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Al-Beltagi M, Saeed NK, Bediwy AS, Elbeltagi R. Breaking the cycle: Psychological and social dimensions of pediatric functional gastrointestinal disorders. World J Clin Pediatr 2025; 14:103323. [DOI: 10.5409/wjcp.v14.i2.103323] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/14/2024] [Revised: 12/14/2024] [Accepted: 01/02/2025] [Indexed: 03/18/2025] Open
Abstract
BACKGROUND Functional gastrointestinal disorders (FGIDs) in children present with chronic symptoms like abdominal pain, diarrhea, and constipation without identifiable structural abnormalities. These disorders are closely linked to gut-brain axis dysfunction, altered gut microbiota, and psychosocial stress, leading to psychiatric comorbidities such as anxiety, depression, and behavioral issues. Understanding this bidirectional relationship is crucial for developing effective, holistic management strategies that address physical and mental health.
AIM To examine the psychiatric impacts of FGIDs in children, focusing on anxiety and depression and their association with other neurodevelopmental disorders of childhood, such as attention-deficit/hyperactivity disorder, emphasizing the role of the gut-brain axis, emotional dysregulation, and psychosocial stress. Key mechanisms explored include neurotransmitter dysregulation, microbiota imbalance, central sensitization, heightening stress reactivity, emotional dysregulation, and symptom perception. The review also evaluates the role of family dynamics and coping strategies in exacerbating FGID symptoms and contributing to psychiatric conditions.
METHODS A narrative review was conducted using 328 studies sourced from PubMed, Scopus, and Google Scholar, covering research published over the past 20 years. Inclusion criteria focused on studies examining FGID diagnosis, gut-brain mechanisms, psychiatric comorbidities, and psychosocial factors in pediatric populations. FGIDs commonly affecting children, including functional constipation, abdominal pain, irritable bowel syndrome, gastroesophageal reflux, and cyclic vomiting syndrome, were analyzed concerning their psychological impacts.
RESULTS The review highlights a strong connection between FGIDs and psychiatric symptoms, mediated by gut-brain axis dysfunction, dysregulated microbiota, and central sensitization. These physiological disruptions increase children’s vulnerability to anxiety and depression, while psychosocial factors - such as chronic stress, early-life trauma, maladaptive family dynamics, and ineffective coping strategies - intensify the cycle of gastrointestinal and emotional distress.
CONCLUSION Effective management of FGIDs requires a biopsychosocial approach integrating medical, psychological, and dietary interventions. Parental education, early intervention, and multidisciplinary care coordination are critical in mitigating long-term psychological impacts and improving both gastrointestinal and mental health outcomes in children with FGIDs.
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Affiliation(s)
- Mohammed Al-Beltagi
- Department of Paediatrics, Faculty of Medicine, Tanta University, Tanta 31511, Alghrabia, Egypt
- Department of Pediatric, University Medical Center, King Abdulla Medical City, Arabian Gulf University, Manama 26671, Bahrain
| | - Nermin K Saeed
- Medical Microbiology Section, Department of Pathology, Salmaniya Medical Complex, Governmental Hospitals, Manama 26671, Bahrain
- Medical Microbiology Section, Department of Pathology, The Royal College of Surgeons in Ireland - Bahrain, Busaiteen 15503, Muharraq, Bahrain
| | - Adel S Bediwy
- Department of Pulmonology, Faculty of Medicine, Tanta University, Tanta 31527, Alghrabia, Egypt
- Department of Pulmonology, University Medical Center, King Abdulla Medical City, Arabian Gulf University, Manama 26671, Bahrain
| | - Reem Elbeltagi
- Department of Medicine, Royal College of Surgeons in Ireland - Bahrain, Busaiteen 15503, Muharraq, Bahrain
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Bujarska M, Bora G, Li BUK, Banda J, Thomas D, Simpson P, Karrento K. Diagnostic characteristics of pediatric cyclic vomiting syndrome. J Pediatr Gastroenterol Nutr 2025; 80:417-425. [PMID: 39737559 DOI: 10.1002/jpn3.12448] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/03/2024] [Revised: 08/29/2024] [Accepted: 10/11/2024] [Indexed: 01/01/2025]
Abstract
OBJECTIVES Cyclic vomiting syndrome (CVS) remains a diagnostic challenge due to its nonspecific presentation despite consensus-based diagnostic criteria. There is a need for improved, evidence-based diagnostic criteria. We hypothesized that symptoms differ quantitatively between children with CVS versus other vomiting conditions and that current diagnostic criteria are not sufficiently sensitive for diagnosing CVS. METHODS Observational, prospective study of children ages 3-18 years with unexplained vomiting episodes evaluated in the outpatient gastroenterology clinic, emergency department, and inpatient units at Children's Wisconsin. Parents completed symptom surveys at 0, 3, and 6 months. Diagnostic workup and treatment response were monitored by chart review. A final diagnosis (CVS vs. non-CVS) was assigned for group comparisons and receiver operating characteristics (ROC) analysis of diagnostic cutoffs. RESULTS Of 108 subjects enrolled, 46 CVS and 54 non-CVS patients were analyzed. The groups reported overall different episode frequencies with more CVS versus non-CVS (81% vs. 55%) having ≥4 episodes/preceding 12 months (p = 0.013). CVS patients also had longer vomiting episodes (p = 0.03). To distinguish CVS from non-CVS, ROC analyses demonstrated the highest sensitivity for a frequency of 4-10 episodes/12 months (p = 0.002) and a duration threshold of >2 h (p < 0.001). CVS patients reported specific episode characteristics: photophobia (p = 0.003), diaphoresis (p = 0.002), multiple emeses/hour (p = 0.001), stereotypical episodes (p = 0.008), and continued retching after gastric emptying (p = 0.008). Less than half of CVS patients met Rome IV and North American Society for Pediatric Gastroenterology, Hepatology & Nutrition diagnostic criteria. CONCLUSIONS Children with CVS display a distinctive vomiting pattern and clinical features compared to other vomiting conditions. Our findings will help improve current diagnostic criteria.
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Affiliation(s)
- Malgorzata Bujarska
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Geetanjali Bora
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Children's National Hospital, Washington, DC, USA
| | - B U K Li
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Julie Banda
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Danny Thomas
- Division of Emergency Medicine, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Pippa Simpson
- Division of Quantitative Health Sciences, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
| | - Katja Karrento
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA
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Frazier R, Ghoshal UC, Remes-Troche J, Robin D, Shun E, Aravind T. Exploring cyclic vomiting syndrome (CVS) worldwide: Current epidemiological insights and recent developments. Neurogastroenterol Motil 2025; 37:e14932. [PMID: 39370604 DOI: 10.1111/nmo.14932] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/14/2024] [Revised: 08/22/2024] [Accepted: 09/15/2024] [Indexed: 10/08/2024]
Abstract
BACKGROUND Cyclic vomiting syndrome (CVS), a disorder of gut-brain interaction, presents with recurrent episodes of severe nausea and vomiting. It is often associated with missed or delayed diagnoses and substantial healthcare utilization. Despite historical recognition dating back to the 19th century, epidemiological insights remain limited, with research predominantly originating from specific regions, such as the US. CVS prevalence and incidence rates vary widely and are hindered by inconsistent methodologies and disease recognition. PURPOSE This review aims to provide a comprehensive overview of CVS prevalence and incidence rates. It reviews the currently available data and identifies gaps in knowledge. Understanding the global epidemiology of CVS, increasing awareness of the disease, and fostering global collaboration are crucial. Other pertinent issues include disparities in outcomes, particularly among African Americans and Hispanics in the United States, underscoring the need to understand the social determinants of health that drive disease outcomes. This understanding can inform targeted interventions to address these barriers and achieve equitable healthcare both in the United States and globally.
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Affiliation(s)
- Rosita Frazier
- Division of Gastroenterology and Hepatology, Mayo Clinic Arizona, Scottsdale, Arizona, USA
| | - Uday C Ghoshal
- Department of Gastroenterology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
| | - Jose Remes-Troche
- Digestive Physiology and Motility Lab, Veracruzana University, Xalapa, Mexico
| | - Dover Robin
- Cyclical Vomiting Syndrome Association, Letchworth, UK
| | - Emoto Shun
- Department of Family Nursing, Division of Health Sciences and Nursing, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
| | - Thavamani Aravind
- Division of Pediatric Gastroenterology, Hepatology and Nutrition, UH Rainbow Babies and Children's Hospital, Case Western Reserve University, Cleveland, Ohio, USA
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Falsaperla R, Scalia B, Collotta AD, Giacchi V, Cimino C, Ruggieri M. Treatment Options for Cyclic Vomiting Syndrome: A Real-World, Single-Center Experience with Systematic Literature Review and Meta-Analysis. J Clin Pharmacol 2024; 64:227-239. [PMID: 37867371 DOI: 10.1002/jcph.2374] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/27/2023] [Accepted: 10/10/2023] [Indexed: 10/24/2023]
Abstract
The optimal therapeutic management of cyclic vomiting syndrome (CVS) remains elusive. The objective of this study was to document our clinical experience in the Pediatric Department of San Marco Hospital and to survey the literature on pediatric CVS treatment, aiming to update the guidance on the most effective treatment strategies for this not-so-uncommon condition. Data from 70 patients with CVS, admitted to our Pediatric Department between September 2011 and December 2021, were aggregated and included in the study. A systematic review of the literature was conducted in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines. The quality of the included studies was assessed using the Quality Assessment of Diagnostic Accuracy Studies 2 (QUADAS-2) tool and the A Measurement Tool to Assess Systematic Reviews 2 (AMSTAR-2) method. Treatment responses, as observed both in the literature and in our own experience, are variable. In our cohort, topiramate demonstrated superiority over other pharmacological treatments, exhibiting an efficacy of 85% in the patients treated. A universally accepted treatment protocol for pediatric CVS has yet to be established. The efficacy of first-line treatments is generally suboptimal, suggesting that topiramate might serve as a safe and effective primary therapeutic option for pediatric CVS.
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Affiliation(s)
- Raffaele Falsaperla
- Neonatal Intensive Care Unit, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
- Unit of Pediatrics and Pediatric Emergency, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
- Department of Educational Science, Chair of Pediatrics, A.O. Policlinico, "G. Rodolico - San Marco", Catania, Italy
| | - Bruna Scalia
- Neonatal Intensive Care Unit, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
| | - Ausilia Desiree Collotta
- Unit of Pediatrics and Pediatric Emergency, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
| | - Valentina Giacchi
- Neonatal Intensive Care Unit, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
| | - Carla Cimino
- Neonatal Intensive Care Unit, Azienda Ospedaliero Universitaria Policlinico "G. Rodolico - San Marco", San Marco Hospital, Catania, Italy
| | - Martino Ruggieri
- Unit of Rare Diseases of the Nervous System in Childhood, Department of Clinical and Experimental Medicine, Section of Pediatrics and Child Neuropsychiatry, University of Catania, Azienda Ospedaliero Universitaria Policlinico"G. Rodolico - San Marco", Catania, Italy
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Thavamani A, Velayuthan S, Patel D, Al-Hammadi N, Sferra TJ, Sankararaman S. Association of Anxiety and Gastrointestinal Comorbidities in Repeat Hospital Admissions in Pediatric Cyclic Vomiting Syndrome. Am J Gastroenterol 2023; 118:1439-1445. [PMID: 37052354 DOI: 10.14309/ajg.0000000000002292] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/15/2022] [Accepted: 03/28/2023] [Indexed: 04/14/2023]
Abstract
INTRODUCTION Cyclic vomiting syndrome (CVS) is a functional gastrointestinal disorder with recurrent episodes of intense nausea and vomiting and thus may require frequent hospitalizations. There is paucity of data exploring the association of psychiatric and gastrointestinal comorbidities in repeat hospitalizations among pediatric patients with CVS. METHODS We analyzed the Pediatric Health Information System database and included all patients up to 18 years of age with a diagnosis of CVS between 2016 and 2020. We excluded patients with chronic conditions, which mimic CVS. The primary outcome variable was 90-day admission rate, which was defined as a visit to emergency department or admission to observation/inpatient unit with a primary diagnosis of CVS within 90 days after an index CVS hospitalization. RESULTS We evaluated a total of 2,604 hospitalizations represented by 1,370 unique individuals. The overall 90-day admission rate was 28.5%, which steadily decreased from 35.7% in 2016 to 23% in 2019 ( P < 0.001). Patients in the repeat hospitalization cohort were slightly older and more often men. Patients with repeat admissions had an increased proportion of anxiety and other gastrointestinal disorders. Multivariable logistic regression showed that anxiety, gastroesophageal reflux disease, functional dyspepsia, and abdominal migraine were associated with increased odds of repeat admissions. DISCUSSION Ninety-day admission rates in pediatric CVS are decreasing overall, although still contributing to significant healthcare expenditure. Anxiety and gastrointestinal comorbidities were associated with increased risk of repeat admissions. Further prospective studies are needed to better understand the complex interactions of these comorbidities and their management affecting the natural course of CVS.
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Affiliation(s)
- Aravind Thavamani
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, UH Rainbow Babies and Children's Hospital, Case Western Reserve University School of Medicine, Cleveland, Ohio, USA
| | - Sujithra Velayuthan
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, UH Rainbow Babies and Children's Hospital, Case Western Reserve University School of Medicine, Cleveland, Ohio, USA
| | - Dhiren Patel
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, SSM Health Cardinal Glennon Children's Medical Center, Saint Louis University School of Medicine, St. Louis, Missouri, USA
| | - Noor Al-Hammadi
- The Advanced HEAlth Data (AHEAD) Research Institute, Saint Louis University School of Medicine, St. Louis, Missouri, USA
| | - Thomas J Sferra
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, UH Rainbow Babies and Children's Hospital, Case Western Reserve University School of Medicine, Cleveland, Ohio, USA
| | - Senthilkumar Sankararaman
- Division of Pediatric Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, UH Rainbow Babies and Children's Hospital, Case Western Reserve University School of Medicine, Cleveland, Ohio, USA
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Haghighat M, Gholami Shahrebabak M, Dehghani SM, Ataollahi M, Amin Farzaneh N, Hamzeloo Hoseinabadi S, Javaherizadeh H. Relapse Rate of Clinical Symptoms After Stopping Treatment in Children with Cyclic Vomiting Syndrome. Middle East J Dig Dis 2023; 15:32-36. [PMID: 37547164 PMCID: PMC10404072 DOI: 10.34172/mejdd.2023.317] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/10/2022] [Accepted: 11/15/2022] [Indexed: 08/08/2023] Open
Abstract
Background: Cyclic vomiting syndrome (CVS) is a chronic functional gastrointestinal disorder. It is characterized by recurrent episodes of vomiting typically separated by periods of symptom-free or baseline health. The present study aimed at evaluating the effectiveness of propranolol and the relapse rate of clinical symptoms after stopping treatment in children suffering from CVS. Methods: Records of 504 patients below the age of 18 years with CVS who were treated with propranolol from March 2008 to March 2018 were reviewed. The duration of follow-up was 10 years. Results: The average age of CVS affliction was 4.3 years and the average age at the diagnosis was 5.8 years. All subjects were treated with propranolol (for an average of 10 months). 92% of treated subjects were cured, causing a dramatic decrease in the rate of vomiting (P < 0.001). Only an average of 10.5% of the studied subjects (53 people) showed a relapse of symptoms after stopping the treatment. The results of a 10-year follow-up period of the patients showed that 24 had abdominal migraine and 6 had migraine headaches, all of whom lacked the symptoms of disease relapse (prognostic evaluation). Conclusion: The findings of this investigation show that the duration of treating CVS with propranolol could be shortened to 10 months with a low percent of symptoms relapse and this shortening may be effective in preventing the undesirable side effects of the drug. The presence of abdominal migraine and migraine headaches in patients after treatment accomplishment and the lack of disease relapse can be prognostic measures for this disease, which require intensive attention.
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Affiliation(s)
- Mahmoud Haghighat
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Maryam Gholami Shahrebabak
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Seyed Mohsen Dehghani
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Maryam Ataollahi
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Nazanin Amin Farzaneh
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Samaneh Hamzeloo Hoseinabadi
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Hazhir Javaherizadeh
- Clinical Sciences Research Institute, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran
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Frazier R, Venkatesan T. Current understanding of the etiology of cyclic vomiting syndrome and therapeutic strategies in its management. Expert Rev Clin Pharmacol 2022; 15:1305-1316. [PMID: 36259482 DOI: 10.1080/17512433.2022.2138341] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022]
Abstract
INTRODUCTION Cyclic vomiting syndrome is a chronic debilitating disorder of the gut-brain interaction and is characterized by recurrent episodes of nausea and vomiting.Recent studies indicate that it is common and affects 2% of the US population. Unfortunately, there is significant heterogeneity in the management of these patients in the medical community. This review article aims to bridge this gap and will review the epidemiology and etiology with a focus on management of CVS. AREAS COVERED This article reviews the epidemiology, and pathophysiology of CVS and impact on patients. It also discusses management based on recent guidelines based on which is intended for the busy clinician. A literature search was done using PubMed and key words "cyclic vomiting", "management", "etiology", and pathophysiology were used to identify articles of importance. EXPERT OPINION CVS is a complex, poorly understood disorder of gut-brain interaction (DGBI) and has a significant negative impact on patients, families and the healthcare system. Recent guidelines recommend a multidisciplinary approach to management using prophylactic therapy in moderate-severe CVS and abortive medication for acute flares. However more research is needed to better understand the pathophysiology and develop targeted therapies for CVS.
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Affiliation(s)
- Rosita Frazier
- Gastroenterology and Hepatology, Mayo Clinic, Scottsdale, Arizona
| | - Thangam Venkatesan
- Division of Gastroenterology, Hepatology and Nutrition, The Ohio State University, Columbus, OH
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Isoldi S, Di Nardo G, Mallardo S, Parisi P, Raucci U, Tambucci R, Quitadamo P, Salvatore S, Felici E, Cisarò F, Pensabene L, Banzato C, Strisciuglio C, Romano C, Fusco P, Rigotti F, Sansotta N, Caimmi S, Savasta S, Zuin G, Di Stefano M, Provera S, Campanozzi A, Rossi P, Gatti S, Corpino M, Alvisi P, Martelossi S, Suppiej A, Gandullia P, Verrotti A, Terrin G, Pacenza C, Fornaroli F, Comito D, D'Arrigo S, Striano P, Raviglione F, Carotenuto M, Orsini A, Belcastro V, Di Corcia G, Raieli V, Ferilli MAN, Ruscitto C, Spadoni E, Grosso S, D'Alonzo R, Papa A, Pavone P, Meli M, Velardita M, Mainetti M, Vanacore N, Borrelli O. Cyclic vomiting syndrome in children: a nationwide survey of current practice on behalf of the Italian Society of Pediatric Gastroenterology, Hepatology and Nutrition (SIGENP) and Italian Society of Pediatric Neurology (SINP). Ital J Pediatr 2022; 48:156. [PMID: 36042474 PMCID: PMC9429644 DOI: 10.1186/s13052-022-01346-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/16/2022] [Accepted: 08/10/2022] [Indexed: 11/23/2022] Open
Abstract
Background Cyclic Vomiting Syndrome (CVS) is a rare functional gastrointestinal disorder, which has a considerable burden on quality of life of both children and their family. Aim of the study was to evaluate the diagnostic modalities and therapeutic approach to CVS among Italian tertiary care centers and the differences according to subspecialties, as well as to explore whether potential predictive factors associated with either a poor outcome or a response to a specific treatment. Methods Cross-sectional multicenter web-based survey involving members of the Italian Society of Pediatric Gastroenterology, Hepatology and Nutrition (SIGENP) and Italian Society of Pediatric Neurology (SINP). Results A total of 67 responses were received and analyzed. Most of the respondent units cared for less than 20 patients. More than half of the patients were referred after 3 to 5 episodes, and a quarter after 5 attacks. We report different diagnostic approaches among Italian clinicians, which was particularly evident when comparing gastroenterologists and neurologists. Moreover, our survey demonstrated a predilection of certain drugs during emetic phase according to specific clinic, which reflects the cultural background of physicians. Conclusion In conclusion, our survey highlights poor consensus amongst clinicians in our country in the diagnosis and the management of children with CVS, raising the need for a national consensus guideline in order to standardize the practice. Supplementary Information The online version contains supplementary material available at 10.1186/s13052-022-01346-y.
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Affiliation(s)
- Sara Isoldi
- Maternal and Child Health Department, Santa Maria Goretti Hospital, Sapienza-University of Rome, Latina, Italy
| | - Giovanni Di Nardo
- Department of Neuroscience, Mental Health and Sense Organs (NESMOS), Faculty of Medicine & Psychology, Sant'Andrea Hospital, Sapienza University of Rome, 00189, Rome, Lazio, Italy
| | - Saverio Mallardo
- Maternal and Child Health Department, Santa Maria Goretti Hospital, Sapienza-University of Rome, Latina, Italy
| | - Pasquale Parisi
- Department of Neuroscience, Mental Health and Sense Organs (NESMOS), Faculty of Medicine & Psychology, Sant'Andrea Hospital, Sapienza University of Rome, 00189, Rome, Lazio, Italy.
| | - Umberto Raucci
- Pediatric Emergency Department, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Renato Tambucci
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Paolo Quitadamo
- Department of Pediatrics, A.O.R.N. Santobono-Pausilipon, Naples, Italy
| | - Silvia Salvatore
- Pediatric Department, Ospedale "F. Del Ponte", University of Insubria, Varese, Italy
| | - Enrico Felici
- Unit of Pediatrics, The Children Hospital, Azienda Ospedaliera SS Antonio e Biagio e Cesare Arrigo, Alessandria, Italy
| | - Fabio Cisarò
- Pediatric Gastroenterology Unit, Regina Margherita Children's Hospital, Azienda Ospedaliera-Universitaria, Città della Salute e della Scienza di Torino, Turin, Italy
| | - Licia Pensabene
- Pediatric Unit, Department of Medical and Surgical Sciences, University "Magna Graecia" of Catanzaro, Catanzaro, Italy
| | - Claudia Banzato
- Department of Surgical Sciences, Dentistry, Gynecology and Pediatrics, Pediatric Division, University of Verona, Verona, Italy
| | - Caterina Strisciuglio
- Department of Woman, Child, General and Specialistic Surgery, University of Campania "Luigi Vanvitelli", Naples, Italy
| | - Claudio Romano
- Pediatric Gastroenterology Unit, Department of Human Pathology in Adulthood and Childhood "G. Barresi", University of Messina, Messina, Italy
| | - Patrizia Fusco
- Pediatric Unit, Montis Regalis Hospital, Mondovì, Cuneo, Italy
| | | | - Naire Sansotta
- Paediatric Hepatology, Gastroenterology and Transplantation, Hospital Papa Giovanni XXIII, Bergamo, Italy
| | - Silvia Caimmi
- Pediatric Clinic, Foundation IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy
| | - Salvatore Savasta
- Pediatric Clinic, Foundation IRCCS Policlinico San Matteo, University of Pavia, Pavia, Italy
| | - Giovanna Zuin
- Pediatric Department, University of Milano Bicocca, FMBBM, San Gerardo Hospital, Monza, Italy
| | - Marina Di Stefano
- Department of Pediatrics, IRCCS San Raffaele Scientific Institute, Milan, Italy
| | - Silvia Provera
- Department of Pediatrics, IRCCS Sacro Cuore Don Calabria Hospital, Negrar, Verona, Italy
| | - Angelo Campanozzi
- Department of Medical and Surgical Sciences, Pediatric Unit, University of Foggia, Foggia, Italy
| | - Paolo Rossi
- Department of Pediatrics, Pediatric Gastroenterology and Liver Unit, Sapienza University of Rome, Rome, Italy
| | - Simona Gatti
- Department of Pediatrics, Università Politecnica delle Marche, Ancona, Italy
| | - Mara Corpino
- Gastroenterologia Pediatrica, Clinica Pediatrica e Malattie Rare, Ospedale Pediatrico Microcitemico A. Cao, ARNAS Brotzu, Cagliari, Italy
| | - Patrizia Alvisi
- Department of Paediatrics, Ospedale Maggiore, Azienda USL, Bologna, Italy
| | | | - Agnese Suppiej
- Pediatric Section, Department of Medical Sciences, University of Ferrara, Ferrara, Italy
| | - Paolo Gandullia
- UOC Gastroenterologia, IRCCS Istituto Giannina Gaslini, Genoa, Italy
| | | | - Gianluca Terrin
- Department of Maternal and Child Health, Policlinico Umberto I, Sapienza University of Rome, Rome, Italy
| | - Caterina Pacenza
- Department of Pediatrics, San Giovanni di Dio Hospital, Crotone, Italy
| | - Fabiola Fornaroli
- Gastroenterology and Endoscopy Unit, Department of Medicine and Surgery, University of Parma, Parma, Italy
| | | | - Stefano D'Arrigo
- Developmental Neurology Division, Fondazione IRCCS Istituto Neurologico Carlo Besta, Milan, Italy
| | - Pasquale Striano
- Pediatric Neurology Unit, IRCCS Istituto Giannina Gaslini, Genoa, Italy
| | | | - Marco Carotenuto
- Clinic of Child and Adolescent Neuropsychiatry, Department of Mental Health and Physical and Preventive Medicine, Università degli Studi della Campania "Luigi Vanvitelli", Caserta, Italy
| | - Alessandro Orsini
- Paediatric Neurology Section, Paediatric Department, Pisa University Hospital, University of Pisa, Pisa, Italy
| | | | | | - Vincenzo Raieli
- Child Neuropsychiatry Unit - ISMEP- ARNAS CIVICO, Palermo, Italy
| | - Michela Ada Noris Ferilli
- Department of Neuroscience, Paediatric Headache Center, Bambino Gesù Children Hospital, IRCCS, Rome, Italy
| | - Claudia Ruscitto
- Child Neurology and Psychiatry Unit, Systems Medicine Department, Tor Vergata University Hospital of Rome, Rome, Italy
| | - Elisabetta Spadoni
- Pediatric Department, Azienda Ospedaliera Nord-Ovest of Tuscany, San Luca Hospital of Lucca, Lucca, Italy
| | - Salvatore Grosso
- Molecular Medicine and Development, University of Siena, Siena, Italy
| | - Renato D'Alonzo
- Pediatric and Neonatological Unit, Maternal and Child Department, Nuovo Ospedale San Giovanni Battista, Foligno, Italy
| | - Amanda Papa
- Infantile Neuropsychiatry Departement Maggiore della Carità University Hospital, Novara, Italy
| | - Piero Pavone
- Section of Pediatrics and Child Neuropsychiatry, Department of Clinical and Experimental Medicine, University of Catania, Catania, Italy
| | - Mariaclaudia Meli
- Azienda Policlinico, "Rodolico-San Marco Hospital", University of Catania, Catania, Italy
| | - Mario Velardita
- Department of Pediatrics, Hospital "Salvatore e Saverio Gravina", Caltagirone, Italy
| | - Martina Mainetti
- Department of Pediatrics, Santa Maria delle Croci Hospital, Ravenna, Italy
| | - Nicola Vanacore
- National Centre for Epidemiology, Surveillance and Health Promotion, National Institute of Health, Rome, Italy
| | - Osvaldo Borrelli
- Division of Neurogastroenterology and Motility, Department of Pediatric Gastroenterology, University College London (UCL) Institute of Child Health and Great Ormond Street Hospital, London, UK
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Veenin K, Wattanasirichaigoon D, Suktitipat B, Noojarern S, Lertrit P, Tim-Aroon T, Kaewsutthi S, Treepongkaruna S. Association of Mitochondrial DNA Polymorphisms With Pediatric-Onset Cyclic Vomiting Syndrome. Front Pediatr 2022; 10:876436. [PMID: 35685918 PMCID: PMC9170898 DOI: 10.3389/fped.2022.876436] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/15/2022] [Accepted: 04/22/2022] [Indexed: 11/25/2022] Open
Abstract
BACKGROUND Cyclic vomiting syndrome (CVS) is a functional gastrointestinal disorder characterized by recurrent stereotypic episodes of vomiting. The pathophysiology of CVS remains obscure. Previous studies have supported the hypotheses of mitochondrial dysfunction. However, data on association studies between mitochondrial DNA (mtDNA) polymorphisms and pediatric-onset CVS are limited and inconsistent. The aims of this study were to describe clinical characteristics, evaluate association of mtDNA polymorphisms 16519T and 3010A with pediatric-onset CVS and identify new mtDNA candidate variants. METHODS This study involved Thai patients diagnosed with CVS according to the Rome III or IV criteria before the age of 15 years. Patients' demographic data, clinical characteristics, previous investigations and treatment outcomes were obtained. Blood samples were collected for next-generation (whole exome) sequencing, followed by analysis of chromosome M (mitochondrial. Variants were filtered according to clinical significance using ClinVar and MITOMAP. mtDNA polymorphisms in 148 normal Thai individuals were used as controls. RESULTS Forty-eight children were enrolled in the clinical study, and 30 participated in the genetic analysis. The median age at onset and median age at diagnosis was 3.0 (1.5-5.6) and 6.3 (3.0-8.6) years, respectively. Maternal history of migraine was positive in 16.7%. About 45.7% (21 of 46) of the patients achieved complete clinical remission, with the mean symptom duration of 5.9 ± 3.3 years. The prevalence of mtDNA variants 16519T and 3010A among the patient group and Thai general population (control) were as follows: 40.0% (12/30) vs. 27.7% (P = 0.18) and 6.7% (2/30) vs. 0.7% (P = 0.07), respectively. Five known pathogenic variants were identified in 6 patients, including mtDNA 8528C in one patient who also had infantile hypertrophic cardiomyopathy. Six likely pathogenic variants were found but without statistical significance. We identified 11 variants with significant prevalence in the patient group. Though, these variants were classified as variants of unknown significance (VUS), several of them were located in mt functional regions and therefore they deserve further investigations as new candidates for association with pediatric CVS. CONCLUSION There were no associations of mtDNA polymorphisms 16519T and 3010A with CVS in our pediatric cohort. Five pathogenic variants and 11 VUS were found associated with pediatric-onset CVS.
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Affiliation(s)
- Kirana Veenin
- Division of Gastroenterology, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Duangrurdee Wattanasirichaigoon
- Division of Medical Genetics, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Bhoom Suktitipat
- Department of Biochemistry, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand
| | - Saisuda Noojarern
- Division of Medical Genetics, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Patcharee Lertrit
- Department of Biochemistry, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand
| | - Thipwimol Tim-Aroon
- Division of Medical Genetics, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
| | - Supannee Kaewsutthi
- Department of Biochemistry, Faculty of Medicine, Siriraj Hospital, Mahidol University, Bangkok, Thailand
| | - Suporn Treepongkaruna
- Division of Gastroenterology, Department of Pediatrics, Faculty of Medicine Ramathibodi Hospital, Mahidol University, Bangkok, Thailand
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10
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Nivatsi M, Aslanidou I, Mantadakis E. Highly effective use of aprepitant in an adolescent girl with severe cyclic vomiting syndrome. BMJ Case Rep 2021; 14:e241132. [PMID: 33664041 PMCID: PMC7934711 DOI: 10.1136/bcr-2020-241132] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 02/21/2021] [Indexed: 01/16/2023] Open
Abstract
Cyclic vomiting syndrome (CVS) is a debilitating functional gastrointestinal disorder. Diagnosis is based on the Rome III criteria. There are no evidence-based guidelines for the management of paediatric CVS, although ondansetron and antimigraine medications are frequently tried. We describe a 13-year-old adolescent girl with severe CVS and numerous hospital admissions for dehydration because of cyclic vomiting. She had failed oral ondansetron therapy. Oral aprepitant (125 mg in the first, and 85 mg on the second and third days), a neurokinin 1 receptor antagonist that has been approved for preventing chemotherapy-induced vomiting or postoperative emesis, was tried in our patient at home during the first prodromal signs of an upset stomach. She had a dramatic response to it, with no further episodes of vomiting since its start. There is an urgent need for randomised clinical studies to assess the efficacy of available treatment options, including aprepitant in patients with severe CVS.
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Affiliation(s)
- Maria Nivatsi
- Department of Paediatrics, University General Hospital of Alexandroupolis, Alexandroupolis, Thrace, Greece
| | - Ilona Aslanidou
- Department of Paediatrics, University General Hospital of Alexandroupolis, Alexandroupolis, Thrace, Greece
| | - Elpis Mantadakis
- Department of Paediatrics, Democritus University of Thrace Faculty of Medicine, Alexandroupolis, Thrace, Greece
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11
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Kovacic K, Li BUK. Cyclic vomiting syndrome: A narrative review and guide to management. Headache 2021; 61:231-243. [PMID: 33619730 DOI: 10.1111/head.14073] [Citation(s) in RCA: 16] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/18/2020] [Revised: 01/03/2021] [Accepted: 01/06/2021] [Indexed: 12/18/2022]
Abstract
OBJECTIVES/BACKGROUND Cyclic vomiting syndrome (CVS) is a disabling disorder of gut-brain interaction manifested by stereotypical and severe episodes of nausea and vomiting. Prevalence data indicate that CVS affects 1-2% of children and there has been a recent dramatic rise in diagnosed adults. METHODS This narrative review summarizes relevant literature pertaining to pediatric and adult CVS and provides a guide to management based on extensive clinical experience. RESULTS More timely diagnosis is facilitated by an expert consensus diagnostic approach and limited testing. Some diagnostic tests of exclusion remain essential. These include an upper gastrointestinal (GI) contrast study to exclude intestinal malrotation and basic laboratory screening. An abdominal ultrasound is recommended to exclude renal hydronephrosis in children and biliary disease in adults. Exclusion of metabolic/genetic conditions is warranted in those with specific warning signs, presentation in infants/toddler age, and in those with refractory disease. In the absence of chronic GI symptoms, referral to a GI specialist for upper endoscopy is generally not necessary in children but recommended in adults. A large subset termed migraine-equivalent CVS display strong clinical and genetic features of migraine. A unifying pathophysiologic core concept involves neuronal hyperexcitability and aberrant central modulation of autonomic signals. This is coupled with multiple susceptibility factors including mitochondrial dysfunction/cellular energy deficits, a hyper-responsive hypothalamic-pituitary-adrenal axis and many comorbidities that increase vulnerability to triggering events. CVS episodes are frequently triggered by stressors and intercurrent illnesses. Lifestyle and non-pharmacological interventions thus play a pivotal role in successful management. Pharmacological therapies are categorized into abortive, supportive/rescue, and prophylactic treatments. The majority respond particularly well to migraine-focused treatment strategies. CONCLUSION Despite improved characterization and understanding, CVS remains classified as a functional disorder of brain-gut interaction that is often disjointly managed by generalists and subspecialists. Early recognition, evaluation, and management will facilitate care and improve outcomes. Further research into its natural history with common progression to migraine headaches, neuroendocrine mechanisms, and the pathophysiologic relation to migraine diathesis is much needed.
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Affiliation(s)
- Katja Kovacic
- Department of Pediatrics, Division of Gastroenterology, Hepatology and Nutrition, Medical College of Wisconsin, Milwaukee, WI, USA
| | - B U K Li
- Department of Pediatrics, Division of Gastroenterology, Hepatology and Nutrition, Medical College of Wisconsin, Milwaukee, WI, USA
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12
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Raucci U, Borrelli O, Di Nardo G, Tambucci R, Pavone P, Salvatore S, Baldassarre ME, Cordelli DM, Falsaperla R, Felici E, Ferilli MAN, Grosso S, Mallardo S, Martinelli D, Quitadamo P, Pensabene L, Romano C, Savasta S, Spalice A, Strisciuglio C, Suppiej A, Valeriani M, Zenzeri L, Verrotti A, Staiano A, Villa MP, Ruggieri M, Striano P, Parisi P. Cyclic Vomiting Syndrome in Children. Front Neurol 2020; 11:583425. [PMID: 33224097 PMCID: PMC7667239 DOI: 10.3389/fneur.2020.583425] [Citation(s) in RCA: 12] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/14/2020] [Accepted: 09/14/2020] [Indexed: 12/13/2022] Open
Abstract
Cyclic Vomiting Syndrome (CVS) is an underdiagnosed episodic syndrome characterized by frequent hospitalizations, multiple comorbidities, and poor quality of life. It is often misdiagnosed due to the unappreciated pattern of recurrence and lack of confirmatory testing. CVS mainly occurs in pre-school or early school-age, but infants and elderly onset have been also described. The etiopathogenesis is largely unknown, but it is likely to be multifactorial. Recent evidence suggests that aberrant brain-gut pathways, mitochondrial enzymopathies, gastrointestinal motility disorders, calcium channel abnormalities, and hyperactivity of the hypothalamic-pituitary-adrenal axis in response to a triggering environmental stimulus are involved. CVS is characterized by acute, stereotyped and recurrent episodes of intense nausea and incoercible vomiting with predictable periodicity and return to baseline health between episodes. A distinction with other differential diagnoses is a challenge for clinicians. Although extensive and invasive investigations should be avoided, baseline testing toward identifying organic causes is recommended in all children with CVS. The management of CVS requires an individually tailored therapy. Management of acute phase is mainly based on supportive and symptomatic care. Early intervention with abortive agents during the brief prodromal phase can be used to attempt to terminate the attack. During the interictal period, non-pharmacologic measures as lifestyle changes and the use of reassurance and anticipatory guidance seem to be effective as a preventive treatment. The indication for prophylactic pharmacotherapy depends on attack intensity and severity, the impairment of the QoL and if attack treatments are ineffective or cause side effects. When children remain refractory to acute or prophylactic treatment, or the episode differs from previous ones, the clinician should consider the possibility of an underlying disease and further mono- or combination therapy and psychotherapy can be guided by accompanying comorbidities and specific sub-phenotype. This review was developed by a joint task force of the Italian Society of Pediatric Gastroenterology Hepatology and Nutrition (SIGENP) and Italian Society of Pediatric Neurology (SINP) to identify relevant current issues and to propose future research directions on pediatric CVS.
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Affiliation(s)
- Umberto Raucci
- Pediatric Emergency Department, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Osvaldo Borrelli
- Division of Neurogastroenterology and Motility, Department of Pediatric Gastroenterology, University College London (UCL) Institute of Child Health and Great Ormond Street Hospital, London, United Kingdom
| | - Giovanni Di Nardo
- Chair of Pediatrics, Department of Neuroscience, Mental Health and Sense Organs (NESMOS), Faculty of Medicine & Psychology, Sant'Andrea Hospital, Sapienza University of Rome, Rome, Italy
| | - Renato Tambucci
- Digestive Endoscopy and Surgery Unit, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Piero Pavone
- Section of Pediatrics and Child Neuropsychiatry, Department of Clinical and Experimental Medicine, University of Catania, Catania, Italy
| | - Silvia Salvatore
- Pediatric Department, Ospedale “F. Del Ponte,” University of Insubria, Varese, Italy
| | | | | | - Raffaele Falsaperla
- Neonatal Intensive Care and Pediatric Units, S. Marco Hospital, Vittorio Emanuele Hospital, Catania, Italy
| | - Enrico Felici
- Unit of Pediatrics, The Children Hospital, Azienda Ospedaliera SS Antonio e Biagio e Cesare Arrigo, Alessandria, Italy
| | - Michela Ada Noris Ferilli
- Division of Neurology, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Salvatore Grosso
- Clinical Pediatrics, Department of Molecular Medicine and Development, University of Siena, Siena, Italy
| | - Saverio Mallardo
- Pediatric Department, Santa Maria Goretti Hospital, Sapienza University of Rome, Latina, Italy
| | - Diego Martinelli
- Division of Metabolism, Department of Pediatric Specialties, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Paolo Quitadamo
- Department of Pediatrics, A.O.R.N. Santobono-Pausilipon, Naples, Italy
| | - Licia Pensabene
- Pediatric Unit, Department of Medical and Surgical Sciences, University “Magna Graecia” of Catanzaro, Catanzaro, Italy
| | - Claudio Romano
- Pediatric Gastroenterology Unit, Department of Human Pathology in Adulthood and Childhood “G. Barresi”, University of Messina, Messina, Italy
| | | | - Alberto Spalice
- Child Neurology Division, Department of Pediatrics, “Sapienza,” University of Rome, Rome, Italy
| | - Caterina Strisciuglio
- Department of Woman, Child, General and Specialistic Surgery, University of Campania “Luigi Vanvitelli,” Naples, Italy
| | - Agnese Suppiej
- Pediatric Section, Department of Medical Sciences, University of Ferrara, Ferrara, Italy
| | - Massimiliano Valeriani
- Division of Neurology, Bambino Gesù Children's Hospital, Institute for Research, Hospitalization and Health Care (IRCCS), Rome, Italy
| | - Letizia Zenzeri
- Emergency Pediatric Department, Santobono-Pausilipon Children's Hospital, Naples, Italy
| | - Alberto Verrotti
- Department of Pediatrics, University of L'Aquila, L'Aquila, Italy
| | - Annamaria Staiano
- Section of Pediatrics, Department of Translational Medical Science, “Federico II” University of Naples, Naples, Italy
| | - Maria Pia Villa
- Chair of Pediatrics, Department of Neuroscience, Mental Health and Sense Organs (NESMOS), Faculty of Medicine & Psychology, Sant'Andrea Hospital, Sapienza University of Rome, Rome, Italy
| | - Martino Ruggieri
- Unit of Rare Diseases of the Nervous System in Childhood, Section of Pediatrics and Child Neuropsychiatry, Department of Clinical and Experimental Medicine, University of Catania, Catania, Italy
| | - Pasquale Striano
- Department of Neurosciences, Rehabilitation, Ophthalmology, Genetics, Maternal and Child Health, University of Genoa, Genova, Italy
- Institute for Research, Hospitalization and Health Care (IRCCS) “G. Gaslini” Institute, Genova, Italy
| | - Pasquale Parisi
- Chair of Pediatrics, Department of Neuroscience, Mental Health and Sense Organs (NESMOS), Faculty of Medicine & Psychology, Sant'Andrea Hospital, Sapienza University of Rome, Rome, Italy
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13
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Sikand M, Sharma P. Psychological intervention in cyclic vomiting syndrome in adolescents: A case series. J Child Adolesc Ment Health 2020; 31:182-188. [PMID: 31805841 DOI: 10.2989/17280583.2019.1674660] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Abstract
Objective: Cyclic vomiting syndrome (CVS) is difficult to diagnose, thus there is often a delay in diagnosis or a misdiagnosis. In the absence of an adequate understanding of the pathophysiology of the syndrome, it is under-recognised and treatment is difficult. The present case series aimed to assess and manage three adolescents with CVS.Method: The Children's Apperception Test was administered on the three Asian adolescents who were referred for the management of CVS to the Department of Clinical Psychology at a tertiary care hospital in New Delhi, India. A treatment module was developed to treat CVS in these adolescents.Results: A strong link was found between the psychological stressors and their physical manifestations in the episodes of vomiting. Therapeutic management with a focus on behavioural modification, adaptive coping skills, and a healthy therapeutic relationship was found to be efficacious in gradually remitting this condition.Conclusions: Thus, the focus of treatment in cases of CVS should be to understand the psychological underpinning and help the adolescents to incorporate healthy coping strategies.
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Affiliation(s)
- Mehak Sikand
- Department of Clinical Psychology, National Institute of Mental Health and Neurosciences, Bengaluru, India
| | - Pragya Sharma
- Department of Clinical Psychology, Dr Ram Manohar Lohia Hospital, New Delhi, India
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14
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McAbee GN, Morse AM, Cook W, Tang V, Brosgol Y. Neurological Etiologies and Pathophysiology of Cyclic Vomiting Syndrome. Pediatr Neurol 2020; 106:4-9. [PMID: 32107138 DOI: 10.1016/j.pediatrneurol.2019.12.001] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/23/2019] [Revised: 12/05/2019] [Accepted: 12/06/2019] [Indexed: 12/28/2022]
Abstract
Cyclic vomiting syndrome is an idiopathic chronic periodic disorder of childhood which may persist into the adult years. Although cyclic vomiting syndrome is considered a central nervous system disorder, it is often managed by a pediatric gastroenterologist. The practitioner should not assume a gastrointestinal or non-neurological cause of symptoms especially if there are coexisting neurological symptoms and signs or if vomiting does not bring relief; this suggests a possible central nervous system cause, which may necessitate a pediatric neurology consultation. Examples of central nervous system causes of cyclic vomiting syndrome that can have subjective and objective neurological findings include abdominal migraine, certain types of epilepsy, structural lesions (tumors, Chiari malformation, demyelinating disease), mitochondrial disease, autonomic disorders, fatty acid/organic acid disorders, urea cycle defects, and cannabinoid hyperemesis syndrome. Improved familiarity with cyclic vomiting syndrome and its mimics may improve the time to appropriate diagnosis and may reduce morbidity related to cyclic vomiting syndrome.
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Affiliation(s)
- Gary N McAbee
- Division of Child Neurology, Department of Pediatrics, Maimonides Children's Hospital, Brooklyn, New York.
| | - Anne Marie Morse
- Division of Child Neurology, Department of Pediatrics, Janet Weis Children's Hospital, Geisinger Medical Center, Danville, Pennsylvania
| | - Ward Cook
- Division of Child Neurology, Department of Pediatrics, Janet Weis Children's Hospital, Geisinger Medical Center, Danville, Pennsylvania
| | - Vivian Tang
- Division of Pediatric Gastroenterology, Department of Pediatrics, Maimonides Children's Hospital, Brooklyn, New York
| | - Yuri Brosgol
- Division of Child Neurology, Department of Pediatrics, Maimonides Children's Hospital, Brooklyn, New York
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15
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Redon S, Donnet A. Dramatic Response to Nebivolol in an Adult Case of Cyclic Vomiting Syndrome. Headache 2019; 59:1827-1828. [DOI: 10.1111/head.13619] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 07/18/2019] [Indexed: 12/01/2022]
Affiliation(s)
- Sylvain Redon
- Department of Evaluation and Treatment of Pain, FHU INOVPAIN CHU Timone, AP‐HM Marseille France
| | - Anne Donnet
- Department of Evaluation and Treatment of Pain, FHU INOVPAIN CHU Timone, AP‐HM Marseille France
- INSERM U‐1107 CHU de Clermont‐Ferrand Clermont‐Ferrand France
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16
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Sharaf RN, Venkatesan T, Shah R, Levinthal DJ, Tarbell SE, Jaradeh SS, Hasler WL, Issenman RM, Adams KA, Sarosiek I, Stave CD, Li BUK, Sultan S. Management of cyclic vomiting syndrome in adults: Evidence review. Neurogastroenterol Motil 2019; 31 Suppl 2:e13605. [PMID: 31241818 PMCID: PMC6899798 DOI: 10.1111/nmo.13605] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/25/2022]
Abstract
BACKGROUND This evidence review was conducted to inform the accompanying clinical practice guideline on the management of cyclic vomiting syndrome (CVS) in adults. METHODS We followed the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) framework and focused on interventions aimed at prophylactic management and abortive treatment of adults with CVS. Specifically, this evidence review addresses the following clinical questions: (a) Should the following pharmacologic agents be used for prophylaxis of CVS: amitriptyline, topiramate, aprepitant, zonisamide/levetiracetam, or mitochondrial supplements? (b) Should the following pharmacologic agents be used for abortive treatment: triptans or aprepitant? RESULTS We found very low-quality evidence to support the use of the following agents for prophylactic and abortive treatment of CVS: amitriptyline, topiramate, aprepitant, zonisamide/levetiracetam, and mitochondrial supplements. We have moderate certainty of evidence for the use of triptans as abortive therapy. We found limited evidence to support the use of ondansetron and the treatment of co-morbid conditions and complementary therapies. CONCLUSIONS This evidence review helps inform the accompanying guideline for the management of adults with CVS which is aimed at helping clinicians, patients, and policymakers, and should improve patient outcomes.
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Affiliation(s)
- Ravi N. Sharaf
- Division of GastroenterologyDepartment of MedicineWeill Cornell Medical CenterNew YorkNew York
| | - Thangam Venkatesan
- Division of GastroenterologyDepartment of MedicineMedical College of WisconsinMilwaukeeWisconsin
| | - Raj Shah
- Division of Gastroenterology and Liver DiseaseDepartment of Internal MedicineCase Western Reserve University School of MedicineClevelandOhio
| | - David J. Levinthal
- Department of MedicineDivision of Gastroenterology, Hepatology, and NutritionUniversity of Pittsburgh Medical CenterPittsburgPennsylvania
| | - Sally E. Tarbell
- Department of Psychiatry and Behavioral SciencesNorthwestern Feinberg University School of MedicineChicagoIllinois
| | - Safwan S. Jaradeh
- Department of Neurology and Neurological SciencesStanford University Medical CenterStanfordCalifornia
| | - William L. Hasler
- Division of GastroenterologyDepartment of MedicineUniversity of Michigan Medical SchoolAnn ArborMichigan
| | - Robert M. Issenman
- Division of Pediatric GastroenterologyDepartment of PediatricsMcMaster UniversityHamiltonOntario
| | | | - Irene Sarosiek
- Division of GastroenterologyDepartment of Internal MedicineTexas Tech University Health Sciences CenterLubbockTexas
| | | | - B U. K. Li
- Division of Pediatric Gastroenterology, Hepatology and NutritionDepartment of PediatricsMedical College of WisconsinMilwaukeeWisconsin
| | - Shahnaz Sultan
- Division of GastroenterologyDepartment of MedicineUniversity of MinnesotaMinneapolis Veterans Affairs Healthcare SystemMinneapolisMinnesota
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17
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Venkatesan T, Levinthal DJ, Tarbell SE, Jaradeh SS, Hasler WL, Issenman RM, Adams KA, Sarosiek I, Stave CD, Sharaf RN, Sultan S, Li BUK. Guidelines on management of cyclic vomiting syndrome in adults by the American Neurogastroenterology and Motility Society and the Cyclic Vomiting Syndrome Association. Neurogastroenterol Motil 2019; 31 Suppl 2:e13604. [PMID: 31241819 PMCID: PMC6899751 DOI: 10.1111/nmo.13604] [Citation(s) in RCA: 33] [Impact Index Per Article: 5.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/06/2019] [Revised: 03/17/2019] [Accepted: 03/18/2019] [Indexed: 12/15/2022]
Abstract
The increasing recognition of cyclic vomiting syndrome (CVS) in adults prompted the development of these evidence-based guidelines on the management of CVS in adults, which was sponsored by the American Neurogastroenterology and Motility Society (ANMS) and the Cyclic Vomiting Syndrome Association (CVSA). GRADE (Grading of Recommendations, Assessment, Development, and Evaluation) framework was used and a professional librarian performed the literature search. The expert committee included the President of the CVSA who brought a patient perspective into the deliberations. The committee makes recommendations for the prophylaxis of CVS, treatment of acute attacks, diagnosis, and overall management of CVS. The committee strongly recommends that adults with moderate-to-severe CVS receive a tricyclic antidepressant (TCA), such as amitriptyline, as a first-line prophylactic medication and receive topiramate or aprepitant as alternate prophylactic medications. Zonisamide or levetiracetam and mitochondrial supplements (Coenzyme Q10, L-carnitine, and riboflavin) are conditionally recommended as alternate prophylactic medications, either alone or concurrently with other prophylactic medications. For acute attacks, the committee conditionally recommends using serotonin antagonists, such as ondansetron, and/or triptans, such as sumatriptan or aprepitant to abort symptoms. Emergency department treatment is best achieved with the use of an individualized treatment protocol and shared with the care team (example provided). The committee recommended screening and treatment for comorbid conditions such as anxiety, depression, migraine headache, autonomic dysfunction, sleep disorders, and substance use with referral to appropriate allied health services as indicated. Techniques like meditation, relaxation, and biofeedback may be offered as complementary therapy to improve overall well-being and patient care outcomes.
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Affiliation(s)
- Thangam Venkatesan
- Division of Gastroenterology and HepatologyMedical College of WisconsinMilwaukeeWisconsin
| | - David J. Levinthal
- Division of Gastroenterology, Hepatology and NutritionUniversity of Pittsburgh Medical CenterPittsburghPennsylvania
| | - Sally E. Tarbell
- Department of Psychiatry and Behavioral SciencesNorthwestern Feinberg School of MedicineChicagoIllinois
| | | | | | - Robert M. Issenman
- Division of Pediatric GastroenterologyMcMaster UniversityHamiltonOntarioCanada
| | | | - Irene Sarosiek
- Division of GastroenterologyTexas Tech University Health Sciences CenterEl PasoTexas
| | | | - Ravi N. Sharaf
- Division of GastroenterologyDepartment of Healthcare Policy and ResearchWeill Cornell Medical CenterNew YorkNew York
| | | | - B U. K. Li
- Division of GastroenterologyDepartment of PediatricsMedical College of WisconsinMilwaukeeWisconsin
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BAGHERIAN Z, YAGHINI O, SANEIAN H, BADIHIAN S. Comparison of the Efficacy of Amitriptyline and Topiramate in Prophylaxis of Cyclic Vomiting Syndrome. IRANIAN JOURNAL OF CHILD NEUROLOGY 2019; 13:37-44. [PMID: 30613204 PMCID: PMC6296701] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Subscribe] [Scholar Register] [Received: 10/10/2016] [Revised: 12/16/2017] [Accepted: 12/27/2017] [Indexed: 11/05/2022]
Abstract
OBJECTIVES Cyclic vomiting syndrome (CVS) is a chronic functional gastrointestinal disorder with no certain treatment. We aimed to compare the efficacy of amitriptyline and topiramate on prophylactic therapy of CVS. MATERIALS & METHODS This randomized clinical trial (registration number: IRCT2015102316844N2) was conducted during 2016 in Isfahan, central Iran. The inclusion criteria were CVS patients (based on Rome III) aging 3-15 yr with normal physical examination, no metabolic disorder, and no gastrointestinal obstruction or renal impairment. Recruited patients were divided into two groups of amitriptyline (1 mg/kg/d) and topiramate (1-2 mg/kg/d) and were followed for 3-months. The outcome was evaluated by comparing severity of attacks (monthly frequency and duration of attacks) before and after intervention. RESULTS Thirty-six children entered each group and two patients left the amitriptyline group. Patients and disease characteristics were similar between groups before intervention (P>0.05). The frequency of attacks (standard deviation) after intervention in amitriptyline and topiramate group was 0.91 (0.40) and 1.07 (0.55), respectively (P=0.368) and the duration of attacks (SD) after intervention were 3.43 (2.46) and 4.90 (3.03), respectively (P=0.017). Twenty-three patients (68%) in amitriptyline group and 14 patients (39%) in topiramate group stopped having attacks after intervention (P=0.016). CONCLUSION Amitriptyline is a better choice to reduce severity of CVS attacks compared to topiramate, in a short-term evaluation. Studies with longer follow-up are required to investigate these findings in a longer period.
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Affiliation(s)
- Zahra BAGHERIAN
- Child Growth and Development Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Omid YAGHINI
- Child Growth and Development Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Hossein SANEIAN
- Child Growth and Development Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Shervin BADIHIAN
- Child Growth and Development Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
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Kovacic K, Sood M, Venkatesan T. Cyclic Vomiting Syndrome in Children and Adults: What Is New in 2018? Curr Gastroenterol Rep 2018; 20:46. [PMID: 30159612 DOI: 10.1007/s11894-018-0654-5] [Citation(s) in RCA: 23] [Impact Index Per Article: 3.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/08/2023]
Abstract
PURPOSE OF REVIEW Cyclic vomiting syndrome (CVS) is a disabling functional gastrointestinal disorder characterized by severe vomiting episodes that alternate with symptom-free periods. The purpose of this review is to summarize current knowledge and highlight most recent data on prevalence, diagnosis, management, and impact of CVS in children and adults. RECENT FINDINGS Originally thought to be a pediatric disorder, the past decade has witnessed a considerable increase in CVS diagnosed in adults. Despite improved recognition of CVS, without a delineated pathophysiology and specific biomarker, it remains classified as a functional gastrointestinal disorder. Migraines and CVS share a common pathway based on several studies and response to migraine therapy. Recent work has begun to expand the list of comorbidities and identify plausible mechanisms and new therapeutic avenues. This review seeks to highlight best practices and novel therapies for CVS based on expert consensus and review of available literature.
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Affiliation(s)
- Katja Kovacic
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Children's Hospital of Wisconsin, Medical College of Wisconsin, 9000 W. Wisconsin Avenue, Milwaukee, WI, 53226, USA.
| | - Manu Sood
- Division of Gastroenterology, Hepatology and Nutrition, Department of Pediatrics, Children's Hospital of Wisconsin, Medical College of Wisconsin, 9000 W. Wisconsin Avenue, Milwaukee, WI, 53226, USA
| | - Thangam Venkatesan
- Division of Gastroenterology and Hepatology, Medical College of Wisconsin, Milwaukee, WI, USA
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Ye Z, Xue A, Huang Y, Wu Q. Children with cyclic vomiting syndrome: phenotypes, disease burden and mitochondrial DNA analysis. BMC Gastroenterol 2018; 18:104. [PMID: 29969994 PMCID: PMC6029397 DOI: 10.1186/s12876-018-0836-5] [Citation(s) in RCA: 11] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/04/2017] [Accepted: 06/26/2018] [Indexed: 12/15/2022] Open
Abstract
BACKGROUND Cyclic vomiting syndrome (CVS) is characterized by repeated, stereotypical vomiting episodes. It is possibly associated with mitochondrial DNA (mtDNA) variants. We examined the phenotype, disease burden, treatment and performed mtDNA analysis in pediatric CVS. METHODS This retrospective study included 42 children with CVS in a tertiary care center. Information regarding medical history, clinical features, laboratory tests, and treatment were collected. mtDNA sequencing was performed among 13 patients. RESULTS Mean age of onset among patients was 4.0±3.4 years, and mean age at diagnosis was 6.7±4.2 years. CVS episodes in onset and features were stereotypic. Recognizable prodromes were reported in 54.8% patients. Neuroimaging showed previously unknown intracranial abnormalities. Gastrointestinal infection was found in four patients. Mean duration of hospitalization was 7.0±2.4 days, and mean hospitalization cost was 10,891 RMB. Sequencing showed that 4/13 patients had C16519T mtDNA polymorphism, and 2/13 patients had G3010A mtDNA polymorphism. CONCLUSIONS Cyclic vomiting syndrome is a disabling disorder, which causes huge disease burdens to the patients and their families. Early clinical suspicion and prompt diagnosis are crucial. mtDNA polymorphisms were found in some patients, but they were not significantly associated with pediatric CVS.
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Affiliation(s)
- Ziqing Ye
- Department of Gastroenterology, Children’s Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102 China
| | - Aijuan Xue
- Department of Gastroenterology, Children’s Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102 China
| | - Ying Huang
- Department of Gastroenterology, Children’s Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102 China
| | - Qiye Wu
- Department of Gastroenterology, Children’s Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102 China
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Hayes WJ, VanGilder D, Berendse J, Lemon MD, Kappes JA. Cyclic vomiting syndrome: diagnostic approach and current management strategies. Clin Exp Gastroenterol 2018; 11:77-84. [PMID: 29520160 PMCID: PMC5833754 DOI: 10.2147/ceg.s136420] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/26/2022] Open
Abstract
Cyclic vomiting syndrome (CVS) is a disorder characterized by episodes of nausea and vomiting lasting for 1-5 days followed by asymptomatic periods. The etiology of CVS is unknown, but it shares similar characteristics to migraine headaches. CVS is generally classified as having four phases: prodromal, acute/vomiting/hyperemesis, recovery, and remission/interepisodic. Current management strategies include trigger avoidance, abortive and prophylactic medication therapies, and supportive care. The goal of therapy for the remission phase is prophylaxis of further episodes. Antidepressant, antiepileptic, and antimigraine medications show an overall reduction or remission of CVS symptoms in more than 70% of patients. This article provides a summary of diagnostic strategies and reviews current management strategies for CVS.
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Affiliation(s)
- William J Hayes
- Department of Pharmacy Practice, South Dakota State University, Regional Health Medical Clinic, Rapid City, SD, USA
| | - Deidra VanGilder
- South Dakota State University, College of Pharmacy and Allied Health Professions, Brown Clinic, Watertown, SD, USA
| | - Joseph Berendse
- Department of Pharmacy Practice, South Dakota State University, College of Pharmacy and Allied Health Professions, VA Black Hills Health Care System, Fort Meade, SD, USA
| | - Michael D Lemon
- Department of Pharmacy Practice, South Dakota State University, College of Pharmacy and Allied Health Professions, VA Black Hills Health Care System, Fort Meade, SD, USA
| | - John A Kappes
- Department of Pharmacy Practice, South Dakota State University, College of Pharmacy and Allied Health Professions, Rapid City Regional Hospital, Rapid City, SD, USA
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Badihian N, Saneian H, Badihian S, Yaghini O. Prophylactic Therapy of Cyclic Vomiting Syndrome in Children: Comparison of Amitriptyline and Cyproheptadine: A Randomized Clinical Trial. Am J Gastroenterol 2018; 113:135-140. [PMID: 28719594 DOI: 10.1038/ajg.2017.194] [Citation(s) in RCA: 17] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/05/2016] [Accepted: 05/21/2017] [Indexed: 12/11/2022]
Abstract
OBJECTIVES Cyclic vomiting syndrome (CVS) is a common functional gastrointestinal disorder characterized by recurrent episodes of nausea and vomiting. There is no definite treatment for the condition, although some medications are recommended. We aimed to compare the efficacy of amitriptyline and cyproheptadine in prophylactic therapy of CVS. METHODS This is a single-blinded randomized clinical trial conducted during 2015-2016 in Isfahan, Iran. Sixty-four children who were 3-15 years old, with a diagnosis of CVS (based on Rome III criteria), were included in the study and were randomly divided into two groups of amitriptyline and cyproheptadine. They were followed for 6 months, looking for frequency and duration of attacks as the primary outcome. RESULTS The mean monthly frequency of attacks in the last 2 months of the study in the amitriptyline and cyproheptadine group were 0.38±0.55 and 0.59±0.71, respectively (P-value=0.197), after intervention. The mean duration of attacks between amitriptyline and cyproheptadine group were 1.41±2.86 and 1.81±2.22 h, respectively (P-value=0.212). In the amitriptyline group 65.6% of patients reported 100% remission, whereas in the cyproheptadine group 50% reported full remission (P-value=0.206). CONCLUSIONS There was no superiority of one of the medications over the other. We did not find any age-related effect on the efficacy of these medications as well.
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Affiliation(s)
- Negin Badihian
- Faculty of Medicine, Najafabad Branch, Islamic Azad University, Najafabad, Iran
| | - Hossein Saneian
- Child Growth and Development Research Center, Research Institute for Primordial Prevention of Non-Communicable Disease, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Shervin Badihian
- Child Growth and Development Research Center, Research Institute for Primordial Prevention of Non-Communicable Disease, Isfahan University of Medical Sciences, Isfahan, Iran.,Students' Research Center, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
| | - Omid Yaghini
- Child Growth and Development Research Center, Research Institute for Primordial Prevention of Non-Communicable Disease, Isfahan University of Medical Sciences, Isfahan, Iran
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Haghighat M, Memari H, Honar N, Dehghani SM, Imanieh MH, Injoo SJ, Javaherizadeh H. The efficacy and duration of treatment with propranolol in children with cyclic vomiting syndrome in southern Iran. PRZEGLAD GASTROENTEROLOGICZNY 2017; 12:291-295. [PMID: 29358999 PMCID: PMC5771454 DOI: 10.5114/pg.2017.72105] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 09/05/2016] [Accepted: 11/18/2016] [Indexed: 02/05/2023]
Abstract
INTRODUCTION Cyclic vomiting syndrome is a functional disorder characterised by repeated episodes of nausea and vomiting with symptom-free intervals between the attacks. Cyclic vomiting syndrome is a migraine equivalent; therefore, anti-migraine medications are effective for cyclic vomiting syndrome prophylaxis, but duration of treatment is not clear. AIM To determine the efficacy and duration of drug therapy in cyclic vomiting syndrome. MATERIAL AND METHODS This study was conducted on 206 cases of cyclic vomiting syndrome, who were treated with propranolol as prophylaxis. After they were symptom-free for at least 9 months, propranolol was tapered and discontinued and then they were followed for at least 6 months after drug withdrawal for evaluation of recurrence. RESULTS The study subjects included 127 boys and 79 girls. The mean age of symptom onset was 3.4 years (range: 3 months to 14.5 years) and the mean age at the time of diagnosis was 5.7 years (range: 8.4 months to 18 years). Among the 206 patients in whom propranolol was discontinued, only 16 (7.8%) subjects developed recurrence of symptoms in the 6-month follow-up period. CONCLUSIONS There is no need to continue prophylaxis of cyclic vomiting syndrome for a long time. It is also possible to make the duration of drug therapy shorter, which is useful for the convenience of the patients and prevention of drug side effects.
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Affiliation(s)
- Mahmoud Haghighat
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Hajar Memari
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Naser Honar
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Seyed Mohsen Dehghani
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Mohammad-Hadi Imanieh
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Seyed-Javad Injoo
- Gastroenterohepatology Research Center, Nemazee Teaching Hospital, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Hazhir Javaherizadeh
- Research Center for Infectious Diseases of Digestive System [Alimentary Tract Research Center], Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran
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Recurrent Gastrointestinal Disturbance: Abdominal Migraine and Cyclic Vomiting Syndrome. Curr Neurol Neurosci Rep 2017; 17:21. [DOI: 10.1007/s11910-017-0731-4] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
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Fejzo MS, Myhre R, Colodro-Conde L, MacGibbon KW, Sinsheimer JS, Reddy MVPL, Pajukanta P, Nyholt DR, Wright MJ, Martin NG, Engel SM, Medland SE, Magnus P, Mullin PM. Genetic analysis of hyperemesis gravidarum reveals association with intracellular calcium release channel (RYR2). Mol Cell Endocrinol 2017; 439:308-316. [PMID: 27663074 PMCID: PMC6464816 DOI: 10.1016/j.mce.2016.09.017] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/09/2016] [Revised: 08/19/2016] [Accepted: 09/19/2016] [Indexed: 02/07/2023]
Abstract
Hyperemesis Gravidarum (HG), severe nausea/vomiting in pregnancy (NVP), can cause poor maternal/fetal outcomes. Genetic predisposition suggests the genetic component is essential in discovering an etiology. We performed whole-exome sequencing of 5 families followed by analysis of variants in 584 cases/431 controls. Variants in RYR2 segregated with disease in 2 families. The novel variant L3277R was not found in any case/control. The rare variant, G1886S was more common in cases (p = 0.046) and extreme cases (p = 0.023). Replication of G1886S using Norwegian/Australian data was supportive. Common variants rs790899 and rs1891246 were significantly associated with HG and weight loss. Copy-number analysis revealed a deletion in a patient. RYR2 encodes an intracellular calcium release channel involved in vomiting, cyclic-vomiting syndrome, and is a thyroid hormone target gene. Additionally, RYR2 is a downstream drug target of Inderal, used to treat HG and CVS. Thus, herein we provide genetic evidence for a pathway and therapy for HG.
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Affiliation(s)
- Marlena Schoenberg Fejzo
- Department of Medicine, David Geffen School of Medicine, University of California, Los Angeles, CA, USA; Department of Maternal-Fetal Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA.
| | - Ronny Myhre
- Norwegian Institute of Public Health, Oslo Norway
| | - Lucía Colodro-Conde
- Psychiatric Genetics Laboratory, QIMR Berghofer Medical Research Institute, Brisbane, Australia
| | | | - Janet S Sinsheimer
- Departments of Biostatistics, Biomathematics, & Human Genetics, David Geffen School of Medicine, University of California, Los Angeles, CA, USA
| | - M V Prasad Linga Reddy
- Department of Human Genetics, David Geffen School of Medicine, University of California, Los Angeles, CA, USA
| | - Päivi Pajukanta
- Department of Human Genetics, David Geffen School of Medicine, University of California, Los Angeles, CA, USA
| | - Dale R Nyholt
- Institute of Health and Biomedical Innovation, Queensland University of Technology, Brisbane, Australia
| | - Margaret J Wright
- Queensland Brain Institute and Centre for Advanced Imaging, University of Queensland, Brisbane, Australia
| | - Nicholas G Martin
- Genetic Epidemiology Laboratory, QIMR Berghofer Medical Research Institute, Brisbane, Australia
| | - Stephanie M Engel
- Department of Epidemiology, Gillings School of Global Public Health, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA
| | - Sarah E Medland
- Psychiatric Genetics Laboratory, QIMR Berghofer Medical Research Institute, Brisbane, Australia
| | - Per Magnus
- Norwegian Institute of Public Health, Oslo Norway
| | - Patrick M Mullin
- Department of Maternal-Fetal Medicine, Keck School of Medicine, University of Southern California, Los Angeles, CA, USA
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Sezer OB, Sezer T. A New Approach to the Prophylaxis of Cyclic Vomiting: Topiramate. J Neurogastroenterol Motil 2016; 22:656-660. [PMID: 27302967 PMCID: PMC5056575 DOI: 10.5056/jnm16035] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/07/2016] [Revised: 04/23/2016] [Accepted: 04/27/2016] [Indexed: 11/20/2022] Open
Abstract
BACKGROUND/AIMS The aim of this study was to compare the efficacy and tolerability of topiramate and propranolol in preventing pediatric cyclic vomiting syndrome. METHODS A retrospective medical-record review of patients who underwent prophylaxis after receiving a diagnosis of cyclic vomiting syndrome was performed. Patients who completed at least 12 months of treatment were included in the analysis. Responder rate, and adverseevent rates were also calculated from all patients. Response to treatment was assessed as the total number of vomiting attacks per year. Patients in whom the frequency of vomiting attack reduced greater or equal to 50% were defined as responders, and the remaining patients were classified as nonresponders. RESULTS A total of 38 patients who were treated prophylactically with either topiramate (16 patients) or propranolol (22 patients) were identified. Fifty-nine percent of the patients in the propranolol group and 81% of the patients in the topiramate group reported freedom from attacks. A decrease of more than 50% in attacks per year occurred in 23% of patients in the propranolol group and 13% of patients in the topiramate group. The responder rates were 81% for propranolol group and 94% for topiramate group (P = 0.001). Despite minor adverse effects (drowsiness, nervousness, and dizziness) observed in a few patients, the adverse event rates were not significantly different between the 2 groups (P = 0.240). CONCLUSIONS The efficacy of topiramate was superior to propranolol for the prophylaxis of pediatric cyclic vomiting syndrome.
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Affiliation(s)
- Oya B Sezer
- Department of Pediatric Gastroenterology and Nutrition, Kecioren Training and Research Hospital, Ankara,
Turkey
| | - Taner Sezer
- Department of Pediatric Neurology, Baskent University Faculty of Medicine, Ankara,
Turkey
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Haghighat M, Shahramian I, Dehghani SM, Ataollahi M, Bahmanyar M. A New Guidelines For Using Erythromycin In Management Of Childhood Cyclic Vomiting Syndrome. INTERNATIONAL JOURNAL OF BASIC SCIENCE IN MEDICINE 2016. [DOI: 10.15171/ijbsm.2016.14] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/09/2022] Open
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Definitions and Outcome Measures in Pediatric Functional Upper Gastrointestinal Tract Disorders: A Systematic Review. J Pediatr Gastroenterol Nutr 2016; 62:581-7. [PMID: 26348686 DOI: 10.1097/mpg.0000000000000973] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/19/2022]
Abstract
OBJECTIVES Functional disorders of the upper gastrointestinal tract are frequently diagnosed in children. Four different clinical entities are addressed by the Rome III committee: functional dyspepsia (FD), cyclic vomiting syndrome (CVS), adolescent rumination syndrome (ARS), and aerophagia. Management of these disorders is often difficult leading to a wide variety in therapeutic interventions. We hypothesize that definitions and outcome measures in these studies are heterogeneous as well. Our aim is to systematically assess how these disorders and outcomes are defined in therapeutic randomized controlled trials (RCTs). STUDY DESIGN CENTRAL, Embase, and MEDLINE/PubMed were searched from inception to February 25, 2015. Search terms were FD, CVS, ARS, and aerophagia. Therapeutic RCTs, or systematic reviews of RCTs, in English language including subjects ages 4 to 18 years (0-18 years for CVS) were evaluated. Quality was assessed using the Delphi list. RESULTS A total of 1398 articles were found of which 8 articles were included. Seven concerned FD and 1 concerned CVS. In all of the studies, Rome criteria or similar definitions were used; all the studies however used different outcome measures. Seventy-five percent of the trials were of good methodological quality. Only 57% used validated pain scales. CONCLUSIONS Different outcome measures are used in therapeutic trials on functional disorders of the upper gastrointestinal tract. There is a clear paucity of trials evaluating different treatment regimens regarding CVS, ARS, and aerophagia. Uniform definitions, outcome measures, and validated instruments are needed to make a comparison between intervention studies possible.
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Nagarwala J, Dev S, Markin A. The Vomiting Patient: Small Bowel Obstruction, Cyclic Vomiting, and Gastroparesis. Emerg Med Clin North Am 2016; 34:271-91. [PMID: 27133244 DOI: 10.1016/j.emc.2015.12.005] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/20/2023]
Abstract
Vomiting and abdominal pain are common in patients in the emergency department. This article focuses on small bowel obstruction (SBO), cyclic vomiting, and gastroparesis. Through early diagnosis and appropriate management, the morbidity and mortality associated with SBOs can be significantly reduced. Management of SBOs involves correction of physiologic and electrolyte disturbances, bowel rest and removing the source of the obstruction. Treatment of acute cyclic vomiting is primarily directed at symptom control, volume and electrolyte repletion, and appropriate specialist follow-up. The mainstay of therapy for gastroparesis is metoclopramide.
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Affiliation(s)
- Jumana Nagarwala
- Department of Emergency Medicine, Henry Ford Hospital, 2799 West Grand Boulevard, CFP-258, Detroit, MI 48202, USA; Department of Emergency Medicine, Wayne State University School of Medicine, Detroit, MI, USA.
| | - Sharmistha Dev
- Departments of Emergency Medicine and Internal Medicine, University of Michigan, Ann Arbor, MI, USA
| | - Abraham Markin
- Department of Emergency Medicine, Henry Ford Hospital, 2799 West Grand Boulevard, CFP-258, Detroit, MI 48202, USA; Department of Internal Medicine, Henry Ford Hospital, 2799 West Grand Boulevard, Detroit, MI 48202, USA
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Benninga MA, Faure C, Hyman PE, St James Roberts I, Schechter NL, Nurko S. Childhood Functional Gastrointestinal Disorders: Neonate/Toddler. Gastroenterology 2016; 150:S0016-5085(16)00182-7. [PMID: 27144631 DOI: 10.1053/j.gastro.2016.02.016] [Citation(s) in RCA: 351] [Impact Index Per Article: 39.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/08/2016] [Accepted: 02/09/2016] [Indexed: 02/08/2023]
Abstract
In 2006, a consensus concerning functional gastrointestinal intestinal disorders (FGIDs) in infants and toddlers was described. At that time little evidence regarding epidemiology, pathophysiology, diagnostic work-up, treatment strategies and follow-up was available. Consequently the criteria for the clinical entities were more experience than evidence based. In the past decade, new insights have been gained in the different FGIDs in these age groups. Based on those, further revisions have been made to the criteria. The description of infant colic has been expanded to include criteria for the general pediatrician and specific criteria for researchers. The greatest change was the addition of a paragraph regarding the neurobiology of pain in infants and toddlers, including the understanding of the neurodevelopment of nociception and of the wide array of factors that may impact the pain experience.
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Affiliation(s)
- Marc A Benninga
- Pediatric Gastroenterology, Emma Children's Hospital / Academic Medical Center, Amsterdam, The Netherlands.
| | - Christophe Faure
- Pediatric Gastroenterology, Sainte-Justine Hospital, Montreal, Qc, Canada
| | - Paul E Hyman
- Pediatric Gastroenterology, Children's Hospital, New Orleans, USA
| | - Ian St James Roberts
- Thomas Coram Research Unit, UCL Institute of Education, University College London, UK
| | - Neil L Schechter
- Pediatric pain center, Boston Children's Hospital, Boston Ma, USA
| | - Samuel Nurko
- Center for Motility and Functional Gastrointestinal Disorders, Boston Children's Hospital, Boston Ma, USA
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Is Helicobacter pylori infection a risk factor for childhood periodic syndromes? Int J Pediatr Adolesc Med 2015; 2:19-23. [PMID: 30805431 PMCID: PMC6372365 DOI: 10.1016/j.ijpam.2015.03.002] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/25/2014] [Accepted: 09/28/2014] [Indexed: 11/23/2022]
Abstract
Background and objectives Helicobacter pylori (H. pylori) infection has been assumed to have roles in various extra-digestive diseases. The current study was designed to evaluate the incidence of H. pylori infection in patients with cyclic vomiting syndrome and its possible role in the etiology of this disease. Design and setting In this case-control study, 120 cases with diagnoses of cyclic vomiting or abdominal migraine who were registered at the Gastroenterology Clinic at Shiraz University of Medical Sciences from 2010 to 2013 were enrolled. Materials and methods Primarily information regarding the patients' diseases were collected with a data gathering sheet, and fresh morning stool samples were collected from the patients and examined for H. pylori stool antigen with the H. pylori Ag EIA test kit. The results were compared with those of healthy children from the control group. Results A total of 120 patients with cyclic vomiting (47.5%) and abdominal migraine (52.5%) with a mean age of 7.1 ± 3.4 (range 2-16 years) and a male-to-female ratio of 1.6 were included. The HPs Ag tests were positive in only 7 (5.8%) patients in our case group, and the HPs Ag tests were positive in 13 (13%) of the children in the control group; this difference was statistically insignificant. Conclusion Our study did not support H. pylori infection as an etiological factor in CV or AM.
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Haghighat M, Dehghani SM, Shahramian I, Imanieh MH, Teimouri A, Noori NM. Combination of erythromycin and propranolol for treatment of childhood cyclic vomiting syndrome: a novel regimen. GASTROENTEROLOGY AND HEPATOLOGY FROM BED TO BENCH 2015; 8:270-7. [PMID: 26468347 PMCID: PMC4600517] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Download PDF] [Subscribe] [Scholar Register] [Indexed: 11/24/2022]
Abstract
AIM This study aimed to evaluate the erythromycin efficacy in childhood cyclic vomiting syndrome. BACKGROUND Cyclic vomiting syndrome (CVS) is an unusual cause of episodic emesis in children and erythromycin is an effective treatment. PATIENTS AND METHODS In this prospective study, 301 patients with a final diagnosis of CVS enrolled in two separated groups. The first group received erythromycin for 7 days and propranolol for at least 9 months (n=155). The second group was treated with propranolol alone for at least 9 months (n=146). These two groups were compared for response to the treatment and the recurrence of symptoms after treatment completion. Relationship of response, recurrence, and characteristics of the disease was assessed. RESULTS Both groups showed a significant difference in terms of response to treatment (P=0.002), however the recurrence after treatment completion had no considerable difference (P=0.563). There was no relationship between CVS characteristics and these two items (response and recurrence). CONCLUSION In our point of view, the addition of erythromycin to standard propranolol treatment can improve the response to treatment, although it has no significant effect on recurrence of CVS symptoms. We suggest the use of erythromycin for 7 days in addition to CVS standard therapy.
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Affiliation(s)
- Mahmood Haghighat
- Gastroenterohepatology Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Seyed Mohsen Dehghani
- Gastroenterohepatology Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Iraj Shahramian
- Gastroenterohepatology Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Mohammad Hadi Imanieh
- Gastroenterohepatology Research Center, Shiraz University of Medical Sciences, Shiraz, Iran
| | - Alireza Teimouri
- Children and Adolescents Health Research Center, Zahedan University of Medical Sciences, Zahedan, Iran
| | - Noor Mohammad Noori
- Children and Adolescents Health Research Center, Zahedan University of Medical Sciences, Zahedan, Iran
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Smolensky MH, Portaluppi F, Manfredini R, Hermida RC, Tiseo R, Sackett-Lundeen LL, Haus EL. Diurnal and twenty-four hour patterning of human diseases: acute and chronic common and uncommon medical conditions. Sleep Med Rev 2014; 21:12-22. [PMID: 25129839 DOI: 10.1016/j.smrv.2014.06.005] [Citation(s) in RCA: 85] [Impact Index Per Article: 7.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/26/2014] [Revised: 06/12/2014] [Accepted: 06/12/2014] [Indexed: 01/30/2023]
Abstract
The symptom intensity and mortality of human diseases, conditions, and syndromes exhibit diurnal or 24 h patterning, e.g., skin: atopic dermatitis, urticaria, psoriasis, and palmar hyperhidrosis; gastrointestinal: esophageal reflux, peptic ulcer (including perforation and hemorrhage), cyclic vomiting syndrome, biliary colic, hepatic variceal hemorrhage, and proctalgia fugax; infection: susceptibility, fever, and mortality; neural: frontal, parietal, temporal, and occipital lobe seizures, Parkinson's and Alzheimer's disease, hereditary progressive dystonia, and pain (cancer, post-surgical, diabetic neuropathic and foot ulcer, tooth caries, burning mouth and temporomandibular syndromes, fibromyalgia, sciatica, intervertebral vacuum phenomenon, multiple sclerosis muscle spasm, and migraine, tension, cluster, hypnic, and paroxysmal hemicranial headache); renal: colic and nocturnal enuresis and polyuria; ocular: bulbar conjunctival redness, keratoconjunctivitis sicca, intraocular pressure and anterior ischemic optic neuropathy, and recurrent corneal erosion syndrome; psychiatric/behavioral: major and seasonal affective depressive disorders, bipolar disorder, parasuicide and suicide, dementia-associated agitation, and addictive alcohol, tobacco, and heroin cravings and withdrawal phenomena; plus autoimmune and musculoskeletal: rheumatoid arthritis, osteoarthritis, axial spondylarthritis, gout, Sjögren's syndrome, and systemic lupus erythematosus. Knowledge of these and other 24 h patterns of human pathophysiology informs research of their underlying circadian and other endogenous mechanisms, external temporal triggers, and more effective patient care entailing clinical chronopreventive and chronotherapeutic strategies.
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Affiliation(s)
- Michael H Smolensky
- Department of Biomedical Engineering, Cockrell School of Engineering, The University of Texas at Austin, Austin, TX, USA.
| | - Francesco Portaluppi
- Hospital S. Anna and Department of Medical Sciences, University of Ferrara, Ferrara, Italy
| | - Roberto Manfredini
- Hospital S. Anna and Department of Medical Sciences, University of Ferrara, Ferrara, Italy
| | - Ramon C Hermida
- Bioengineering & Chronobiology Laboratories, University of Vigo, Campus Universitario, Vigo, Spain
| | - Ruana Tiseo
- Hospital S. Anna and Department of Medical Sciences, University of Ferrara, Ferrara, Italy
| | - Linda L Sackett-Lundeen
- Department of Laboratory Medicine & Pathology, University of Minnesota, HealthPartners Institute for Education and Research and the Department of Pathology, Regions Hospital, St. Paul, MN, USA
| | - Erhard L Haus
- Department of Laboratory Medicine & Pathology, University of Minnesota, HealthPartners Institute for Education and Research and the Department of Pathology, Regions Hospital, St. Paul, MN, USA
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Approach to the diagnosis and treatment of cyclic vomiting syndrome: a large single-center experience with 106 patients. Pediatr Neurol 2014; 50:569-73. [PMID: 24842256 DOI: 10.1016/j.pediatrneurol.2014.02.009] [Citation(s) in RCA: 24] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/18/2013] [Revised: 02/05/2014] [Accepted: 02/12/2014] [Indexed: 12/27/2022]
Abstract
BACKGROUND Cyclic vomiting syndrome is characterized by repeated, stereotypical vomiting episodes. The diagnosis is made by exclusion of other organic diseases, which can lead to extensive testing. It has been suggested that these patients can have mitochondrial dysfunction. The aim of the study was to examine the evaluation of our cyclic vomiting patients and to determine whether they had associated, undiagnosed metabolic abnormalities. METHODS This retrospective study included 106 patients aged <21 years at diagnosis. Information regarding medical history, laboratory, and imaging studies were collected. Metabolic studies in plasma and urine were obtained when patients were well and when patients were in a vomiting cycle, including plasma amino acids, acylcarnitines, and urine organic acids. RESULTS The mean age at diagnosis was 8.9 ± 5.0 years. Neuroimaging revealed previously unknown intracranial abnormalities in <10% of patients, none of whom explained the vomiting signs. Abdominal ultrasounds revealed abnormalities in 15% of patients during an acute episode and 7% of patients when well. Sixty-one patients had an upper gastrointestinal series, all of which were normal. A total of 92% of patients had laboratory testing with 38% indicating abnormalities possibly suggesting mitochondrial dysfunction. CONCLUSIONS This large, single-center study further evaluated the need for more focused evaluation in patients with suspected cyclic vomiting syndrome. Thirty-eight percent of our patients had abnormalities in blood and/or urine suggesting mitochondrial dysfunction, which requires more detailed investigation in the future.
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Hejazi RA, McCallum RW. Cyclic vomiting syndrome: treatment options. Exp Brain Res 2014; 232:2549-52. [PMID: 24862509 DOI: 10.1007/s00221-014-3989-7] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2014] [Accepted: 05/07/2014] [Indexed: 11/29/2022]
Abstract
Cyclic vomiting syndrome is a disorder characterized by recurrent episodes of severe nausea and vomiting separated by symptom-free periods. Our aims were to review treatments of adult cyclic vomiting syndrome as well as to identify areas for further clinical research and the unanswered questions in this field. We conducted a PubMed search using such keywords as "cyclic vomiting syndrome," "nausea," "vomiting," "treatment," "trigger factors" and "tricyclic antidepressants" and combined this information with the knowledge and clinical research from the authors. Available data show that in adult cyclic vomiting syndrome, there is an impressive and sustained response to high-dose tricyclic antidepressants. In up to 13% who are regarded as poor responders to tricyclic antidepressants, a predictable profile can be identified related to coexisting psychological disorders, marijuana use, poorly controlled migraine headache or chronic narcotic use. Cyclic vomiting syndrome in adults is being an increasingly recognized entity. Tricyclic antidepressants are the main treatment for controlling symptoms. Eliminating and addressing trigger factors are an essential part of management.
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Affiliation(s)
- Reza A Hejazi
- Department of Internal Medicine, Paul L. Foster School of Medicine, Texas Tech University Health Sciences Center, 4800 Alberta Ave, El Paso, TX, 79905, USA,
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Altun H, Gül Ö, Öksüz AN, Şahin N. Cyclic vomiting syndrome treated successfully with fluoxetin. Turk Arch Pediatr 2014; 49:88-9. [PMID: 26078641 DOI: 10.5152/tpa.2014.1373] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/06/2013] [Accepted: 10/30/2013] [Indexed: 11/22/2022]
Affiliation(s)
- Hatice Altun
- Department of Child and Adolescent Psychiatry, Kahramanmaraş University Faculty of Medicine, Kahramanmaraş, Turkey
| | - Özlem Gül
- Department of Pediatrics, Kahramanmaraş University Faculty of Medicine, Kahramanmaraş, Turkey
| | - Ali Nuri Öksüz
- Department of Psychiatry, Kahramanmaraş University Faculty of Medicine, Kahramanmaraş, Turkey
| | - Nilfer Şahin
- Department of Child and Adolescent Psychiatry, Muğla Sıtkı Koçman University Education and Research Hospital, Muğla, Turkey
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Cyclic vomiting syndrome in adults. Wien Med Wochenschr 2013; 163:514-6. [DOI: 10.1007/s10354-013-0250-0] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/06/2013] [Accepted: 10/25/2013] [Indexed: 12/17/2022]
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Abstract
Cyclic vomiting syndrome (CVS) is a rare abnormality of the neuroendocrine system that affects 2% of children. It is a frequently missed diagnosis in the emergency department and may require a number of emergency department visits before the diagnosis is made. The objectives of this review are to identify the clinical features that suggest a diagnosis of CVS and to review the literature on its management. The MEDLINE and EMBASE databases were searched from January 1948 to October 2011 using the keywords 'Cyclic' or 'Vomiting'. Papers were excluded if they did not follow the consensus guidelines or if they were case reports. This review analysed 1093 cases of cyclic vomiting in 25 papers that fulfilled the inclusion criteria. All except one paper were retrospective studies. The size of these cohort studies ranged from three to 181 patients, with a mean patient size of 29. This review found that over 40% of patients have headaches/migraines, with associated anxiety and depression in ≈ 30% of cases. There is a family history of headaches/migraines in 38.9%, and this association was much stronger in the adult CVS cohort compared with the paediatric cohort. Compared with paediatric CVS, adults have a longer duration of attacks and they occur more frequently (5.9 vs. 3.4 days, 14.4 vs. 9.6 episodes/year). Limited data are available on the treatment of the acute phase of CVS, but in adults, sumatriptan has been shown to be effective. For prophylactic treatment, tricyclics are effective in both adult and paediatric CVS, with a clinical response in 75.5 and 67.6% of patients, respectively, in nonplacebo-controlled cohort studies. Furthermore, propranolol has been shown to be useful in children. CVS is an intractable illness with a major impact on the patient's quality of life. There is a long duration between the onset of symptoms and the diagnosis of the condition. There is a high association with headaches/migraines and anxiety/depression. The symptoms are more severe in adult-onset CVS. Tricyclic antidepressants have good efficacy in reducing the frequency/duration or the intensity of attacks. There is limited evidence on the acute management of CVS.
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Abstract
INTRODUCTION Cyclic vomiting syndrome (CVS) is a condition characterized by recurrent, stereotyped, attacks of intense nausea and intractable vomiting with no identifiable cause. The diagnosis is made according to the Rome III criteria; however, the condition remains a heterogenous group of symptoms and few studies have analysed patient-reported features in detail. Tricyclic antidepressants may benefit some patients, but there are no data on the long-term outcomes of this condition. METHODS This is a retrospective cohort study of all patients treated at two London teaching hospitals between 1994 and 2011. Information was obtained by means of inpatient case notes, clinic letters and telephone interviews using a standardized questionnaire. RESULTS Twenty-eight patients were identified, 17 with adult-onset CVS and 11 with childhood-onset CVS. In the adult-onset CVS cohort, the average age of onset was 30 years; the prevalence of headache was 57.1% and anxiety/depression 21.7%. Vomiting attacks occurred on average 10 times a year with a mean duration of 55.3 h. In the childhood-onset CVS cohort, the average age of onset was 5 years and the prevalence of headaches was 44.4%. Vomiting attacks occurred on average 25.5 times a year with a mean duration of 54.5 h. During acute attacks, patients rated morphine-based medications as the most efficacious. Overall, 65.4% of our patients were not treated with long-term prophylactic medications and, of these, 42.1% felt that their symptoms had improved and 26.3% reported complete resolution. CONCLUSION CVS is a debilitating illness with frequent attacks of prolonged duration. There is a high association with anxiety/depression as well as headaches. Antimigraine medication, β-blockers and antiepileptic treatment may be successful at attenuating attacks; however, in this long-term study, the prognosis of patients not treated with these medications was also good. Overall, 61.9% of patients showed a gradual improvement in symptoms and 23.8% had complete resolution after a mean of 7 years.
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Contopoulos-Ioannidis DG, Seto I, Hamm MP, Thomson D, Hartling L, Ioannidis JPA, Curtis S, Constantin E, Batmanabane G, Klassen T, Williams K. Empirical evaluation of age groups and age-subgroup analyses in pediatric randomized trials and pediatric meta-analyses. Pediatrics 2012; 129 Suppl 3:S161-84. [PMID: 22661763 DOI: 10.1542/peds.2012-0055j] [Citation(s) in RCA: 28] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/24/2022] Open
Abstract
BACKGROUND An important step toward improvement of the conduct of pediatric clinical research is the standardization of the ages of children to be included in pediatric trials and the optimal age-subgroups to be analyzed. METHODS We set out to evaluate empirically the age ranges of children, and age-subgroup analyses thereof, reported in recent pediatric randomized clinical trials (RCTs) and meta-analyses. First, we screened 24 RCTs published in Pediatrics during the first 6 months of 2011; second, we screened 188 pediatric RCTs published in 2007 in the Cochrane Central Register of Controlled Trials; third, we screened 48 pediatric meta-analyses published in the Cochrane Database of Systematic Reviews in 2011. We extracted information on age ranges and age-subgroups considered and age-subgroup differences reported. RESULTS The age range of children in RCTs published in Pediatrics varied from 0.1 to 17.5 years (median age: 5; interquartile range: 1.8-10.2) and only 25% of those presented age-subgroup analyses. Large variability was also detected for age ranges in 188 RCTs from the Cochrane Central Register of Controlled Trials, and only 28 of those analyzed age-subgroups. Moreover, only 11 of 48 meta-analyses had age-subgroup analyses, and in 6 of those, only different studies were included. Furthermore, most of these observed differences were not beyond chance. CONCLUSIONS We observed large variability in the age ranges and age-subgroups of children included in recent pediatric trials and meta-analyses. Despite the limited available data, some age-subgroup differences were noted. The rationale for the selection of particular age-subgroups deserves further study.
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Affiliation(s)
- Despina G Contopoulos-Ioannidis
- Department of Pediatrics, Division of Infectious Diseases, Stanford Prevention Research Center, Stanford University School of Medicine, Stanford, California 94305, USA.
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[Stereotypical episodes of vomiting for 11 years in a 33-year-old patient]. Internist (Berl) 2011; 53:345-50. [PMID: 22146936 DOI: 10.1007/s00108-011-2962-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/14/2022]
Abstract
A 33-year-old man was admitted because of severe vomiting. For the last 11 years, he had suffered recurrent stereotypical episodes of vomiting lasting 3-4 days. The episodes of vomiting occurred 10-15 times a year. Moreover his brother and his mother had similar symptoms. Thus, (familial) cyclic vomiting syndrome was diagnosed. With the help of antiemetic and sedative drugs, the acute vomiting episode was stopped. Prophylactic therapy with amitriptyline was started, which led to a symptom-free period of 3.5 years without a new episode of vomiting.
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Hejazi RA, McCallum RW. Review article: cyclic vomiting syndrome in adults--rediscovering and redefining an old entity. Aliment Pharmacol Ther 2011; 34:263-73. [PMID: 21668458 DOI: 10.1111/j.1365-2036.2011.04721.x] [Citation(s) in RCA: 56] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 01/02/2023]
Abstract
BACKGROUND Cyclic vomiting syndrome is a disorder characterised by recurrent episodes of severe nausea and vomiting separated by symptom-free periods. AIMS To review the history, epidemiology, clinical aspects, pathophysiology, diagnosis and treatments of adult cyclic vomiting syndrome as well as to identify areas for further clinical research and the unanswered questions in this field. METHODS We conducted a PubMed search using such keywords as cyclic vomiting syndrome; nausea; vomiting; pathophysiology; diagnosis; treatment; trigger factors; gastric emptying test; autonomic nerve function test; gastrointestinal hormones; outcome and natural history and combined this information with the knowledge and extensive clinical research and publications from the authors. RESULTS Available data show that in adult cyclic vomiting syndrome, severe epigastric and sometimes diffuse abdominal pain accompanies most cycles of nausea and vomiting interspersed with periods of symptomatic remission. Psychological disorders, specifically anxiety and depression are common, and gastric emptying is actually rapid in approximately 60% of patients and normal in the remainder. There is an impressive and sustained response to high-dose tricyclic antidepressants. In up to 15% who are regarded as poor responders to tricyclic antidepressants, a predictable profile can be identified related to co-existing psychological disorders, marijuana use, poorly controlled migraine headache or chronic narcotic use. CONCLUSIONS Cyclic vomiting syndrome in adults is an entity that is being increasingly recognised, but the need to educate Internists, Gastroenterologists and Emergency Department staff remains an ongoing challenge.
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Affiliation(s)
- R A Hejazi
- Department of Internal Medicine, Paul L. Foster School of Medicine, Texas Tech University Health Sciences Center, El Paso, TX 79905, USA
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Lin YP, Ni YH, Weng WC, Lee WT. Cyclic Vomiting Syndrome and Migraine in Children. J Formos Med Assoc 2011; 110:382-7. [DOI: 10.1016/s0929-6646(11)60056-9] [Citation(s) in RCA: 23] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/06/2010] [Revised: 04/19/2010] [Accepted: 05/19/2010] [Indexed: 11/30/2022] Open
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Cyclic Vomiting Syndrome in Taiwanese Children. J Formos Med Assoc 2011; 110:14-8. [DOI: 10.1016/s0929-6646(11)60003-x] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/11/2009] [Revised: 12/01/2009] [Accepted: 02/01/2010] [Indexed: 01/10/2023] Open
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Hubbard TA, Crisp CA. Cessation of cyclic vomiting in a 7-year-old girl after upper cervical chiropractic care: a case report. J Chiropr Med 2010; 9:179-83. [PMID: 22027110 PMCID: PMC3206580 DOI: 10.1016/j.jcm.2010.07.006] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/15/2010] [Accepted: 07/19/2010] [Indexed: 10/19/2022] Open
Abstract
OBJECTIVE The purpose of this case report is to describe chiropractic management of a child with cyclic vomiting syndrome. CLINICAL FEATURES A 7-year-old girl had a history of cyclic vomiting episodes for the past 4 1/2 years. She also had a 2-month history of headaches and stomachache. INTERVENTION AND OUTCOME The patient received low-force chiropractic spinal manipulation to her upper cervical spine. There was improvement in her symptoms within an hour after the chiropractic manipulation. Her symptoms only returned after direct trauma to her neck. The recurring symptoms again disappeared immediately after treatment. CONCLUSION This case study suggests that there may be a role for the use of chiropractic spinal manipulative therapy for treating cyclic vomiting syndrome. Controlled studies are necessary to aid our understanding of this finding.
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Affiliation(s)
- Todd A. Hubbard
- Assistant Professor, Palmer College of Chiropractic, Davenport, IA
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Efficacy of Nebivolol and Amitriptyline in the Prophylaxis of Cyclic Vomiting Syndrome. Neurologist 2010; 16:313-4. [DOI: 10.1097/nrl.0b013e3181b120a6] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
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Abstract
This review focuses on so-called "periodic syndromes of childhood that are precursors to migraine," as included in the second edition of the International Classification of Headache Disorders. Presentation is characterized by an episodic pattern and intervals of complete health. Benign paroxysmal torticollis is characterized by recurrent episodes of head tilt, secondary to cervical dystonia, with onset between ages 2-8 months. Benign paroxysmal vertigo presents as sudden attacks of vertigo lasting seconds to minutes, accompanied by an inability to stand without support, between ages 2-4 years. Cyclic vomiting syndrome is distinguished by its unique intensity of vomiting, affecting quality of life, whereas abdominal migraine presents as episodic abdominal pain occurring in the absence of headache. Their mean ages of onset are 5 and 7 years, respectively. Diagnostic criteria and appropriate evaluation represent the key issues. Therapeutic recommendations include reassurance, lifestyle changes, and prophylactic as well as acute antimigraine therapy.
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Affiliation(s)
- Latha Chandran
- State University of New York at Stony Brook, Stony Brook, NY, USA
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