Clinical Case Conference
Hair Apparent: Rapunzel Syndrome
Ariel S. Frey, B.A., M.A.T.
Initial Psychiatric Consultation
Milissa McKee, M.D., M.P.H.
Robert A. King, M.D.
Andrés Martin, M.D., M.P.H.
Rapunzel had magnificent long hair, fine as spun
gold.
—The Grimm Brothers, Rapunzel (1)
Rapunzel’s hair fell to the ground like a rainbow.
It was as strong as a dandelion
and as strong as a dog leash.
—Anne Sexton, Rapunzel (2)
History of the Problem
“[It] had a long tail
that extended past the
duodenum, a rare
complication…
referred to as
Rapunzel syndrome.”
Emily (all names and identifiers have
been changed to protect confidentiality)
was a 7-year-old girl seen by her pediatrician for a routine annual examination. Although Emily had voiced no
complaints, her mother had noted that
she had appeared paler than usual.
Upon physical examination, her pediatrician noted a nontender palpable
mass in the child’s stomach, leading
him to order an abdominal ultrasound.
The study showed no evidence of any abnormalities, although the technician did note that Emily must have just
eaten because her stomach was full. However, her mother
reported that Emily had not eaten since breakfast, 4
hours earlier.
On reevaluation the next day, Emily’s pediatrician still
palpated the same mass, leading him to order a computed tomography (CT) scan of the abdomen. The abdominal CT demonstrated a free-floating mass that filled the
entire stomach and was surrounded by a thin rim of contrast material (Figure 1). Emily’s pediatrician did not think
the mass was consistent with a neoplasm but was concerned that it could be a conglomeration of something
she had ingested. Upon further questioning, Emily reported no stomach pain, nausea, vomiting, reflux, diarrhea, flatulence, recent illnesses, or fever. Her mother reported no change in her bowel habits and stated that
Emily never wanted to eat much in one sitting.
Given the large size and appearance of the mass on
tomography, a decision was made to remove it surgically. Just as the elective surgery was scheduled, Emily
was referred for evaluation by the child psychiatry
consultation-liaison team.
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The child psychiatry team (A.S.F. and A.M.) met with
Emily and her mother the morning before surgery. By
her own and her mother’s account, she was happy and
well adjusted. She had many friends and was enrolled in
a mainstream public school second-grade class that she
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
that family life had been generally good during the marriage and that her husband had been a good father;
Emily was especially close to him. Emily’s father, however, had a substance abuse problem that led to dissolution of the marriage 2 years before admission. After this,
the parents’ relationship became strained, and Emily
had not seen her father in a year.
Emily was briefly in counseling to help her deal with the
changes in her life surrounding her parents’ separation,
but the counselor felt that she was coping well and did not require further psychotherapy. Kim, however, expressed
private concern that Emily was not dealing with the loss of her father. Meanwhile, Emily had started at a new school
and was doing well academically and
socially.
With this background information,
further evaluation was deferred until
after the surgery, when the identity of
the mass would be elucidated.
Surgical Procedure
Because the mass in Emily’s stomach appeared too
large to be removed laparoscopically, a gastrotomy was
performed instead through a 7-cm median xiphoumbilical incision. The mass was found to be a trichobezoar, or
hairball, 45 cm long and 8 cm in diameter (Figure 2). The
hair mass was cast in the shape of the stomach, which it
filled, and extended down through the distal end of the
duodenum, past the ligament of Treitz.
Trichobezoars
The smooth surface of hair does not allow for its propagation through peristalsis. Thus, when hair is ingested, it
gets trapped in the mucosa of the stomach. As more hair is
added, the resulting mass causes the stomach to cease
peristalsis completely. This mass is a trichobezoar (3), first
described in 1779 by Baudomant (4).
Trichobezoars are nearly impossible to diagnose by
plain film alone. Ultrasound has been shown to be effective in diagnosing bezoars in up to 88% of cases if a “clean”
acoustic shadow, which represents a solid mass, can be visualized (4, 5). However, a calcified mass, neuroblastoma,
aneurysm, abscess, or fecal material can appear similarly
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CLINICAL CASE CONFERENCE
FIGURE 1. Abdominal Computed Tomography Showing a Mass in the Stomach of a 7-Year-Old Childa
Right
a
Left
Orally ingested contrast material has been displaced against the lining of the gastric wall.
on an ultrasound (4). An abdominal CT with contrast has
been shown to diagnose 97% of bezoars (5), which appear
as free-floating filling defects in the stomach (3).
Although trichobezoars can be removed in several different ways, Emily’s was too large to avoid open surgery.
Smaller trichobezoars can be removed by endoscopy after
fragmentation by water pick, laser, or extracorporeal
shock-wave lithotripsy (6, 7) or by enzymatically dissolving the mass. Finally, small bezoars can be removed laparoscopically through a small incision (6). However, none
of these techniques are successful for very large bezoars
(over 20 cm), which usually require removal through open
surgery (3).
Trichotillomania, Trichophagia,
and Rapunzel Syndrome
Clinical Presentation
Trichotillomania was first described in the literature in
1889 (3) and first recognized by APA as a distinct disorder
in 1987 (8). The prevalence of trichotillomania is 0.6%–
1.6% if DSM-IV criteria are used (9). Among those who suffer from trichotillomania, only 30% will engage in trichophagia, or eating their hair, and of these, only some 1%
will go on to eat their hair to the extent requiring surgical
Am J Psychiatry 162:2, February 2005
removal (10). In the case of Emily, the trichobezoar had a
long tail that extended past the duodenum, a rare complication of trichotillomania referred to as Rapunzel syndrome in the surgical literature.
Trichotillomania has many different clinical presentations. Patients with trichotillomania usually pull hair from
the scalp, but eyelashes, eyebrows, legs, armpits, and pubic regions are also targets for pulling. Although by definition, trichotillomania entails hair pulling to the point of
alopecia, there is a much larger number of individuals
who pull their hair but not to the point of noticeable thinning (11, 12). Not all hair pullers experience the sense of
tension before pulling followed by relief afterward that is
described in the DSM-IV definition; one study showed
that among college students, although 1.5% of men and
3.4% of women had chronic hair pulling, only 0.6% met
the full DSM criteria for trichotillomania (9, 13).
Clinical studies of trichotillomania delineate two types
of the disorder, which may coexist in one individual (14).
The “focused” type is trichotillomania in which time and
attention are set aside specifically for the purpose of hair
pulling. This type of hair pulling is associated with tension before and relief after pulling, and with negative affect. More common is the “automatic” or “sedentary”
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,
or watching television (15, 16). Patients with the latter
type are unaware that they are pulling their hair at all or
become aware only when a clump of hair “appears” in
their hands (13). Many patients with trichotillomania
have other associated habits; about 48%–70% have some
form of oral behavior after pulling hair, such as running
the hair across their lips, biting the root, or eating the hair
(13, 16).
Developmental Features
Trichotillomania is far more common in girls than boys.
The age at onset of trichotillomania is bimodal; it begins
either in early childhood or in adolescence (15).
Of the early-onset cases, one-third develop it before the
age of 10, and 14% develop it before the age of 7. In chil-
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dren who begin hair pulling before the age of 8, the disorder is usually benign and self-limited, although not all
cases disappear. Most of these cases are of the sedentary
type, and the children, when verbal, often deny pulling
(15). In one case, a 28-month-old girl was found to be pulling her own hair, and upon review of her history, it was
found that she had pulled and eaten her dolls’ hair as early
as 6 months of age. Thus, in early-onset trichotillomania,
pulling and eating dolls’ hair can be a first symptom of trichotillomania (17). Although habits such as thumb sucking have been noted in early-onset trichotillomania, other
comorbid features have not been found (15).
Trichotillomania occurring in the later school years and
adolescence is more likely to be chronic and accompanied
by comorbid psychiatric disorders, such as depression,
anxiety, obsessive-compulsive disorder (OCD), body dysAm J Psychiatry 162:2, February 2005
CLINICAL CASE CONFERENCE
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 substance abuse (13, 15, 18). As is generally true, clinical samples of patients seeking treatment for trichotillomania
appear to have higher rates of comorbidity than nonreferred community groups (11).
Complications
The physical complications of trichotillomania include
rash or infection on the scalp, repetitive stress injury, and
carpal tunnel syndrome (19). When trichophagia is present,
complications can also include gingivitis and, secondary
to formation of a trichobezoar, gastrointestinal blockage
and perforation or secretory diarrhea and malabsorption
of nutrients, leading to vitamin deficiencies (10, 19, 20).
While rare, the formation of trichobezoars is the most serious complication of trichotillomania. Case reports of chilAm J Psychiatry 162:2, February 2005
dren with trichobezoars or Rapunzel syndrome are sparse,
and many link the trichophagia either to early childhood
deprivation or abuse (7, 10, 21), psychiatric comorbid conditions (21, 22), or mental retardation (23, 24). While there
are case studies of individuals with trichobezoars who
seemingly had none of these associated conditions (25–
32), cases severe enough to require surgical removal are
certainly rare.
A Parent’s Hair
The cutting of hair represents a restitution offered
the mother, whom the subject fancies to have harmed
in some fashion.
—Jeffrey J. Anderson (33)
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CLINICAL CASE CONFERENCE
Further Psychiatric Consultation
Upon discovering that Emily’s stomach mass was a
trichobezoar, the psychiatric team reapproached her.
Emily clearly had several nonspecific vulnerabilities putting her at risk for a psychiatric disorder: she had recently lost her father and had changed schools. Earlier,
her father had been periodically absent and had a history of substance abuse. Furthermore, she had been witness to significant marital discord. However, these alone
did not shed light on Emily’s trichophagia.
Kim was completely taken aback by the finding in her
daughter’s stomach, since she had never seen Emily eat
or pull her own hair. She remembered that when Emily
was 3, she had had the habit of chewing on her hair and
her dolls’ hair, and this had concerned her. Her pediatrician, however, had not been concerned. Subsequently,
Emily appeared to have stopped these behaviors. And
yet, Emily’s hair had seemed to grow very slowly, and
she had needed few haircuts during her 7 years. Emily,
when interviewed alone, agreed that she used to chew
her hair when she was “little.” Upon further questioning,
she admitted that she had in the past sometimes twirled
her hair around a finger and that hair would at times
break off, and she would eat it. She denied a sense of
tension or an urge before pulling or a sense of relief afterward. She said that she no longer pulled her hair but
could remember doing it in the first grade. When asked
why she had hair in her stomach, Emily was unable to
explain how the hair had gotten there.
Kim and Emily’s older sister both had very long hair,
reaching past their waists. Although Kim assumed that
she did shed hair around the house, she doubted that
Emily was eating her hair and stated that she always kept
her hairbrush in her purse, where Emily would not have
access to it.
Determining with certainty the provenance of the hair
in the bezoar was a complicated process. The length of
individual strands (over 25 inches for the longer ones)
pointed to the mother’s and sister’s hair, as Emily’s own
had never been over 12 inches long. The damage to the
follicles and their cells did not permit DNA analysis that
would have been confirmatory, and light microscopy did
not help in differentiation. Scanning electron microscopy did offer useful information to distinguish between
the daughter’s and mother’s hair. Specifically, the different widths and surface scaling patterns of the two allowed for ready differentiation of fresh hair clippings, as
well as of hair samples taken from the bezoar itself (Figure 3). In summary, the combination of gross pathology
and scanning electron microscopy techniques confirmed
that hair in the bezoar had derived from at least two
sources: Emily and her mother.
Psychiatric Formulation
Emily appeared to be an intelligent, cheerful child who
reported no negative affect and was either very secretive
about her trichophagia or entirely unaware of it. She had
no alopecia or hair thinning, but had clearly had trichophagia of her own and her mother’s hair.
Emily seemed to be functioning well outside the area of
her hair eating and possible hair pulling. The etiology of
her trichophagia and apparent trichotillomania can be
considered from multiple viewpoints, all informed by the
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social stressors in her life. Socially, there were factors to
predispose her to psychopathology: an unstable family
constellation, exposure to marital arguments, the loss of
her father, and her change of school may all have contributed (14). From a dynamic perspective, her hair eating
could reflect a desire to hold on to and keep inside what
she had lost—perhaps her father. Rather than outwardly
expressing her feelings of loss, Emily may be conceptualized as physically holding on to her past life by literally ingesting it and holding it within. Another explanation, from
a cognitive psychological perspective, might posit that
Emily ate her hair because she had unopposed impulses
to do so. These internal feelings may have come to be
because she felt a need for control in a world that was
changing around her while she was powerless. Or these
compulsive urges could have been related to a biological
predisposition, maybe mediated through vulnerable serotonergic pathways. An argument can also be made from a
behavioral perspective that perhaps having once tried
pulling and eating her hair, Emily found it soothing and
continued to maladaptively engage in it for the desired
positive effect. Emily’s reasons for starting to pull and eat
her hair probably have aspects of all of these explanations,
as well as other idiosyncratic reasons.
Another way to conceptualize Emily’s trichotillomania
accompanied by trichophagia is to group it with compulsive forms of disordered eating that may serve the function of self-soothing, namely bulimia, pica, and rumination. Both trichophagia and pica involve an urge to eat
nonnutritive substances. The literature on pica distinguishes between voluntary pica (eating in response to
what is available) and involuntary pica (eating in response
to a compulsion or addiction) (34). The possibility that
some cases of pica may respond to selective serotonin reuptake inhibitors (SSRIs) has led some to view it as a form
of compulsion and to theorize that pica belongs on the
same compulsivity-impulsivity spectrum that includes trichotillomania, trichophagia, and OCD (35).
Thus, a parsimonious view is that Emily probably had
trichotillomania and trichophagia compounded by pica—
eating her own hair, as well as her mother’s, just as she had
earlier chewed on her dolls’ hair, similar to the case noted
in the literature (17). As her onset was clearly before the
age of 8, her trichotillomania can be characterized as an
early-onset form. As is characteristic of that form, she apparently did not experience any urge or sense of relief in
pulling, as required by the DSM-IV criteria for the disorder. Like many children with this form of trichotillomania,
she appeared to have no other psychiatric comorbid conditions. Emily also experienced significant social stressors
in her home, such as has been reported to often accompany the onset of childhood trichotillomania (14).
Reclaiming Rapunzel
Rapunzel found the means of escaping her predicament with her own body.
—Bruno Bettelheim (36, p. 17)
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CLINICAL CASE CONFERENCE
But what more reliable source to recovery do we
have than our own body?
—Bruno Bettelheim (36, p. 149)
Treatment Alternatives
There is no formal set of guidelines for the treatment of
trichotillomania in children. Common treatment modalities include medication and cognitive behavior therapy
(14), although their empirical support base for children is
limited. Support groups, which appear useful and well accepted in adults, are also increasingly available to younger
patients through advocacy groups, such as the Trichotillomania Learning Center in Santa Cruz, Calif. (37).
A study of 123 patients with trichotillomania with a mean
age at onset of 11 years found that of the 42% who had been
treated, there was no difference in responses to psychotherapy, behavior therapy, clomipramine, or fluoxetine.
Furthermore, none of the treatments provided significant
and lasting improvement of symptoms (38). In the absence
of convincing evidence for the effectiveness of medication
in childhood trichotillomania, many clinicians would opt
for habit-reversal therapy as the initial, most benign intervention (38), with the possible subsequent addition of an
SSRI or clomipramine, especially if there is comorbid depression, anxiety, or OCD.
Habit-reversal training is a form of behavioral therapy in
which the focus is on eliminating an unwanted chronic
habit. It may include a cognitive component, in which the
patient and therapist address the thoughts associated with
the behavior and replace these with more positive
thoughts. Azrin et al. (39) demonstrated that habit-reversal
training reduced the symptoms of trichotillomania in
74%, while negative practice (deliberately doing the behavior in mass practice) reduced the symptoms in only
33%. At the 22-month follow-up, nine of the 12 in the
habit-reversal training group had achieved remission,
while only two of the eight had in the negative practice
group. Both this original and subsequent simplified versions of habit-reversal training have proven effective in
achieving near or complete remission of trichotillomania
in children (40–42). A study of 16 subjects comparing cognitive behavior therapy to clomipramine and placebo
found that cognitive behavior therapy was significantly
better than clomipramine or placebo in reducing the
symptoms of trichotillomania, while clomipramine and
placebo did not significantly differ from each other in producing clinically meaningful improvement (43). A study
comparing fluoxetine to behavior therapy in 43 subjects
also showed the best outcome in those using behavior
therapy (44). Similar behavioral interventions have also
been adapted successfully for the treatment of pica, particularly among individuals with developmental disabilities (45), among whom the prevalence of the condition has
been reported to be as high as 25.8% (46).
Clinical Follow-Up and Prognosis
Emily was referred for outpatient psychiatric services
after her discharge from the hospital and uneventful re-
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covery from abdominal surgery. Seen at 1 and 2 months
after discharge, she quickly thrived. She returned and readapted rapidly to school and family life. She appeared
happy and carefree, reported no symptoms of depression or anxiety, and continued to deny pulling or eating
of hair—her own or anyone else’s. Despite the watchful
vigilance of her mother, sister, and grandparents, she
was not found to engage in hair pulling or hair eating,
and no areas of hair thinning or loss became evident.
There was no evidence that trichotillomania, trichophagia, or pica had recurred.
In light of these facts, specific treatment as outlined was
not instituted at a specialty clinic, as had originally been
considered necessary. Rather, Emily was referred for supportive individual and family counseling at a more conveniently located clinic and for periodic monitoring through
the surgery and specialized child psychiatric services.
Given that her hair-eating behavior may have continued
undetected or eventually recurred, minimally invasive follow-up tests were to be performed biannually to ensure
that no hair reaccumulation took place. These included
abdominal ultrasound or simple abdominal X-rays after
swallowing barium. Endoscopy or CT would be avoided
unless absolutely necessary, given their intrusive nature
and higher radiation dose, respectively.
The overall prognosis for Emily is favorable. She has a
supportive and loving family; she has many friends, does
well academically, and has a positive affect. Moreover, the
natural history of trichotillomania is that most girls who
have onset before age 8 outgrow the disorder by adolescence (15). Nevertheless, she also has factors that will continue to remain challenging, and recurrent trichobezoars
have been previously reported in the literature.
Overall, a hopeful outlook seems justified. A longerterm treatment goal will be to empower Emily to use alternative and more adaptive tools for dealing with the
stressors in her life—in essence, to learn to use her mind,
rather than her stomach or her hair, as an instrument for
coping. With familial and professional help and support,
we can hope that Emily will be able to overcome her trichotillomania, pica, and trichophagia, to reclaim control
over her behavior and her body, and to expunge “Rapunzel” back into the fairy-tale books where it belongs.
Received June 30, 2004; accepted Aug. 11, 2004. From the Child
Study Center, Yale University School of Medicine; and the Departments of Child Psychiatry and Pediatric Surgery, Yale-New Haven
Hospital, New Haven, Conn. Address correspondence and reprint
requests to Dr. Martin, Child Study Center, Yale University School of
Medicine, 230 South Frontage Rd., New Haven, CT 06520–7900;
[email protected] (e-mail).
Supported in part by a Career Developmental Award (MH-01792) to
Dr. Martin.
The authors thank “Emily” and her family for their cooperation,
and Drs. Parsia Vagefi, Allan Goldstein, Diane Findley, Ronald Salem,
Paul Fiedler, Zeev Kain, Michael Kashgarian, and Miguel ReyesMugica for their comments on earlier drafts of this article.
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