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Type 1 diabetes complicated with uncontrollable adult cyclic vomiting syndrome: a case report
© Ogiso et al. 2015
- Received: 28 April 2015
- Accepted: 17 September 2015
- Published: 23 September 2015
We herein describe the case of a 29-year-old woman with type 1 diabetes from 10 years of age who developed adult cyclic vomiting syndrome. Beginning at 25 years of age, she was frequently hospitalized for stress-induced vomiting. Her vomiting episodes developed acutely and remitted after severe vomiting of more than 30 times a day for a few days. The vomiting periods were accompanied by leukocytosis with a predominance of neutrophils, high blood pressure and fever. In addition, it was noted that her levels of both adrenocorticotropic hormone and antidiuretic hormone during the vomiting attacks increased and subsequently dramatically decreased immediately after symptom improvement; therefore, she was diagnosed with adult-type cyclic vomiting syndrome in accordance with the diagnostic criteria of Rome III, a system developed to classify functional gastrointestinal disorders. Though glycemic control had improved with continuous subcutaneous insulin infusion therapy, the vomiting frequency increased due to the failure of drug treatments and general psychotherapy to terminate the vomiting attacks, making discharge difficult and greatly interfering with everyday life. Eventually, hypnotherapy and miniature garden therapy were prescribed, which significantly reduced the vomiting frequency, making it possible to discharge her from inpatient medical care.
In the treatment of this patient with type 1 diabetes and adult-type cyclic vomiting syndrome, continuous subcutaneous insulin infusion therapy and comprehensive psychotherapy were effective.
- Type 1 diabetes mellitus
- Cyclic vomiting syndrome (CVS)
- Continuous subcutaneous insulin infusion therapy (CSII)
- Sandplay therapy
In type 1 diabetes mellitus, it has been shown that the frequency of complications is reduced by the maintenance of good glycemic control . In addition, it has been reported that patients with type 1 diabetes have more frequent complications with feeding behavior abnormalities and eating disorders [2, 3], as well as a significantly faster progression of complications . It has especially been noted that complications of anorexia nervosa significantly increase mortality .
Continuous subcutaneous insulin infusion therapy (CSII), since being used in many cases registered in the Diabetes Control and Complication Trial (DCCT) , has come to be widely recognized worldwide. CSII was demonstrated to have stronger hypoglycemic action in type 1 diabetes than multiple daily insulin injections (MDI). The insulin requirement was also decreased and was revealed to have a stabilizing effect on blood glucose levels . The improvement with CSII was reported to be especially effective in patients with a hemoglobinA1c (HbA1c) value of more than 8 % . However, in adolescent cases of type 1 diabetes, disease management with CSII is not as effective because glycemic control has also been shown to be unstable due to many other factors . On the other hand, it has been demonstrated through meta-analysis that psychological interventions are associated with improvement in glycemic control . In recent years, approaches that focus on psychological health have been important. Psychotherapies such as hypnotherapy and play therapy were included during traditional medical care. Hypnotherapy was developed based on the research of Erickson, a well-known psychiatrist and psychotherapist in the United States and has also been reported as being efficacious for Parkinson’s disease , migraine , anxiety and depression . Sandplay therapy was developed by Swiss Jungian psychologist D.M. Kalff and has been defined as a psychotherapeutic method that enables patients to arrange miniature figures in a sandtray to create a “sandworld” corresponding to various dimensions of their social reality .
Here, we discuss a patient with type 1 diabetes complicated by ACVS who had repeated vomiting attacks that were especially difficult to control. We report that both CSII and comprehensive psychotherapy, including hypnotherapy and sandplay therapy, were effective.
The patient was a 29-year-old woman who had been in good health until 1995, at which time she was diagnosed at 10 years of age with type 1 diabetes with a positive glutamate decarboxylase (GAD) antibody, and insulin treatment was started. Although her mother had a history of migraines, no history of diabetes in her family was observed.
Because she was bullied in elementary school due to diabetes, she developed overeating behaviors and her blood glucose levels became unstable; frequent hospitalizations were required and became pivotal to her daily life. Unfortunately, the patient continued to experience bullying even in junior high school, and her mother was often absent from home due to work; so the patient began overeating due to loneliness and anxiety. She was then allowed admission to a pediatric long-term care facility where she was also bullied by other patients. In addition, she was repeatedly hospitalized due to high plasma glucose levels. Ultimately, she entered a nursing-high school at the local hospital and graduated from junior high school. Following admission to the high school, to perform education through distance learning, she graduated at 20 years of age. During this period, she was hospitalized for a psychosomatic illness when she was 17 years old. She was transferred from the local hospital to another hospital’s pediatric unit. Furthermore, she began to display signs of retinopathy and overt proteinuria.
In September 2007, at 23 years old, the patient first visited our hospital through referral from the pediatrician at the previous hospital as she had become an adult and glycemic control was becoming increasingly difficult.
In December 2007, the patient, due to vomiting in response to stressful events, was first admitted to our hospital. On physical examination, her height was 158 cm, weight was 56.9 kg, BMI was 22.8 kg/m2, body temperature was 38.4 °C, blood pressure was 146/91 mmHg, and pulse was 117 bpm and regular. Her cardiopulmonary examination was normal. She had no abnormal abdominal findings. Her bilateral Achilles tendon reflexes were absent and bilateral lower extremity vibration sensation was also absent. On ophthalmologic examination, she had diabetic microangiopathies and growth arrest retinopathy. She demonstrated peripheral and autonomic neuropathy and had evidence of diabetic nephropathy of the third stage, i.e., overt proteinuria had been diagnosed, and her estimated glomerular filtration rate was 139.7 mL/min/1.73m2. She had no evidence of chronic thyroiditis (anti-thyroglobulin antibodies 11 IU/mL and anti-thyroid peroxidase antibodies 7 IU/mL; normal range: ≤ 28 IU/mL and ≤ 16 IU/mL, respectively).
Laboratory data for the vomiting attack period and after improvement (April 2009)
Vomiting period (Day 1)
Remission period (Day 6)
Total ketone bodies
Subsequently, around March 2011, the patient’s vomiting episodes worsened and were more prolonged. She had a prolonged period of hospitalization, and her daily life again became difficult. Although her nutrition was managed by nasogastric tube feedings and central venous nutrition, her body weight decreased significantly to 38.0 kg with a body mass index (BMI) of 15.2 kg/m2.
In February 2012, we started hypnotherapy. The treatment was limited by poor language comprehension (Wechsler Adult Intelligence Scale-Third Edition [WEIS-III] score was 60 points), and a developmental disorder was inferred. Even so, the patient was able to be discharged because vomiting frequency was reduced by a combination of hypnotherapy and sandplay therapy (Fig. 2b). Today, her episodes of vomiting have not completely remitted, but it was possible to shift her to outpatient management, and her HbA1c has also been maintained at 7 %.
In our case, violent vomiting began more than 30 times a day without warning, and both nausea and vomiting disappeared within a week. Furthermore, there were more than 3 intermittent vomiting episodes per year, and there was a period of remission of nausea and vomiting between each episode. Migraine history was observed in the patient’s mother. From the above, she was considered to have met all of the diagnostic criteria. However, we also believed it was necessary to exclude celiac disease from the differential diagnosis, although the frequency is very low in Japan.
In this case, vomiting attacks triggered by stress appeared to evolve into a ketotic state with the observation of abnormally high levels of ACTH and ADH, which improved rapidly, along with recovery of vomiting (Table 1). Moreover, the elevation of leukocytes (predominantly neutrophils), hypertension and elevation of body temperature were observed during the vomiting period. We speculated that the increased neutrophil predominance of leukocytes without an increase in C-reactive protein (CRP) and the significant elevation of both blood pressure and fever reflected hypercortisolemia and the enhancement of catecholamine release as well as PGE2 production (catecholamines and PGE2 were not measured), respectively. Sato et al. reported the case of an 8-year-old girl with periodic attacks of vomiting. At the initiation of the attack, ACTH and ADH levels were prominently increased (610 pg/mL and about 82 pg/mL, respectively), followed by hypercortisolemia (51-80 μg/dL) . Nakazato et al. described a 48-year-old woman with several attacks of vomiting. Her abnormal laboratory results included leukocytes (12,500/μL), CRP (0.54 mg/dL) and increased ACTH (196 pg/mL) . Data from the cases shown in these reports were similar to those in our case (Table 1). In association with hormonal variation, vomiting attacks have often been found to overlap with menstruation. Shin et al. reported a case of cyclic vomiting syndrome in an adult patient characterized by stereotypical vomiting attacks occurring during every menstrual period . Therefore, we considered it likely that this patient possessed elements of menstrual-related CVS.
A satisfactory therapeutic method for the treatment of ACVS has not yet been reported. So far, relaxation for stress prevention and medications such as tricyclic antidepressants with CRF inhibitory action, benzodiazepines with an anti-anxiety effect, 5-HT3 receptor antagonists with an antiemetic effect, antihistamines, D2 receptor antagonists and anti-migraine drugs (5-HT10 agonists) have been tried. However, many of them were ineffective in this case. In addition, very little antiemetic effect was observed with the administration of diazepam, metoclopramide and domperidone. Only haloperidol administration was relatively effective, but the mechanism was unknown. On the other hand, appropriate methods of coping with stress were considered to be important in the prevention of the onset of vomiting. Although we worked in conjunction with a clinical psychologist to improve the patient’s coping skills after remission of a vomiting attack, it was not possible to reduce the frequency of vomiting episodes. This is in contrast to the reported efficacy of hypnotherapy in the suppression of nausea and vomiting during cancer chemotherapy  as well as its remarkable antiemetic effect on hyperemesis gravidarum .
We have reported a case of a patient with type 1 diabetes with ACVS. When the characteristic vomiting episodes are seen in patients with type 1 diabetes, it is necessary to take into account the possible overlay of ACVS. A combination of CSII, comprehensive psychotherapy including hypnotherapy and sandplay therapy and a commitment to the patient as a team, including a clinical psychologist, should be considered useful in attaining glycemic control and vomiting control for such patients.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
We wish to thank Prof. Hitoshi Ishii and Prof. Akira Kaito for their help with editing the manuscript and also the patient and her mother for their permission to publish this manuscript. Furthermore, we thank Shannon Hach, MD who provided medical editing services on behalf of Forte, Inc.
Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
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