Anorexia nervosa in identical triplets.

The cause of anorexia nervosa (AN) is unclear, but is likely multifactorial, including psychological, familial, environmental, societal, genetic, and other biological factors. This case report of identical 12-year-old female triplets simultaneously concordant for AN illustrates the importance of addressing all these components in evaluation and treatment, and the difficulty of determining the relative importance of each factor in the cause of an individual's eating disorder. An overly close relationship and competitiveness between the girls, treated at times as a triplet group rather than as individuals, as well as stressful family dynamics, were probably important antecedents to the girls' AN. The girls encouraged each other and competed to lose weight. Brief individual and family psychotherapy, parent counseling, nutritional counseling, and psychoeducation led to successful treatment. The triplets were encouraged in treatment to compete with and encourage each other to obtain treatment goals, including eating more healthily and achieving healthy weights. A literature review of AN twins studies is also presented, as these studies add to our understanding of the relative importance of shared genes and shared environment in the development of AN. These studies also add insight into treating individuals from families with multiple affected relatives.

Double-blind placebo-controlled administration of fluoxetine in restricting- and restricting-purging-type anorexia nervosa.

BACKGROUND: Anorexia nervosa is an often chronic disorder with high morbidity and mortality. Many people relapse after weight restoration. This study was designed to determine whether a selective serotonin reuptake inhibitor would improve outcome and reduce relapse after weight restoration by contributing to maintenance of a healthy normal weight and a reduction of symptoms. METHODS: We administered a double-blind placebo-controlled trial of fluoxetine to 35 patients with restricting-type anorexia nervosa. Anorexics were randomly assigned to fluoxetine (n = 16) or a placebo (n = 19) after inpatient weight gain and then were observed as outpatients for 1 year. RESULTS: Ten of 16 (63%) subjects remained on fluoxetine for a year, whereas only three of 19 (16%) remained on the placebo for a year (p =.006). Those subjects remaining on fluoxetine for a year had reduced relapse as determined by a significant increase in weight and reduction in symptoms. CONCLUSIONS: This study offers preliminary evidence that fluoxetine may be useful in improving outcome and preventing relapse of patients with anorexia nervosa after weight restoration.

Reactive hypoglycemic coma due to insulin autoimmune syndrome: case report and literature review.

Recurrent episodes of postprandial hypoglycemic symptoms culminated in hypoglycemic coma in a hypertensive but otherwise healthy man while he was taking hydralazine. The patient was found to have an extreme elevation in the immunoreactive insulin level, leading to the discovery of insulin antibodies in the absence of prior exposure to exogenous insulin. Negative results of an anatomic study of the pancreas and an inability to reproduce hypoglycemia during a prolonged fast helped to exclude insulinoma. In contrast, symptomatic hypoglycemia developed in response to oral glucose loading and was associated with an elevation in total and free insulin as well as C-peptide levels. The patient was diagnosed with insulin autoimmune syndrome, which, although a common source of hypoglycemia in Japan, has been well documented in only 15 cases from other countries. HLA typing revealed the patient to be positive for groups Cw4 and DR4, a combination that has been preliminarily associated with insulin autoimmune syndrome in Japan. Unlike the majority of cases previously reported, this patient had no clinical or serologic evidence of an underlying autoimmune disorder and had not been exposed to drugs containing sulfhydryl groups. This case adds to the world literature on insulin autoimmune syndrome, lends support to a postulated HLA association, and documents the presence of insulin autoantibodies in the absence of another underlying autoimmune disorder.

Recurrent hyperparathyroidism from benign neoplastic seeding: a review with recommendations for management.

Benign neoplastic seeding represents a rare cause of hyperparathyroidism found at parathyroid reoperations. It consists of pathologically benign neoplastic parathyroid tissue scattered throughout a previous parathyroid surgical site. We describe a case of recurrent and then persistent hyperparathyroidism occurring in a patient during an 18-year period. The patient's third operation showed nonmalignant parathyroid tissue throughout the right neck bed, which necessitated extensive parathyroid and fibrofatty tissue dissection and a thyroidectomy. This unexpected finding of extensive parathyroid seeding has only been described in detail in ten other patients in the past. A review of these 11 patients shows several features: initial operations were for large parathyroid tumors or cysts in six patients; rupture and spillage of contents occurred in six; and initial hypercalcemia was severe in several patients, perhaps indicating inherently more aggressive tumors. The appearance of benign neoplastic seeding resembled parathyroid carcinoma in its local invasiveness. Therefore the presence of anaplasia, mitoses, capsular invasion, and cellular spindling must be relied on as distinguishing factors. When confronted with this presentation, we recommend removal of all nodal bearing, thyroid, and fibrofatty tissue from the level of the thyroid cartilage to the superior mediastinum and a thyroidectomy if necessary.

Case study: an infection-triggered, autoimmune subtype of anorexia nervosa.

OBJECTIVE: Certain cases of anorexia nervosa (AN) may be similar to the recently described subtype of childhood-onset obsessive-compulsive disorder hypothesized to be one of the pediatric infection-triggered autoimmune neuropsychiatric disorders (PITANDs). METHOD: Three clinical cases are reported. The first patient is a 12-year-old boy whose AN worsened acutely after a group A beta-hemolytic streptococcal (GABHS) infection. His symptoms were alleviated after antibiotic treatment. Two other patients with possible PITANDs-related AN are described. RESULTS: An infection-triggered process may contribute to the pathogenesis of a subtype of AN. CONCLUSIONS: Future research is needed to explore the nature of PITANDs and their relationship with AN.

Interviewing prepubertal children about suicidal ideation and behavior.

OBJECTIVE: Much of the literature on assessment of suicidal children has focused on identifying risk factors associated with suicidal ideation and behavior in this population. Unique problems encountered in interviewing prepubertal children about suicidal ideation and behavior are examined in this paper. METHOD: Observations of problems encountered in interviewing prepubertal children about suicidal ideation and behavior were gleaned in the context of interviews of children admitted to a child psychiatry inpatient unit and interviews of the parents of these children. RESULTS: Unique problems include difficulties in assessment of suicidal intent, impact of cognitive development, particularly of the concept of death, interaction between current emotional state and memory of previous suicidal episodes, characteristics of play associated with suicidal states, effects of parents' attitudes toward assessment on information gathering, and the impact of certain risk factors on cognition and behavior during the interview. CONCLUSION: Interviewing children about suicidal ideation and behavior necessitates that the clinician attend to multiple elements of the interview simultaneously. These interviews are further complicated by the stressful thoughts and feelings that can be raised in both clinician and child in reaction to exploring the child's suicidal ideation and behavior. Additional research is needed to refine the process of reliable interviewing of children about suicidal ideation and behavior and to develop instruments both to quantitate the different elements of these interviews and to guide the clinicians conducting them.

An abused psychotic preadolescent at risk for Huntington's disease.

An abused 10-year-old girl with a family history of Huntington's disease developed incapacitating abdominal pain with concomitant behavioral symptomatology suggestive of dementia. The pseudoneurologic nature of her symptoms was clarified through exhaustive evaluation and did not appear to be that of early-onset Huntington's disease. Assessment included pediatric, psychiatric, neurologic, and gynecologic examination; extensive radiologic and laboratory tests; and chronobiology studies. Successful treatment necessitated the integration of numerous therapeutic modalities including dynamically oriented psychotherapy, psychopharmacologic intervention, physical therapy, behavior modification, and electroconvulsive therapy.

Naltrexone in autistic children: an acute open dose range tolerance trial.

The safety and efficacy of naltrexone was explored in an open acute dose range tolerance trial in 10 hospitalized autistic children, ages 3.42 to 6.50 years (mean, 5.04). Naltrexone was given in ascending doses: 0.5, 1.0, and 2.0 mg/kg/day. Behavioral side effects were observed as early as 1/2 hour after dosing. Ratings on the Children's Psychiatric Rating Scale showed that withdrawal was reduced across all three dose levels; administration of 0.5 mg/kg/day dose resulted in increased verbal production; and the 2.0 mg/kg/day dose resulted in reduction of sterotypies. Mild sedation of brief duration was the only side effect. Electrocardiogram, liver function tests, and all other laboratory studies remained unchanged throughout the study. These preliminary findings require replication in a larger sample of patients under double-blind and placebo controlled condition.

Leuprolide acetate treatment of catamenial pneumothorax.

A 35-year-old nulligravid female with a 20 pack year history of smoking and continuous OC use since age 16 presented with recurrent pneumothoraces coinciding with the onset of menses at age 28. At that time she underwent a right partial pleurectomy and lobectomy, which demonstrated bullous disease but no glandular or stromal elements. Although catamenial respiratory discomfort persisted while on OCs, no pneumothoraces were documented until age 33 at which time she was given the diagnosis of catamenial pneumothorax. A diagnostic laparoscopy failed to demonstrate endometriosis or the presence of diaphragmatic defects. In an effort to preserve her fertility, she began a course of LA-GnRH-a therapy with depot LA. Because of disabling vasomotor and emotional side effects, continuous conjugated estrogens and MPA acetate were given as add-back therapy. She has remained symptom and side effect free for over 2 years on this regimen.

Infection-triggered anorexia nervosa in children: clinical description of four cases.

BACKGROUND: Anorexia nervosa (AN) is a serious illness with no definitive treatment. Clinical and research evidence led to the hypothesis that some children with AN may have a pediatric autoimmune neuropsychiatric disorder associated with streptococcus (PANDAS), similar in pathogenesis to other hypothesized PANDAS disorders. METHODS: Four youngsters (ages, 11-15 years) with PANDAS AN were treated with an open trial of antibiotics, in addition to conventional treatment. They were evaluated for eating disorder and obsessive-compulsive symptoms, and for weight gain. Evidence of streptococcal infection came from clinical evaluation, throat cultures, and two serological tests: anti-deoxyribonuclease B (anti-DNase B) and anti-streptolysin O (ASO) titers. The "rheumatic" marker D8/17 was also measured. This B-cell alloantigen is associated, in several publications, with poststreptococcal autoimmunity: Rheumatic fever (RF), Sydenham's chorea (SC), and possibly PANDAS obsessive compulsive disorder (OCD) and tic disorders. RESULTS: There was clinical evidence of possible antecedent streptococcal infection in all four patients, two of whom had comorbid OCD, with possible infection-triggered AN. All four had the rheumatic marker: A percentage of D8/17-positive B cells of 28-38%, with a mean of 33% (12% or more is considered positive for the marker). The patients responded to conventional treatment plus antibiotics with weight restoration and decreased eating disorder and obsessive-compulsive symptoms. Three needed to gain weight and did so. CONCLUSIONS: There may be a link between infectious disease and some cases of AN, which raises the possibility of new treatment.

Childhood eating disorders.

This paper reviews the recent research literature on childhood eating disorders from a developmental perspective. Although there have been some recent advances in research, much work remains to be done, especially studies specific to child and adolescent eating disorders, as well as definitive follow-up studies. Few data are available on the normal development of eating behavior, or resilience and risk factors for eating pathology. The best-studied areas include epidemiological studies, short-term treatment for bulimia nervosa, and outcome in anorexia nervosa.

A comparison of subgroups of inpatients with anorexia nervosa.

OBJECTIVE: Classification of subgroups of people with anorexia nervosa has been in flux. It has not been clear whether anorexics who only purge should be grouped with pure restricters or with people who both binge and purge. METHODS: We compared 27 restricting-type anorexics (RAN), 26 bulimic anorexics (BAN), and 34 restricting anorexics with purging behaviors (RAN-P). All subjects were underweight and recently admitted to a hospital. We excluded subjects who had not had a diagnosis of anorexia nervosa for at least 1 year duration. RESULTS: The three groups of subjects had similar scores for Depression and Anxiety on the Beck Depression Inventory, the Spielberger State and Trait Anxiety Inventory, and on the EDI subscales aside from higher scores for BAN subjects on the Bulimia subscale. DISCUSSION: These three subgroups of anorexia nervosa have similar degrees of dysphoric moods and core eating disorder symptoms when underweight and malnourished.

Colonic and anorectal function in constipated patients with anorexia nervosa.

OBJECTIVES: Many patients with eating disorders complain of severe constipation. Previous studies have suggested that constipation in patients with anorexia nervosa may be associated with slow colonic transit. However, it is unclear whether a refeeding program will alter colonic transit in these patients. The aim of this study was to investigate colorectal function by measuring colonic transit and anorectal function in anorexic patients with constipation during treatment with a refeeding program. METHODS: We prospectively studied 13 female patients with anorexia nervosa who were admitted to an inpatient treatment unit and compared them to 20 previously studied, age-matched, healthy female control subjects. Patients underwent colonic transit studies using a radiopaque marker technique and anorectal manometry measuring anal sphincter function, rectal sensation, expulsion dynamics, and rectal compliance. Patients were studied both early (< 3 wk) and late (> 3 wk) in their admission. We restudied two patients who had slow colonic transit. All patients also underwent structured interviews. RESULTS: Four of six patients studied within the first 3 wk of their admission had slow colonic transit, defined as > 70 h (108.0 +/- 17.0 h, mean +/- SEM), on initial evaluation. In contrast, none of the seven patients studied later than 3 wk into their admission had slow colonic transit. Two of the four patients with slow transit were restudied later in their admission and were found to have normal transit times. Rectal sensation, internal anal sphincter relaxation threshold, rectal compliance, sphincter pressures, and expulsion pattern were normal in all subjects. CONCLUSIONS: Despite complaints of severe constipation, colonic transit is normal or returns to normal in the majority of patients with anorexia nervosa once they are consuming a balanced weight gain or weight maintenance diet for at least 3 wk.

Tromantadine: inhibitor of early and late events in herpes simplex virus replication.

Unlike amantadine (1-adamantanamine), tromantadine (N-1-adamantyl-N-[2-(dimethyl amino)ethoxy]acetamide hydrochloride) inhibits herpes simplex virus type 1 (KOS strain)-induced cytopathic effect and virus replication with limited toxicity to the cells. Vero and HEp-2 cells tolerated up to 2 mg of tromantadine per 2 X 10(6) cells for 24-, 48-, or 96-h incubation periods with little change in cell morphology. Treatment of the cells with 10 to 50 micrograms of tromantadine reduced herpes simplex virus-induced cytopathic effect. Treatment with 100 to 500 micrograms of tromantadine inhibited herpes simplex virus-induced cytopathic effect and reduced virus production. Complete inhibition of virus production was observed with treatments of 500 micrograms to 1 mg. The antiherpetic activity of tromantadine was dependent upon the viral inoculum size and the time of addition of the compound with respect to infection. Virion synthesis and viral polypeptide synthesis were inhibited by addition of tromantadine at the time of infection or 4 h postinfection. The results are consistent with tromantadine inhibition of an early event in herpes simplex virus infection, before macromolecular synthesis, and a late event, such as assembly or release of virus.

Methylphenidate treatment for bulimia nervosa associated with a cluster B personality disorder.

OBJECTIVES: Psychotherapy and antidepressant medication are helpful to many patients with bulimia nervosa (BN). However, a substantial number of bulimics respond poorly to such treatments. Recent studies suggest that many of the poor responders have cluster B personality disorders. In some ways, the symptomatology of bulimics who have a comorbid cluster B disorder resembles that of patients with attention deficit hyperactivity disorder (ADHD). In particular, individuals in both groups frequently have a high level of impulsivity. Such a resemblance raised the question of whether administration of methylphenidate (MPH), a drug used to treat ADHD, would have therapeutic effects in this subgroup of BN patients. METHODS: In a pilot study, we administered MPH to 2 patients with BN and cluster B traits and found beneficial effects. These patients had not responded to adequate trials of psychotherapy and selective serotonin reuptake inhibitors (SSRIs). RESULTS: MPH treatment was effective. Both Patients had decreased binging and purging. DISCUSSION: MPH may be useful for bulimics with cluster B personality disorder who respond poorly to conventional treatment. Further studies of MPH administration may be worthwhile. Due to the potential risks, however, clinical treatment with this agent is not recommended at this time.

Psychiatric presentations of intracranial cysts.

Eight patients with intracranial cysts presenting with primary psychiatric diagnoses were studied. The cysts were visible on computed tomography (CT) and magnetic resonance imaging (MRI), and they produced neurologic, electroencephalographic (EEG), and neuropsychologic abnormalities. Descriptions of psychiatric manifestations associated with intracranial cysts are rare, and their potential neuropsychiatric significance has been minimized. This study demonstrates that intracranial cysts can cause neuropsychiatric symptoms, that surgical intervention may resolve the psychiatric manifestations in selected cases, and, in cases not warranting surgery, that psychopharmacological therapy to treat the behavioral manifestations is indicated. These cases mandate consideration of intracranial cysts in the diagnostic and therapeutic formulation of atypical neuropsychiatric disorders.