She had pany the onset of childhood trichotillomania (14). no alopecia or hair thinning, but had clearly had tricho- phagia of her own and her mother’s hair. Reclaiming Rapunzel Emily seemed to be functioning well outside the area of her hair eating and possible hair pulling. The etiology of Rapunzel found the means of escaping her predica- her trichophagia and apparent trichotillomania can be ment with her own body. considered from multiple viewpoints, all informed by the —Bruno Bettelheim (36, p. 17)

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But what more reliable source to recovery do we covery from abdominal surgery. Seen at 1 and 2 months have than our own body? after discharge, she quickly thrived. She returned and re- —Bruno Bettelheim (36, p. 149) adapted rapidly to school and family life. She appeared happy and carefree, reported no symptoms of depres- sion or anxiety, and continued to deny pulling or eating Treatment Alternatives of hair—her own or anyone else’s. Despite the watchful There is no formal set of guidelines for the treatment of vigilance of her mother, sister, and grandparents, she trichotillomania in children. Common treatment modali- was not found to engage in hair pulling or hair eating, ties include medication and cognitive behavior therapy and no areas of hair thinning or loss became evident. There was no evidence that trichotillomania, tricho- (14), although their empirical support base for children is phagia, or pica had recurred. limited. Support groups, which appear useful and well ac- In light of these facts, specific treatment as outlined was cepted in adults, are also increasingly available to younger not instituted at a specialty clinic, as had originally been patients through advocacy groups, such as the Trichotillo- considered necessary. Rather, Emily was referred for sup- Learning Center in Santa Cruz, Calif. (37). portive individual and family counseling at a more conve- A study of 123 patients with trichotillomania with a mean niently located clinic and for periodic monitoring through age at onset of 11 years found that of the 42% who had been the surgery and specialized child psychiatric services. treated, there was no difference in responses to psycho- Given that her hair-eating behavior may have continued undetected or eventually recurred, minimally invasive fol- therapy, behavior therapy, clomipramine, or fluoxetine. low-up tests were to be performed biannually to ensure Furthermore, none of the treatments provided significant that no hair reaccumulation took place. These included and lasting improvement of symptoms (38). In the absence abdominal ultrasound or simple abdominal X-rays after of convincing evidence for the effectiveness of medication swallowing barium. Endoscopy or CT would be avoided in childhood trichotillomania, many clinicians would opt unless absolutely necessary, given their intrusive nature for habit-reversal therapy as the initial, most benign inter- and higher radiation dose, respectively. vention (38), with the possible subsequent addition of an The overall prognosis for Emily is favorable. She has a SSRI or clomipramine, especially if there is comorbid de- supportive and loving family; she has many friends, does pression, anxiety, or OCD. well academically, and has a positive affect. Moreover, the Habit-reversal training is a form of behavioral therapy in natural history of trichotillomania is that most girls who which the focus is on eliminating an unwanted chronic have onset before age 8 outgrow the disorder by adoles- habit. It may include a cognitive component, in which the cence (15). Nevertheless, she also has factors that will con- patient and therapist address the thoughts associated with tinue to remain challenging, and recurrent trichobezoars the behavior and replace these with more positive have been previously reported in the literature. thoughts. Azrin et al. (39) demonstrated that habit-reversal Overall, a hopeful outlook seems justified. A longer- training reduced the symptoms of trichotillomania in term treatment goal will be to empower Emily to use al- 74%, while negative practice (deliberately doing the be- ternative and more adaptive tools for dealing with the havior in mass practice) reduced the symptoms in only stressors in her life—in essence, to learn to use her mind, 33%. At the 22-month follow-up, nine of the 12 in the rather than her stomach or her hair, as an instrument for habit-reversal training group had achieved remission, coping. With familial and professional help and support, while only two of the eight had in the negative practice we can hope that Emily will be able to overcome her tri- group. Both this original and subsequent simplified ver- chotillomania, pica, and trichophagia, to reclaim control sions of habit-reversal training have proven effective in over her behavior and her body, and to expunge “Rapun- achieving near or complete remission of trichotillomania zel” back into the fairy-tale books where it belongs. in children (40–42). A study of 16 subjects comparing cog- nitive behavior therapy to clomipramine and placebo Received June 30, 2004; accepted Aug. 11, 2004. From the Child found that cognitive behavior therapy was significantly Study Center, Yale University School of Medicine; and the Depart- better than clomipramine or placebo in reducing the ments of Child Psychiatry and Pediatric Surgery, Yale-New Haven symptoms of trichotillomania, while clomipramine and Hospital, New Haven, Conn. Address correspondence and reprint placebo did not significantly differ from each other in pro- requests to Dr. Martin, Child Study Center, Yale University School of Medicine, 230 South Frontage Rd., New Haven, CT 06520–7900; ducing clinically meaningful improvement (43). A study [email protected] (e-mail). comparing fluoxetine to behavior therapy in 43 subjects Supported in part by a Career Developmental Award (MH-01792) to also showed the best outcome in those using behavior Dr. Martin. therapy (44). Similar behavioral interventions have also The authors thank “Emily” and her family for their cooperation, and Drs. Parsia Vagefi, Allan Goldstein, Diane Findley, Ronald Salem, been adapted successfully for the treatment of pica, par- Paul Fiedler, Zeev Kain, Michael Kashgarian, and Miguel Reyes- ticularly among individuals with developmental disabili- Mugica for their comments on earlier drafts of this article. ties (45), among whom the prevalence of the condition has been reported to be as high as 25.8% (46). References Clinical Follow-Up and Prognosis 1. Grimm Brothers: Rapunzel. Translated by Godwin-Jones R. Emily was referred for outpatient psychiatric services Richmond, Virginia Commonwealth University Department of after her discharge from the hospital and uneventful re- Foreign Languages, 1994–1999

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