JOURNAL OF CHILD AND ADOLESCENT PSYCHOPHARMACOLOGY Volume 25, Number x, 2015 ª Mary Ann Liebert, Inc. Pp. 1–3 DOI: 10.1089/cap.2014.0136
Letter to the Editor
Eating Disorder in Juvenile Fibromyalgia Takako Miyamae, MD, PhD,1 Jintaro Isozaki, MD,2 Masako Kikuchi, MD,3 and Shumpei Yokota, MD, PhD 3
To The Editor:
of antinuclear antibody, anti-Sjo¨gren’s-syndrome-related antigen A (anti-SSA/Ro), and rheumatoid factors were negative. We performed infusion therapy mainly comprising glucose and supplementary amino acids, along with correction of an electrolyte imbalance. A powdered soft drink containing glutamine, fiber, and oligosaccharides was introduced to enable the patient to gradually recover oral ingestion. Although she rejected the smell of food at first, she readily accepted liquid food or drugs in tablet form. She came to mix with other pediatric patients while living in the ward and she looked cheerful with resumption of food intake.
ibromyalgia (FM) is a syndrome of unknown etiology, characterized by symptoms of chronic pain, fatigue, and sleep disorders. These major complaints are typically not associated with findings of chronic inflammation or other organic changes, leaving their pathophysiologic mechanisms unclear. The diagnosis is made according to the criteria proposed by the American College of Rheumatology (ACR) in 1990. The core of the globally applied ACR criteria is having tender points in the 18 loci that are pathognomonic of adult FM (Wolfe et al. 1990). On the other hand, primary juvenile fibromyalgia is diagnosed based on Yunus’s criteria (Yunus et al. 1981). The literature on eating disorders associated with this disease is very limited. Although it was reported by a Ministry of Health, Labour and Welfare (MHLW) research team in 2004 that 24.3% of domestic FM cases, including adults, lost body weight (Matsumoto et al. 2006), we reported ‡ 5% body weight loss in 5 of 20 juvenile fibromyalgia ( JFM) cases (Miyamae et al. 2009). Causes of body weight loss were not explored in either report. Arnold et al. analyzed 108 FM cases for psychiatric disorders, and estimated the odds ratio of eating disorders relative to healthy controls as 2.4 (Arnold et al. 2006). Here, we compare our three JFM cases who markedly lost body weight as a result of eating disorders with anorexia nervosa cases from the past.
Case 2 From August 2007, when the patient was 13 years and 4 months of age, her relationships within her extracurricular club were not going well. In March 2008, appetite loss was precipitated by a school club advisor scolding her, resulting in a 10 kg weight loss in 1 month from 38 to 28 kg. In January 2009, when the patient was 15, a diagnosis of JFM was made after several medical institutions had excluded any organic disease through detailed examination. At that time, the patient weighed 64 kg because of a shift to overeating, but she again refused to eat in October, and was referred to our hospital in December of the same year. She had experienced an 8.3 kg weight loss in the preceding 2 months, from 64 to 55.7 kg ( - 12.9%). Her height was 148.5 cm. Blood values indicated no findings of inflammation (WBC 4400/ lL, CRP 0.1 mg/dL). There were no abnormalities in electrolytes, and liver function was normal. Antinuclear antibody, anti-SSA/Ro antibody, and rheumatoid factors were negative. All 18 of the required 18 tender points were positive. The patient preferred wearing loose clothes, such as a jersey, and avoided tight clothing because of general pain. It was surmised that her JFM onset had had a background of domestic violence by her brother, her mother’s dependence on her and inability to separate herself from her, and her father’s ignoring the family as well as his infidelity. The patient had performed poorly at school, scoring low marks, and she was aggressive in communicating with other patients on the ward and was considered unsociable. She was more careful of her toilette than other girls despite wearing a jersey. When she was experiencing peace of mind, she talked endlessly about her circumstances as well as about her future in an attention-seeking, childlike tone of voice. But when the subject was changed to nutritional improvement, she talked nonstop in a suddenly changed tone of voice. In the hospital, compulsory tube feeding was tried, but failed, and the patient’s body weight loss increased because she negotiated cleverly with medical care providers, such as ‘‘I feel tense’’ or ‘‘It’s really
Case Reports Case 1 A 17-year-old girl had begun to have general pains and headaches when she was 16 years and 2 months old, in January 2007, and was diagnosed as having JFM in May of the same year. She had epigastralgia as well as nausea almost simultaneously to the disease onset. She ate less and less because of hypersensitivity to the taste and smell of food, and was hospitalized in November because of a severe body weight reduction of 15 kg over 10 months, from 45.9 to 30.9 kg, a reduction of 32.7%. Her height was 146.1 cm. Tender points were identified at 18 of the 18 specific loci that are pathognomonic for FM. The patient was a serious student and her scholastic record was good enough for her to enter a local college preparatory school. Her disease seemed to have been precipitated by living with her mother and her mother’s lover, as well as by parting from a boyfriend. Blood values were white blood cell count (WBC) 3200/lL; C-reactive protein (CRP) 0.01 mg/dL; serum amyloid A < 5 lg/mL; and Na 124 mEq/L, K 3.0 mEq/L, and Cl 79 mEq/L. There were no abnormalities in liver function, and results 1
Institute of Rheumatology, Tokyo Women’s Medical University, Tokyo, Japan. Department of Pediatric Psychology, Yokohama City University, Yokohama, Japan. Department of Pediatrics, Yokohama City University, Yokohama, Japan.
MIYAMAE ET AL.
tough.’’ She left the ward without permission several times, and was eventually discharged in June 2010. During her 6 months of hospitalization, she never ate a morsel of food. Case 3 In April 2007 (when the she was 14 years and 5 months old), the patient hit her head, triggering general pain, and a diagnosis of JFM was made. She stayed in our department for *4 months shortly after the onset of JFM. After discharge, the number of school days when she was present increased gradually, but her oral intake of food and body weight gradually decreased from August to October 2008, by which time she had lost 8 kg over 2 months, for a decrease in weight from 40 to 32 kg (20%). Her height was 152.0 cm. She was then readmitted to our department. Blood values showed no evidence of inflammation: WBC 5900/lL, CRP 0.01 mg/dL, and serum amyloid A < 5 lg/mL. There were no abnormalities in electrolytes or liver function. Antinuclear antibody titer was positive at 80, whereas tests of anti-SSA/Ro antibody and rheumatoid factors were negative. All 18 of the required 18 tender points were identified. The patient was a perfectionist and extremely strong at making herself known. Her eating disorder seemed to have been provoked by her father’s infidelity and her parents’ divorce. She had been eating only a few beans a day before hospitalization. In the hospital, behavior therapy was initiated to treat the patient’s eating disorder. A diet was started with a half portion of baby food, gradually increased to a full portion, then increased to half a toddler portion, then to a full-size toddler portion, the to a half- size, and finally to a full-size portion of food appropriate for a school-age child. Hospital staff restricted the patient’s personal belongings and her movements. Restriction of her personal belongings was relaxed every time she achieved the diet goal: Eating a determined amount of food for 3 consecutive days. Movement restriction was relaxed when her body weight increased. Initially, the patient resisted our attempt to increase her food intake as well as body weight, but her resistance was gradually weakened and her full body weight was restored. Discussion Here we describe three girls with JFM that fulfilled ACR criteria for FM (Wolfe et al. 1990), who needed hospitalization for management of eating disorders (Table 1). They were all admitted to the pediatric ward for improvement of their nutritional state with the help of the Department of Pediatric Psychiatry.
Specialists in pediatric psychiatry analyzed the three patients as follows. For Case 1, it was inferred that the patient wanted to appear as ‘‘being sick and weak’’ rather than being motivated by the desire for a slim body to appeal to others, and rather than ‘‘being unable to eat.’’ Case 2 was characterized by a very definite obsession with her body weight and shape as evidence by her obstinate refusal to eat and resistance to increased calorie intake. Her tenacious clinging to ‘‘being underweight’’ impressed us as being different from ‘‘pursuing slimming’’ and ‘‘obesity phobia’’ as seen in anorexia nervosa. In Case 3, multiple episodes of self-injurious behavior were observed in addition to JFM-related pain and an eating disorder. These multiple problematic events never occurred simultaneously. This patient’s psychological structure seemed to be the following. There was a central symptom or episode at a certain time, of which the patient complained. Any other symptom/episode was considerably below clinical thresholds; therefore, the patient mentioned it only if specifically asked about it. None of the three patients fulfilled the American Psychiatric Association’s Diagnostic and Statistical Manual of Mental Disorders, 5th ed. (DSM-V) criteria for the diagnosis of anorexia nervosa (American Psychiatric Association 2013). Although they refused oral intake, they lacked the distorted perception of body weight and shape and the extreme fear of an increase of body weight. JFM is diagnosed as a somatoform disorder in the field of psychiatry, and it sometimes needs to be differentiated from depression or a complication of depression (Usui et al. 2007). Our three cases seemed to have in common being diagnosed by psychiatrists as having an eating disorder phenotype and having hysteria. Intriguingly, all of them complained of the general pain specific for FM less frequently during the periods when they actively had eating disorders or were at a markedly low weight. FM is now understood as a syndrome that presents with a variety of symptoms such as chronic fatigue, sleep disorder, and autonomic neuropathy, which is different from widespread pain syndrome. In the preliminary diagnostic criteria for FM proposed by the ACR in 2010, symptoms other than pain are dealt with as ‘‘somatic symptoms’’ (Wolfe et al. 2010). It is open to question whether or not the eating disorders seen in our cases represented ‘‘somatic symptoms.’’ Regardless, optimal management of eating disorders or other ‘‘somatic symptoms’’ needs to be established by both pediatricians and pediatric psychiatrists following accumulated data from more patients. Guidelines for management of JFM have been proposed recently, and they recommended a psychological/ psychiatric approach without the aid of drugs to relieve or cure
Table 1. Characteristics of Three Juvenile Fibromyalgia Patients Who Developed Eating Disorders Characteristics Duration from JFM diagnosis to onset of eating disorder Body weight loss Trigger to onset
Case 1 17 years, female
Case 2 15 years, female
Eating disorder proceeded
- 32.7% Mother’s lover
- 12.9% Trouble in her extracurricular club Elder brother’s domestic violence Mother’s dependence on the patient
- 20.0% Father’s fickleness
Parting from a boyfriend Eating Disorder Attitude against improvement in oral ingestion JFM, juvenile fibromyalgia.
Case 3 14 years, female
Divorce of parents Father’s fickleness and ignoring the family Anorexia and overeating Negotiated cleverly
Anorexia Negotiated cleverly
EATING DISORDER IN JUVENILE FIBROMYALGIA symptoms (Yokota et al. 2013). Cognitive-behavioral therapy (CBT) could be one of the psychosocial interventions for this condition (Sil 2014). Disclosures No competing financial interests exist. References American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders, 5th ed. Washington, DC: American Psychiatric Association; 2013. Arnold LM, Hudson JI, Keck PE, Auchenbach MB, Javaras KN, Hess EV: Comorbidity of fibromyalgia and Psychiatric disorders. J Clin Psychiatry 67:1219–1255, 2006. Matsumoto Y, Maeda S, Tamakoshi A, Nishioka K: Clinical features of fibromyalgia in Japan (from the results a national epidemiologic survey) [in Japanese]. Rinsho Ryumachi 16:87–92, 2006. Miyamae T, Yokota S: Juvenile fibromyalgia in Japan: From a questionnaire for patients/parents and pediatricians [in Japanese]. J Jpn Pediatr Soc 113:503–507, 2009. Sil S, Arnold LM, Lynch–Jordan A, Ting TV, Peugh J, Cunningham N, Powers SW, Lovell DJ, Hashkes PJ, Passo M, Schikler KN, Kashikar-Zuck S: Identifying treatment responders and predictors of improvement after cognitive-behavioral therapy for juvenile fibromyalgia. Pain 155:1206–1212, 2014. Usui C, Arai H. [Names of somatopathic diseases that have a problematic relationship with psychiatric diseases] Fibromyalgia [in Japanese]. Seishinka Chiryogaku 22:755–760, 2007.
3 Wolfe F, Clauw D, Fitzcharles MA, Goldenberg D, Katz RS, Mease P: The American College of Rheumatology preliminary diagnostic criteria for fibromyalgia and measurement of symptom severity. Arthritis Care Res 62:600–610, 2010. Wolfe F, Smythe HA, Yunus MB, Bennett RM, Bombardier C, Goldenberg DL, Tugwell P, Campbell SM, Abeles M, Clark P, Fam AG, Farber SJ, Fiechtner JJ, Franklin CM, Gatter RA, Hamaty D, Lessard J, Lichtbroun AS, Masi AT, Mccain GA, Reynolds WJ, Romano TJ, Russell IJ, Sheon RP: The American College of Rheumatology. 1990 Criteria for the Classification of Fibromyalgia. Report of the Multicenter Criteria Committee. Arthritis Rheum 33:160–72, 1990. Yokota S, Kikuchi M, Miyamae T: Juvenile fibromyalgia: Guidance for management. Pediatrics Int 55:403–409, 2013. Yunus M, Masi AT, Calabro JJ, Miller KA, Feigenbaum SL: Primary fibromyalgia (fibrositis): Clinical study of 50 patients with matched normal controls. Semin Arthritis Rheum 11:151–171, 1981.
Address correspondence to: Takako Miyamae, MD, PhD Institute of Rheumatology Tokyo Women’s Medical University 10-22, Kawada-cho Shinjuku-ku, Tokyo 162-0054 Japan E-mail: [email protected]