Abstract

To The Editor:
F
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/μL; C-reactive protein (CRP) 0.01 mg/dL; serum amyloid A <5 μg/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 of antinuclear antibody, anti-Sjögren'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.
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/μL, 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 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/μL, CRP 0.01 mg/dL, and serum amyloid A <5 μg/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.
JFM, juvenile fibromyalgia.
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 symptoms (Yokota et al. 2013). Cognitive-behavioral therapy (CBT) could be one of the psychosocial interventions for this condition (Sil 2014).
Footnotes
Disclosures
No competing financial interests exist.
