Baclofen in a Child with Rumination-Regurgitation Disorder

Abstract

Rumination-Regurgitation disorder (RRD) in children refers to repeated regurgitation with rechewing or expulsion of food soon after meal ingestion without any preceding retching or nausea. Management focused on diaphragmatic breathing and other modalities, such as cognitive behavioral therapy, is challenging for toddlers. Here we report a 2-year-old boy with RRD who responded favorably to baclofen, a Gamma Amino Butyric Acid B receptor (GABA_B) agonist.

Introduction

Rumination comes from the Latin ruminare, meaning “to chew the cud.” In humans, rumination is characterized by the effortless regurgitation of food, typically occurring soon after meals. Initially, it was grouped under functional gastrointestinal disorders, but it is now categorized under disorders of the gut-brain axis. ¹ Rumination syndrome in children is characterized, according to the Rome IV criteria, as repeated regurgitation with rechewing or expulsion of food that begins shortly after meal ingestion, does not occur at night, and is not preceded by retching. The three distinct age groups affected are: infants and toddlers, neurodiverse youth with varied psychological, neurological, and neurodevelopmental issues, and neurotypical youth. Rumination is often underdiagnosed, with caregivers seeking multiple healthcare providers and experiencing significant delays in obtaining an accurate diagnosis. The condition has a prevalence of 1.7% in infants and 2.1% in toddlers. ²

Theories associated with its etiology include: a primary pathway involving premonitory urges (similar to those observed in tic disorders), a pathway secondary to ongoing pathophysiology (such as gastroesophageal reflux), and a pathway secondary to psychosocial mechanisms, where rumination persists as a learned behavior in response to contextual cues, such as the sight or smell of food. ²

If untreated, rumination disorder can lead to various complications, including weight loss, malnutrition, dental erosion, halitosis, and electrolyte disturbances, along with significant functional disabilities. Previously, rumination was thought to be common among children and adolescents with developmental abnormalities and learning difficulties. However, it is now increasingly recognized in individuals with normal cognitive abilities. ³ The majority of children failed to respond to any medication or psychological interventions. Here, we present the case of a 2-year-old child with rumination disorder who responded favorably to oral baclofen.

Case Report

A 2-year-and-2-month old typically developing boy presented with complaints of regurgitation of food soon after every meal over the past year. The boy had been well until a year ago when he experienced 4–5 vomiting episodes following a febrile illness. He was admitted to a hospital and treated for the condition, and he recovered completely.

After discharge, the child began frequently pointing to his throat, which the mother interpreted as discomfort related to the previous vomiting episodes or some local throat problem. Gradually, the child began to regurgitate undigested food soon after every meal. Occasionally, he inserted his fingers or entire hand into his mouth, inducing vomiting. When prevented from doing so, he would remain quiet for a few minutes but would resume the behavior when the mother became occupied. Over time, feeding the child became increasingly challenging, and the parents noticed that the child was not gaining weight.

Multiple consultations were sought at different healthcare centers, but each time the parents were told the child had gastroesophageal reflux disease (GERD), and antacids were prescribed. However, the mother observed minimal improvement, and the child’s behavior persisted.

On examination, general physical findings were unremarkable. Anthropometric measures revealed weight-for-height between −2 SD and −3 SD (wasting). There was no evidence of anemia or any other signs of micronutrient deficiency. Systemic examination, including abdominal examination, was unremarkable. During the interview, the child was alert and cheerful on his mother’s lap, attempted to reach for objects on the table, and exhibited no self-induced vomiting behavior. The rest of the neurological examination was unremarkable. Based on these clinical features, a provisional diagnosis of rumination-regurgitation disorder (RRD) (6B85) was made in accordance with the International Classification of Diseases, 11th edition (ICD-11) and Rome-4 criteria. ⁴

Blood investigations, including hemoglobin level, blood urea, serum creatinine, and Serum Glutamic Pyruvic Transaminase (SGPT) levels, were normal, with a normal ultrasound abdomen. The child’s weight was around 10.2 kilograms. He et al.’s research on the pediatric pharmacokinetics of baclofen use in children recommended a maximum dose of 2 mg/kg of oral baclofen for children more than 2 years of age.^5,6 The patient was started on a lower dose of 2.5 ml baclofen syrup once daily at night (0.25 mg/kg) and slowly titrated to 5 ml (0.5 mg/kg) over a period of 1 month. Behavioral measures, including small frequent feedings, distraction techniques such as play soon after meals, along with maintenance of an erect posture in the chair throughout and after feeding, were advised. The mother was advised to maintain a log documenting the number and pattern of regurgitation episodes, as well as the number of days she successfully implemented the behavioral strategies. Ten days later, the parents reported that the child’s self-induced regurgitation behavior had reduced, and he was consuming adequate meals. There was a consistent reduction in the number of episodes as per the mother’s log. At the end of 3 months, there were only 1 episode per month. Hence, slowly, baclofen was tapered to 2.5 ml again and then stopped after 1 month. The child was followed for 3 weeks after the cessation of medicines, and no further episodes were noted.

Discussion

The present case reports a favorable response of RRD to oral baclofen, thus adding potential benefit of baclofen for treating toddlers with RRD. Diaphragmatic breathing serves as a mainstay of treatment for RRD by creating a competing response to abdominal wall contractions. However, in this case, the patient was only 2-year-and-2-month old, making it impractical to attempt this technique. Other evidence-based interventions, such as relaxation techniques, cognitive behavioral therapy, antidepressants, and anxiolytics, were also not feasible due to the child’s age. ² The child had previously been treated with antacids for at least 6 months, but symptoms showed no improvement. Techniques producing competing responses, such as chewing or sucking on hard candy, were also unsuitable due to the child’s age.

The vasovagal reflex relaxes the lower oesophageal sphincter (LES) as food enters the stomach, but predisposes it to acid and food reflux to occur. As a result, the transient lower esophageal sphincter relaxation (TLESR) is the most likely cause of GERD. ⁶ Research evidence supports the efficacy of baclofen in increasing lower esophageal sphincter pressure, reducing transient lower esophageal relaxations, decreasing reflux episodes through Gamma Amino Butyric Acid B receptor (GABA_B) activation, and improving subjective symptoms. Baclofen is effective in pediatric gastroesophageal reflux and was considered an appropriate treatment option for this case. ² Baclofen also suppresses the esophageal sensory neurons and, as a result, despite the persistence of acid reflux, the patient does not feel any heartburn. Baclofen is associated with adverse effects such as drowsiness and giddiness, but these were absent in the case of this child. ⁶

A multidisciplinary approach involving a pediatrician, mental health expert, and dietician is essential, particularly in cases with complications such as weight loss. Treatment begins with confirming the diagnosis, providing reassurance, and psycho-educating parents about the pathophysiology of rumination syndrome, including factors that may exacerbate symptoms.

In this case, the parents were disheartened by the poor response to antacids and the child’s failure to gain weight. A neurobiological explanation of the illness was provided, and the potential benefits of baclofen were discussed. The good response to baclofen in this case may be attributed to the absence of triggering events, such as stress, inflammation, or gastroesophageal reflux.

While previous reports have employed multiple treatment modalities, to our knowledge, this is the first report in a toddler with RRD showing a favorable clinical outcome with baclofen. ²