This case report provides a rare example of clinical linkage between anorexia nervosa and catatonia in an adolescent. It also reviews diagnostic considerations in formulating this highly unusual presentation.
A developmentally normal adolescent boy of high academic achievement without formal psychiatric history presented to the emergency department. He was brought in by his parents, secondary to reported suicidal ideation with a plan to jump off a tall building. The parents reported the patient had minimal sleep over the past 3 to 4 days, with repetitive thoughts, bizarre behaviors, and shift in personality. He had weight loss of about 70 pounds over the past year and restricted food intake. He reported doing 500 stomach crunches, 250 push-ups, and 40 minutes of running on a treadmill per day, a pattern that had developed about 4 to 5 months prior to presentation, without evidence of intensification just prior to presentation. The patient stated he felt tired of everything and that death would be a release. His mood was reported as anxious. Rapid pressured speech with racing thoughts beginning the day prior to presentation quickly developed into behavioral agitation, including yelling and scratching at skin and extremities. His physical examination was unremarkable other than for emaciated appearance.
Initial vital signs included a heart rate of 60 beats per minute, temperature of 37.4°C, blood pressure of 127/88 mm Hg, respirations of 16 per minute, and oxygen saturation of 100% on room air. His height was 182.9 cm, with weight on admission of 53.3 kg, yielding a body mass index (BMI) of 15.93 kg/m2. An electrocardiogram was notable for sinus bradycardia to 54. Initial laboratory evaluation (reference ranges in parentheses) was notable for elevation in bicarbonate to 30 mEq/L (22–29 mEq/L), creatinine of 1 mg/dL (0.4–0.9 mg/dL), blood urea nitrogen of 23 mg/dL (10–20 mg/dL), platelet count of 136 × 109/L (150–400 × 109/L), mildly elevated thyroid-stimulating hormone of 6.15 mIU/L (0.27–4.20 mIU/L) with normal free T4 of 1.12 ng/dL (0.9–1.7 ng/dL), alkaline phosphatase of 40 IU/L (116–468 IU/L), aspartate aminotransferase of 59 U/L (10–35 U/L), alanine aminotransferase of 110 U/L (5–30 U/L), normal urinalysis, and negative urine drug screen. Other normal per tinent laboratory results include sodium of 137 mEq/L (135–145 mEq/L), potassium of 4.5 mEq/L (3.5–5 mEq/L), chloride of 96 mEq/L (95–107 mEq/L), anion gap of 11 mEq/L (<17 mEq/L), glucose of 89 mg/dL (65–139 mg/dL), calcium of 9.3 mg/dL (8.5–10.2 mg/dL), magnesium of 2.2 (1.5–2.9 mEq/L), and phosphorus of 4.5 mg/dL (2.9–5.1 mg/dL). Substance use was consistently denied by both patient and parents. Child psychiatry evaluated the patient daily over his 100+ hours in the emergency department.
Initial evaluation noted agitation, with the patient yelling “I need to eat,” before standing and placing his hands on his parents' and the consultant's heads, then becoming stiff and trembling, and then repeating that phrase. This lasted about 1 minute, after which he laid on the bed. He was nonresponsive until he began to eat with his hands, at which point he was able to participate in the interview. Inpatient psychiatric admission was recommended with concern for diagnosis of mania or psychosis, although there was no available bed. His speech soon diminished to single word communication, with agitation and aggression, after which he began pushing his mother and yelling expletives. The patient became less interactive over the next 24 hours, and repeated circular arm movements for hours after he had been tested using those motions. Attempts to interact with the patient became unsuccessful. He displayed overt features consistent with catatonia, including grimacing, stereotypy, mutism, echopraxia, and stupor.
Neurologic consultation was recommended and obtained, with testing for possible autoimmune encephalitis. Magnetic resonance imaging and computed tomography of the brain along with electroencephalogram were performed and were unremarkable. Lumbar puncture under fluoroscopy encountered technical difficulty and was traumatic, with only a small amount of cerebrospinal fluid collected, which showed 89,000 red blood cells, 48 nucleated cells (0–5), 300 mg/dL of protein (15–45 mg/dL), and xanthochromia (likely secondary to trauma). Laboratory results including creatine kinase, thiamine, and cyanocobalamin were unremarkable. A serum encephalopathy panel of autoimmune antibodies returned entirely negative.
The patient was ultimately admitted to general pediatrics, with assistance required for basic care and incontinence. He continued to have slowed movement, speech, and cognition. When a bed was available, he was transferred to inpatient psychiatry.
After stabilization, the patient was able to participate in an eating disorder-focused interview. He reported anxiety about eating food and the “volume of change” of food, with worries and fears about being “chubby” starting at age 3 or 4 years. He disliked the appearance of his stomach and reported a lot of time focusing on being healthy. He estimated his lowest weight at 120 pounds and highest at 190 pounds, with a goal somewhere between 130 and 150 pounds. Over the past year, he started restricting food without a specific caloric goal in mind other than less. He would frequently check food labels for calorie counts and sugar amounts. He started exercising to lose weight, engaging in resistance training, cardiovascular exercises, push-ups, and sit-ups. He denied engaging in binging or purging including diuretics, laxatives, diet pills, or self-induction of emesis. The patient noted strong cold intolerance, along with constipation. In the few weeks prior to admission, he would not eat breakfast or snacks, but did eat most of his lunch tray at school and ate “until full” at dinner (an example being a whole bag of steamed broccoli).
Per his parents, he experienced significant difficulty with fitting in at school to the point of physical bullying, and a mismatch between academic and social functioning. Retrospectively, they were able to trace a timeline; the changes started after a 4 or 5-inch growth spurt about 9 months prior, where he became taller and thinner and began exercising with his mother. He received positive feedback on changes in his appearance, which reinforced his behavior. His amount of exercise increased in duration and intensity, with a subsequent drop in weight. He developed restriction of entire food areas including carbohydrates, dairy, and sugar, and began eating large amounts of vegetables with proteins. He grew increasingly resistant to parental encouragement to eat more, with both surreptitious and unconcealed engagement in exercise. They further noted a strong self-critical aspect in his personality, without evidence for psychotic thought processes. Both patient and parents denied history of depressive episodes.
During admission, a clinical diagnosis of anorexia nervosa, restricting subtype, severe, was made, given his BMI on admission of 15.93 mg/kg2 (growth curve at third percentile), persistent behavior interfering with weight gain even at this significantly low weight, and lack of recognition of severity of symptoms as evidenced by focus on his behavior as being healthy.
Initial treatment focused on as-needed symptomatic management. Hydroxyzine at a dose of 10 mg every 4 hours as needed for agitation was started by the emergency department, with a recommendation for haloperidol at a dose of 2 mg oral/intramuscular or olanzapine at a dose of 2.5 mg oral/intramuscular for agitation made by the psychiatry department.
A lorazepam challenge (2 mg intravenous) was administered 3 days after presentation, with resultant increased interaction, movement, and speech observed. He was noted to be answering questions appropriately and behaving closer to his baseline. He then began scheduled lorazepam at a dose of 1 mg twice daily. He continued to speak illogically and in a fragmented fashion, with slowed movement coupled with wandering requiring redirection. Lorazepam was increased after 24 hours to 1.5 mg three times daily. Due to concerns of psychosis or mania, risperidone was started at 0.5 mg for 1 dose, then increased to 0.5 mg every morning and 1 mg at bedtime. Trazodone at a dose of 25 mg at bedtime was started to help with sleep. He received a dose of 2 mg of intramuscular haloperidol for agitation overnight. While stationed in the pediatrics department, he gradually became able to consistently eat and use the toilet independently.
Medications were converted to liquid form after a period of refusal by the patient. After an episode of rigidity, risperidone was stopped, but worsened psychotic symptoms led to its resumption so it was titrated back to 0.5 mg three times daily. Movements, speech, and cognition became more fluid over 24 days of inpatient treatment, during which time he limited exercise and ate a normal diet. Lorazepam was tapered to a dose of 1 mg three times daily. At time of discharge, his parents estimated the patient was at 50% of baseline functioning.
Electrolytes were periodically checked throughout his admission to monitor for refeeding as per facility protocol and to evaluate for supplementation needs. As the patient was eating on his own, he was not placed on a formal eating disorder protocol, although his meals were observed. Calorie counts averaged about 3,000 calories per day. Daily multivitamin and polyethylene glycol were started, along with vitamin D supplementation given his low level of 25 ng/mL (20–80 ng/mL). Bone densitometry scan was normal. Vital signs grossly remained within normal limits, with initial bradycardia earlier in the stay and occasional tachycardia. The patient's weight was monitored during his hospital stay, with him weighing 53.3 kg on admission and 58.9 kg at time of inpatient discharge.
He was discharged to an eating disorder partial hospitalization program, where he received 5 more weeks of treatment. Lorazepam dose was tapered slowly from 1 mg three times daily to 0.5 mg three times daily. Risperidone dosing was consolidated to 0.5 mg every morning and 1 mg at bedtime. Trazodone was discontinued. He continued to restore weight under supervised programming and reached 70 kg, with target of 73.5 kg. He gradually developed more insight and continued to return to his baseline.
This case raises a number of diagnostic issues. Catatonia seems unequivocal by the Diagnostic and Statistical Manual of Mental Disorders, fifth edition1 criteria coupled with response to benzodiazepines. Medical testing and evaluation are needed to rule out organic causes in someone without a formal psychiatric history whose first clinical presentation rapidly became catatonic; however, no clear medical cause was ultimately isolated in this case. Response to risperidone proved interesting, as antipsychotic use is often avoided in catatonia due to association with developing neuroleptic malignant syndrome.2 It was originally started for antimanic, antipsychotic activity, and was temporarily stopped due to possible motor rigidity. The patient's symptomatic regression after this discontinuation suggests that risperidone had efficacy despite the patient's catatonic state. Risperidone has also been used in the direct treatment of catatonia.3,4 More traditional catatonia treatment approaches with more robust evidence bases include benzodiazepines, particularly lorazepam, as well as electroconvulsive therapy.2
Even after recovery, it remains difficult to determine definitively what precipitated catatonia, with a fairly broad differential remaining. Although the neurology department concluded that test results were not consistent with an inflammatory process, autoimmune encephalitis against a target not detectable in current laboratory assays would still be possible given abrupt onset of illness and rapid changes in presentation over days. Further, more definitive testing for cerebrospinal fluid autoimmune antibodies was not feasible given the difficulty in obtaining an adequate sample from lumbar puncture. His initial presentation, including days with minimal sleep, illogical thought process and behavior, psychomotor agitation, and suicidal ideation, could be consistent with a manic episode despite no family history of bipolar illness, his young age, and no significant mood symptoms outside of this episode. Bipolar I disorder with psychotic features would be the mood disorder his symptoms most closely resemble. Equally, the patient's initial presentation could fit agitated or excited catatonia that morphed into more traditional catatonia. Schizophrenia spectrum illness would be on the differential despite its early presentation without family history and lack of perceptual disturbance. However, none of these possible precipitants would also explain the symptoms of anorexia nervosa over many months. The complex connections between anorexia and mood and psychotic disorders remain understudied and unclear.5,6
Although almost unreported in the literature, our team's hypothesis is that anorexia nervosa in the context of a provisional mood disorder diagnosis was the most likely precipitating factor here, with the possibility that malnourishment (reflected in subclinical hypothyroidism, vitamin D deficiency, loss of 70 pounds over 1 year, and elevations in liver enzymes/creatinine/blood urea nitrogen), coupled with psychosocial stressors at school and home (it has been theorized that environmental changes may precede the onset of catatonia) and inherited vulnerabilities (family history of depression and anxiety) was enough to precipitate decline into catatonia.7–9 Further study of linkages between catatonia and eating disorders is needed. Eating disorders, particularly in men, remain underdiagnosed; had the agitated and catatonic episodes not occurred, this patient's eating disorder would likely have remained undetected and become more difficult to treat.10 Early intervention is important.
This patient's continued recovery 9 months later without backslide after initial treatment remains promising. He is no longer prescribed the lorazepam, and he has been on a slow taper of risperidone per patient and family preference and based on his experience in the hospital.
- American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 5th ed. American Psychiatric Publishing; 2013.
- Pelzer AC, van der Heijden FM, den Boer E. Systematic review of catatonia treatment. Neuropsychiatr Dis Treat. 2018;14:317–326. doi:10.2147/NDT.S147897 [CrossRef] PMID:29398916
- Hesslinger B, Walden J, Normann C. Acute and long-term treatment of catatonia with risperidone. Pharmacopsychiatry. 2001;34(1):25–26. doi:10.1055/s-2001-15190 [CrossRef] PMID:11229618
- Grenier E, Ryan M, Ko E, Fajardo K, John V. Risperidone and lorazepam concomitant use in clonazepam refractory catatonia: a case report. J Nerv Ment Dis. 2011;199(12):987–988. doi:10.1097/NMD.0b013e3182392d7e [CrossRef] PMID:22134459
- McAulay C, Hay P, Mond J, Touyz S. Eating disorders, bipolar disorders and other mood disorders: complex and under-researched relationships. J Eat Disord. 2019;7(1):32. doi:10.1186/s40337-019-0262-2 [CrossRef] PMID:31528342
- Seeman MV. Eating disorders and psychosis: seven hypotheses. World J Psychiatry. 2014;4(4):112–119. doi:10.5498/wjp.v4.i4.112 [CrossRef] PMID:25540726
- Wolańczyk T, Komender J, Brzozowska A. Catatonic syndrome preceded by symptoms of anorexia nervosa in a 14-year-old boy with arachnoid cyst. Eur Child Adolesc Psychiatry. 1997;6(3):166–169. doi:10.1007/BF00538989 [CrossRef] PMID:9383651
- Moskowitz AK. “Scared stiff ”: catatonia as an evolutionary-based fear response. Psychol Rev. 2004;111(4):984–1002. doi:10.1037/0033-295X.111.4.984 [CrossRef] PMID:15482070
- Ellul P, Choucha W. Neurobiological approach of catatonia and treatment perspectives. Front Psychiatry. 2015;6(182):182. doi:10.3389/fpsyt.2015.00182 [CrossRef] PMID:26733892
- Strother E, Lemberg R, Stanford SC, Turberville D. Eating disorders in men: underdiagnosed, undertreated, and misunderstood. Eat Disord. 2012;20(5):346–355. doi:10.1080/10640266.2012.715512 [CrossRef] PMID:22985232