Authors: Dechantal C. V. Montano, OTR/L, Sara T. Naegele, MS Ed,  Ronald C. Savage, Ed.D.

Introduction

Children with acquired brain injuries face an array of cognitive, behavioral, and sensory-motor problems.   In particular, many of these children have problems with sensory processing, which, in turn, interfere with their neuro-development. Sensory processing (sometimes called “sensory integration” or SI) is a term that refers to the way the nervous system receives messages from the senses and turns them into appropriate motor and behavioral responses. Sensory Processing Disorder (SPD) is a condition that exists when sensory signals do not get organized into appropriate responses. Dr. A. Jean Ayres (Ayres, 2005), an occupational therapist and developmental psychologist, pioneered work in sensory integration dysfunction in the 1960s. She proposed that in order to function the brain needed to receive and interpret sensory information correctly using a seven-sense sensory system, which includes the tactile, visual, auditory, gustatory, olfactory, vestibular and proprioception systems. She likened sensory dysfunction to a neurological “traffic jam” because problems occur when sensory input is not processed or misrouted in the brain. “SPD is not one specific disorder…but rather an umbrella term to cover a variety of neurological disabilities,” explains Carol Kranowitz (Kranowtiz, 2005), an educator and expert in sensory processing and integration. Children with SPD, especially children with acquired brain injuries, will find it difficult to process and act upon information received through their senses, which creates challenges in performing countless everyday tasks. Learning difficulties, motor clumsiness, behavioral problems, anxiety, depression, school failure, and other impacts may result if the disorder is not treated effectively.

SPD is not presently recognized as a distinct and separate group of disorders in The Diagnostic and Statistical Manual V but is recognized as occurring in individuals with other neurological disorders. Most research to date involving SPD has involved individuals with Attention Deficit and Hyperactivity Disorder (ADHD) and Autism Spectrum Disorders (ASD). Few isolated studies on how Sensory Processing Disorder manifests as an image of brain dysfunction have been conducted. This could be in part because its existence as a separate and distinct neurological disability is still quite controversial. According to a recent policy statement put forth by the American Academy of Pediatrics, “the amount of research regarding the effectiveness of sensory integration therapy is limited and inconclusive,” (2015). However, Owen et al. (2013) in a landmark study showed that children who met criteria only for sensory processing disorder have anatomical differences in their brain structure than children who do not present with sensory processing difficulties. Owen and her colleagues at The University of California San Francisco found that children affected specifically with Sensory Processing Disorder (SPD) have abnormal white matter microstructure. This is the first time that a biological basis for the disease, which sets it apart from other neuro-developmental disorders has been found. According to the researchers, one of the reasons SPD has been overlooked until now is that it often occurs in children who also have ADHD or autism. This study also shows that SPD may in fact be a distinct disorder that affects individuals with and without other neurological disabilities. Therefore, more research into this disorder as well as treatment for this disorder needs to be further explored in other populations.

As mentioned above, the common pediatric conditions of study with Sensory Processing Disorder are Autism and ADHD; therefore, the uniqueness of this study is that it focuses on SPD in a population other than those previously studied. The subject is a 14-year-old female who has had early biological insults to the developing brain via infantile stroke and the condition Electrical Status Epilepticus of Sleep (ESES). This is a rare form of epilepsy that produces subclinical (unseen) seizures during sleep. This type of epilepsy is characterized by the presence of generalized 1-3 Hz spike-wave discharges occupying 85% or more of the EEG of non-REM sleep. Overall, she presents with a neuro-developmental disorder due to an acquired brain injury (ABI) occurring both in utero and continuously due to the ESES. Ungerleider (1995) states “recent functional brain imaging studies in humans indicate that learning and memory involve many of the same regions of the cortex that process sensory information and control motor output.” This study shows that there is a potential for a positive correlation between SPD, learning and memory.      Furthermore, findings from a study by Tarapore et al (2013) support the hypothesis since “disruption of the long-range communication between brain regions, as measured by reductions in functional connectivity, would be found in patients [with TBI] compared with matched controls.” The results from this study and Owen et al show that both individuals with traumatic brain injury (TBI) and SPD have reduced connectivity between brain regions. Therefore, SPD could be a prevalent issue either dependent or independent of the brain injury for individuals with ABI; subsequently, the intervention for SPD could be effective for individuals with ABI.

Given the lack of research in the area of SPD for acquired brain injury populations coupled with the researchers’ combined therapeutic and educational experiences an interest was taken into studying the relationship between increased sensory processing skills and academic achievement for a 14-year-old female with infantile stroke and Electrical Status Epilepticus of Sleep (ESES) among other neurological disorders. The objective of this study was to determine the relationship between an increase in sensory processing alertness/engagement and academic achievement for a subject with neuro-developmental disorders specifically ESES. Since there was evidence that Sensory Processing Disorder was indeed a neuro-developmental brain disorder, and manifested in a neurologically similar way as ABI, it was hypothesized that the similarities between the deficit manifestations could respond to an external intervention previously used for individuals with SPD and ultimately improve cognition and learning. As previously stated very few studies have been conducted to investigate this theory; however, Polatajko et al (1991) found that a sensory integrative intervention was as effective as perceptual-motor training,  “when administered for 1 hour, once a week, for 6 months…in improving academic and motor performance in children with learning disabilities associated with SI dysfunction.”

Subject Background
The subject was internationally adopted at seven months old. Her hospital records contained insufficient documentation of parental pre-adoption history; however, according to medical records, her weight was reported to be 700 grams, and she had APGAR scores of 3, 5, and 5. Her hospital birth records noted the amniotic fluid smelled fetid.  It was later that an analysis of an MRI of her brain indicated interruption in blood flow to the brain. This occurred at approximately the 23rd week of gestation.  Based upon a CT scan taken at 9 months, doctors estimated she had lost approximately 30 percent of brain matter. Subject’s current head circumference is 43.3 cm, which is considered to be in the first percentile for a 20-month-old girl and indicates microcephaly.Her motor and cognitive developmental milestones were all delayed or never occurred.  In regards to her gross motor development, her records indicated that she began standing alone while holding onto a stable object at 3.5 years; she began to walk independently on a flat surface at 4.5 years, and was able to navigate stairs with assistance at 6.5 years. In regards to her cognitive development with a focus on communication, her records indicated, she was interested and attracted to sound at 7 months, she spoke her first word at 3 years, she spoke unintelligible words and phrases at 9 years, and began speaking in some intelligible short simple sentences in combination with a high-tech communication device at 13 years.

When initial researchers’ interventions with the subject began, formal education had stopped for an entire academic year. The subject was instead receiving traditional out-patient therapies (OT, PT, and speech), and Augmentative and Alternative Communication (AAC) Training for her Dynavox device for approximately 20 hours each week.

Methodology
Researchers gathered baseline academic achievement through scores indicated on her Individualized Education Plan (IEP) from September 2013. The scores were taken from an independent psycho-education evaluation. Additionally, modified testing using the Brigance Inventory of Early Development III (2013) was completed in order to corroborate results specified in her IEP. Tests included: color identification, identifies uppercase letters, identifies basic personal information, rote counting, reads words from common signs, word recognition grade placement test (modified). Subject responded to questions utilizing her AAC device and verbal responses to closed questioning. Additionally, researchers collected baseline sensory systems data. Both cognitive and sensory intervention was delivered in school and home with participation of medical, educational and technical staff.

Research clinicians used an experimenter designed 7-sense diet and tracking sheet to measure outcomes from the Experimenter Sensory Diet, based on the sensory diet theory from research conducted on subjects with sensory defensiveness by Occupational Therapist Patricia Wilbarger in 1991. “A sensory diet is a form of a home program intervention plan that incorporates organizing sensory input, or utilizes already existing sensory input, into everyday life in order to assist the person to maintain a regulated behavioral state, such as the calm, alert state required during certain school activities (Wilbarger, 1991). Multiple sensory diet strategies may be implemented at regular intervals throughout the day. In particular, these strategies may be performed prior to times that may be considered challenging, in order to prepare or set up the body to maintain an organized state throughout the activity, or they may also be used during activities in order to assist the client to maintain an organized state throughout the activity (Williams & Shellenberger, 1994). In this study, the diet tracking sheets were designed by the research clinicians to measure the observable sensory strategies via Levels of Engagement (LoE) and Levels of Alertness (LoA) on a 5-point scale, and were recorded by a trained technician on specific diet adjustments dependent upon the various phases of the research.

Direct Instruction coupled with aspects of strategy-instruction was utilized during individual/small group instruction. Glang et al (1991) found that after 12 hours of direct instruction, “both Subject made substantial academic progress in their targeted instructional areas.” Direct Instruction is a teacher-directed method of instruction in which cognitive skills are broken down into very basic components and explicitly taught. “Specific techniques include: breaking down the instructions and tasks into small steps, teaching modeling of skill or task, administering feedback repeatedly…allowing independent practice and individually paced instruction. “ In addition, aspects of Strategy Instruction such as “verbal modeling, questioning, reminders to use strategies or procedures,” (Sohlberg, M, et al, 2005) were utilized. Direct Instruction was utilized because it is one of the only researched methods of instruction shown to be effective in teaching Subjects with Acquired Brain Injury.  Glang et al (2008) concluded in a literature review that “Two instructional approaches—Direct Instruction and cognitive strategy intervention—have significant evidence supporting their use with many populations of children, with and without disabilities, and address many of the common needs and learning characteristics of Subjects with TBI, thus showing particular promise for these Subjects.”

Review of Process
Modified 7- sense (visual, tactile, auditory, vestibular, proprioceptive, olfactory, and gustatory) sensory diets were assigned in three phases: The Introductory Phase, The Second Phase (Extremity-Specific Phase), and The Third Phase (Subject Initiation Phase). In the first 4 months, diets were implemented up to 6 times a day on the subject (Head, Upper and Lower Extremities). During the next 8 months, diets were scaled back and implemented to 3 times a day.

During all of the phases, a modified 7-sense tracking sheet was aligned to the specific extremity and the protocol order with (Levels of Alertness) LoA and Levels of Engagement (LoE). Likert Scales for LoA and LoE were developed by the researchers and attached to the protocol.  LoA were measured as: 1 (deep sleep); 2 (sleepy/intentional eye closure), 3 (calm/neutral/awake); 4 (wide-awake); and 5 (hyper-active). LoE were measured as: 1 (unengaged); 2 (engaged 25%); 3 (engaged 50%); 4 (engaged 75%); 5 (engaged 100%). In addition, pictures accompanied each Likert demarcation for added clarity of the descriptions.

In the Introductory Phase, a Proximo-distal, hierarchical arrangement of the sensory protocol was performed on both upper and lower extremities at one session.  The hierarchical arrangement included, in order: modified milking massage (15 repetitions on every extremity), modified Wilbarger brushing (unidirectional, from proximal to distal, 15 repetitions on every extremity with moderate pressure), localized extremity joint vibration with battery-operated 4 prong Homemedics handheld massager (Proximal to distal: UE shoulder, elbow, wrist; LE hip, knee, ankle, 15 counts per joint), modified auditory integration program (50% volume on source, Sound Health Series mix, Sennheiser Closed 202 headphones). Each sensory diet lasted between 15 to 30 minutes in total length.  Each part of the diets was labeled and defined to the subject by provider prior, during and after the diet was completed. This lasted for the first 3 months.

The Second Phase was a division of the Introductory Phase into extremity-specific sessions. One 15- 30-minute session would be dedicated to the Upper Extremity protocol; another 15-30 minute session would be dedicated to the Lower Extremity protocol. Both types would use the modified Auditory Integration Therapy respectively. Diets lasted between 15 to 30 minutes in total length, and these were repeated up to three (3) times a day respectively. Diets were labeled and defined by provider prior, during and after the diet was completed. This was initiated immediately after the Introductory Phase, the next 3 months.

The Third Phase required Subject determination and use of communication strategies embedded in her AAC device. The provider would inquire what body extremity (UE/LE or R/L), and what specific diet was preferred. Diets lasted between 15 to 30 minutes in total length, and these were repeated up to five (5) times a day respectively.  Diets were labeled and defined by Subject choice prior, during and after the diet was completed. This came immediately after the Second Phase, the last 3 months.

Direct Instruction coupled with aspects of strategy-instruction was utilized during individual/small group instruction. The aspects of strategy-instruction included verbal modeling, questioning, and verbal reminders to use support strategies. When subject mastered content with at least 80% accuracy over five consecutive sessions, new topics were introduced. Previous skills were consistently revisited during the first five minutes of lessons as well as incorporated into lessons when applicable to the new skill being taught. Subject’s academic program took place five days a week and consisted of two-three formal 30-minute individual or small group  (2-3 Subjects) periods, and two formal 30-minute whole group (6 Subjects) periods over the course of 480 minutes per day for 12 months.

Post-Test academic achievement and sensory processing was taken one year after baseline. Post-Test academic achievement was taken again via an independent psycho-educational testing and modified Brigance testing which was completed in October 2014. Brigance testing included: knows personal information, Identifies parts of the body, word recognition grade placement test, counts by rote, reading comprehension grade placement, recognizes quantity, understands qualitative concepts, identifies common signs, reads words from common signs, recognizes quantity, adds numbers, subtracts numbers, multiplies numbers, divides numbers. Subject responded to both assessments utilizing a combination of AAC device, open and close verbal questioning and a touch screen device.  A case study by Kirsch, et al (2004) concluded, “ATC interventions can facilitate functional performance and contribute to learning of specific adaptive skills. Wireless, interactive, Web-based interventions appear particularly suited to tasks in the home and community, permitting remote intervention and monitoring of task status.”

Results
At baseline, the subject presented with aversion to the diet protocol (0% for both levels of engagement and alertness). The subject required maximal technical assistance for 2 months (October-November 2013) prior to responding positively to the diet protocol; the protocol was narrowed from a 7 sense to a 5 sense during this time period to accommodate the subject’s sensory processing needs as recorded by the technician on the LoA and LoE tracking sheets. From December 2013 to January 2014, a segmented protocol was introduced, also known as phase-two, to isolate sense stimulation with the head and extremity stimulation. From February 2014-May 2014, the subject continued with phase 2 and the technician recorded trends of responses.

It was also noted at baseline that the subject tested at a pre-kindergarten level for word recognition and kindergarten level for computation. Results corroborated with the independent psycho-education evaluation.

Evidence from assessments demonstrated a positive correlation between increased and consistent levels of alertness and engagement during improvements in segmented diets and academic achievement.

Post-Testing Sensory Diet Tracking results in May-June 2014 showed the subject to be alert at level 3 (calm/neutral/awake) for 86.7% and at level 2 (sleepy, intentional eye-closure) for 13.3% of the recorded sessions. The tracking sheets also showed the subject to be engaged at level 4 (engaged for 75% of the activity) for 68.3%, at level 3 (engaged for 50% of the activity) for 26.7% and at a level 2(engaged for 25% of the activity) for 5% of the recorded sessions. This is in comparison to the results from baseline upon introduction of the protocol for which the subject responded aversively and therefore was reported to be at 0%.

In addition, post-testing cognitive results via the modified Brigance testing had subject at a 3rd grade level on word recognition and computation (addition, subtraction, multiplication and division), in comparison to subject’s baseline testing which was respectively at a Pre-Kindergarten and Kindergarten level.

Conclusion
Based on the results, this case study shows a positive correlation between increased sensory processing alertness/engagement and increased academic achievement when sensory diets are administered. It is showing a significant change in percentage of optimal alertness and engagement for this 14-year old with neuro-developmental disorder specifically ESES Subject who displayed an 87% LoA (wide awake) and 68% LoE (engaged 75%) in the sensory diet protocol with an improvement in performance in modified Brigance testing from a Pre-K work recognition to 3^rd grade, and K Comp to 3^rd grade. This reveals a 4 and 3-grade level jump, respectively, over the course of one year. This case study validates the hypothesis that increased sensory processing alertness/engagement plays a part in increasing academic achievement in an adolescent female with neuro-developmental disorder specifically ESES.

This study utilized a single-subject design, which strengthens the internal validity of the study; however decreases the external validity of the study as it is not generalizable to other individuals within this population as it only pertains to the change evidenced with this particular subject. Furthermore, it needs to be noted that additional traditional therapies, including physical therapy, occupational therapy, speech and language therapy including augmentative and alternative communication therapy as well as modified conductive education, a program designed for children with motor dysfunction, which integrates education and rehabilitation into one program with the goal of achieving ortho-function–the capacity of individuals to respond to biological and social demands (Darrah, 2003)–were performed throughout the Subject’s day, which decreases the internal validity of the study as these interventions were not controlled for and may have played a role in her overall increase in academic achievement.

However, the results of this study indicate that a comorbidity of SPD and subsequent interventions for SPD need to be further explored for individuals with ABI as a way to increase sensory processing and academic achievement. Further work on the efficacy of sensory processing skills needs to be studied within a clinical based setting to determine if the benefits are generalizable to other children within this population as well other populations. Future studies should control for factors including but not limited to therapeutic interventions and duration of interventions on a daily basis.

Finally, according to a recent policy statement put forth by the American Academy of Pediatrics, “the amount of research regarding the effectiveness of sensory integration therapy is limited and inconclusive,” (2015). Therefore, more clinical based research regarding SPD as a distinct diagnosis and as a co-morbidity with the ABI population needs to be conducted in clinical based settings in order to address the controversy and limitations surrounding diagnosis and interventions for SPD either as a distinct or co-morbid diagnosis. This case study and the few that proceeded serve as first step in a relatively unexplored field that stands to yield significant benefits for both individuals with and without ABI.

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