Histopathological Confirmation of Axonal Sprouting in Regenerative Peripheral Nerve Interface.

Symptomatic neuroma represents a debilitating complication after major limb amputation. The regenerative peripheral nerve interface (RPNI) has emerged as a reproducible and practical surgery aimed at mitigating the formation of painful neuroma. Although previous animal studies revealed axonal sprouting, elongation, and synaptogenesis of proximal nerve stump within the muscle graft in RPNI, there is a lack of reports confirming these physiological reactions at the histopathological level in human samples. This report presents a case of below-knee amputation with RPNI due to foot gangrene resulting from polyarteritis nodosa. Subsequently, an above-knee amputation was necessitated due to the exacerbation of polyarteritis nodosa, providing the opportunity for histopathological examination of the RPNI site. The examination revealed sprouting, elongation, and existence of neuromuscular junction of the tibial nerve within the grafted muscle. To the best of our knowledge, this is the first report demonstrating axonal sprouting, elongation, and possibility of synaptogenesis of the nerve stump within the grafted muscle in a human sample.

Motion Capture-based 3-dimensional Measurement of Range of Motion in Patients Undergoing Cervical Laminoplasty.

STUDY DESIGN: A prospective study. OBJECTIVE: To measure 3-dimensional cervical range of motion (ROM) by noninvasive optical tracking-based motion-capture technology in patients undergoing laminoplasty, and to elucidate the postoperative effects of laminoplasty on cervical mobility. SUMMARY OF BACKGROUND DATA: Cervical laminoplasty is a motion-sparing decompression surgery for degenerative cervical myelopathy. Unlike cervical laminectomy and fusion, the true postoperative impact of laminoplasty on neck motion has not been well studied. METHODS: Participants comprised 25 patients undergoing double-door cervical laminoplasty for degenerative cervical myelopathy in a single center. Maximum flexion/extension, left/right rotation, and left/right side bending were recorded using the motion-capture device preoperatively and 3 months postoperatively. ROMs in 3 orthogonal axes were calculated. Preoperative differences in C2-7 Cobb angles on lateral flexion/extension x-rays were also measured as the radiologic ROM to assess reliability. Preoperative and 1-year postoperative Japanese Orthopaedic Association score, Neck Disability Index [NDI], and Euro-QOL were recorded, and correlations with ROMs were assessed. RESULTS: Preoperative mean (±SD) ROMs for flexion/extension, rotation, and side bending were 90±17, 107±16, and 53±17 degrees, respectively. Although radiologic sagittal ROM measurement showed a smaller range than motion capture, averaging 36±13 degrees, a moderate to strong correlation between radiologic and motion capture values was observed (R=0.57, P=0.003). Preoperative NDI showed a negative correlation with coronal ROM (rho=-0.547, P=0.02). Postoperative ROM showed a significant reduction in rotation (95±16 degrees, P=0.002) but not in flexion/extension or side bending. CONCLUSIONS: Three-dimensional motion-capture analysis allowed reliable measurement of cervical ROM. Rotational ROM was significantly reduced after laminoplasty, showing that cervical kinematics are still significantly altered.

Factors Associated With a Lateral Ankle Sprain in Young Female Soccer Players: A Prospective Cohort Study.

Background: Previous studies have attempted to determine if certain risk factors can predict the occurrence of a lateral ankle sprain (LAS) in female soccer players. Unfortunately, there is limited evidence with regard to risk factors associated with an LAS in female soccer players. Purpose: To identify intrinsic risk factors for an LAS among young female soccer players. Study Design: Cohort study; Level of evidence, 2. Methods: Participants were 161 young female soccer players in Japan who were evaluated for LAS risk factors during a preseason medical assessment. The assessment included anthropometric, joint laxity, joint range of motion, muscle flexibility, muscle strength, and balance measurements. Each athlete's history of LASs was also collected. The participants were monitored during a single-yearseason for LASs, as diagnosed by physicians. Results: There were 26 instances of an LAS in 25 players (15.5%) during the season. Injured players were significantly more likely to have sustained a previous ankle sprain (P = .045) and demonstrated significantly worse balance than their peers without an LAS during the double- and single-leg balance tests (P = .008 for both). Athletes with lower hamstring-to-quadriceps muscle strength ratios were also significantly more likely to sustain an LAS (P = .02). Conclusion: Poor balance, a low hamstring-to-quadriceps ratio, and a history of ankle sprains were associated with an increased risk of LASs in young female soccer players in the current study. These findings may be useful for developing a program to prevent LASs in this population.

Lower hamstring to quadriceps muscle strength ratio and lower body weight as factors associated with noncontact anterior cruciate ligament injury in male American football players: A prospective cohort study.

Background: Previous studies have aimed to determine the use of certain risk factors in predicting the occurrence of noncontact anterior cruciate ligament (ACL) injuries. Unfortunately, evidence regarding noncontact ACL injuries in male American football players is limited. This prospective cohort study aimed to identify intrinsic risk factors for noncontact ACL injury among male American football players. Methods: This study evaluated 152 male American football players in Japan for potential noncontact ACL injury risk factors during a preseason medical assessment, including anthropometric, joint laxity, and flexibility, muscle flexibility, muscle strength, and balance measurements. A total of 25 variables were examined. Participants were monitored during each season for noncontact ACL injury, as diagnosed by physicians. Results: Noncontact ACL injuries occurred in 11 knees of 11 players (prevalence; 7.1 %). Injured players were significantly more likely to have lightweight (P = 0.049). No statistically significant between-group differences were found for any other variables. Participants with a lower hamstring to quadriceps (H/Q) ratio (P = 0.04) were more likely to sustain noncontact ACL injuries. Conclusion: Lower H/Q ratio and lower body weight were significantly associated with new-onset noncontact ACL injury in male American football players. These findings will help develop strategies to prevent noncontact ACL injuries in male American football players.

Effect of dual-task interaction combining postural and visual perturbations on cortical activity and postural control ability.

Previous studies have suggested cortical involvement in postural control in humans by measuring cortical activities and conducting dual-task paradigms. In dual-task paradigms, task performance deteriorates and can be facilitated in specific dual-task settings. Theoretical frameworks explaining these dual-task interactions have been proposed and debated for decades. Therefore, we investigated postural control performance under different visual conditions using a virtual reality system, simultaneously measuring cortical activities with a functional near-infrared spectroscopy system. Twenty-four healthy participants were included in this study. Postural stability and cortical activities after perturbations were measured under several conditions consisting of postural and visual perturbations. The results showed that concurrent visual and postural perturbations could facilitate cortical activities in the supplementary motor area and superior parietal lobe. Additionally, visual distractors deteriorated postural control ability and cortical activation of the supplementary motor area. These findings supported the theoretical framework of the "Cross talk model", in which concurrent tasks using similar neural domains can facilitate these task performances. Furthermore, it indicated that the cortical resource capacity and domains activated for information processing should be considered in experiments involving dual-task paradigms and training.

Risk factors for hamstring strain injury in male college American football players -a preliminary prospective cohort study.

BACKGROUND: Given the frequency of hamstring strain injuries (HSI) among male college American football players, several studies have attempted to determine whether certain risk factors can predict their occurrence. However, no consensus on modifiable risk factors for HSIs in male college American football players has yet been reached to prevent these injuries. This study aimed to clarify risk factors for HSI prospectively in college male American football players. METHODS: A total of 78 male college American football players, whose positions were limited to skill positions, were medically assessed for potential risk factors of HSI. The preseason medical assessment included anthropometric measurements, joint laxity and flexibility, muscle flexibility, muscle strength, and balance ability. RESULTS: HSI occurred in a total of 25 thighs from 25 players (32.1%). Injured players had significantly lower hamstring flexibility (p = 0.02) and hamstring to quadriceps strength ratio (H/Q) (p = 0.047) compared to uninjured players. Additionally, injured players had significantly lower general joint laxity scores, especially for the total (p = 0.04), hip (p = 0.007), and elbow (p = 0.04) scores, compared to uninjured players. CONCLUSIONS: Lower hamstring flexibility, lower hamstring to quadriceps strength ratio, and lower general joint laxity score were identified as risk factors for HSI in male college American football players placed in skill positions. The muscle flexibility and H/Q ratio could be useful in preventing HSI in such players.

Intellectual Characteristics in Children With Congenital Unilateral Upper Limb Deficiencies.

Introduction Some children with motor disabilities show low cognitive levels. However, the influence of motor disabilities on children's intelligence remains to be fully elucidated. This study aimed to clarify the intellectual characteristics of children with upper limb deficiencies and the influence of upper limb impairments on intelligence. Methods The participants were 10 children from four to six years of age with congenital unilateral transradial or transcarpal limb deficiencies who received prosthetic interventions. The children's intelligence and adaptive behaviors, including motor skills, were examined using the Wechsler Preschool and Primary Scale of Intelligence and the Vineland Adaptive Behavior Scale, respectively. Results There were no significant characteristics or discrepancies in cognitive level in children with upper limb deficiencies. The Adaptive Behavior Composite Score of the Vineland Adaptive Behavior Scale was significantly positively correlated with the Full-Scale Intelligence Quotient of the Wechsler Preschool and Primary Scale of Intelligence. Conclusions The children with congenital limb deficiencies showed average cognitive levels. Expansion of adaptive behaviors, including appropriate complementation of disabilities, may promote intellectual development in children with motor disabilities.

Relationship between degree of disability, usefulness of assistive devices, and daily use duration: an investigation in children with congenital upper limb deficiencies who use upper limb prostheses.

Upper limb prostheses can help children with congenital upper limb deficiencies (ULDs) perform activities of daily living. Although the degree of disability and prosthesis usefulness may be related to becoming accustomed to wearing a prosthesis, these relationships have not been confirmed. This study was aimed at investigating the relationship between motor function, the usefulness of prostheses in everyday activities, and daily duration of prosthesis use in children with congenital ULDs. Eleven children with congenital transcarpal or transradial ULDs and their caregivers participated in this study. The caregivers were asked to complete a questionnaire concerning their children's everyday activities and the daily duration of prosthesis wearing. The adaptive behavior scale was used to measure motor function prior to intervention. Correlations analyses revealed no significant correlation between the children's adaptive behavior, including motor skills before intervention, and daily duration of prosthesis wearing, but revealed a positive correlation between the number of activities that the children performed more effectively with their prostheses and the daily use duration. To ensure that children with congenital ULDs become accustomed to wearing their prostheses, the prostheses should meet the requirements of everyday activities.

Dynamic Postural Stability Is Decreased During the Single-Leg Drop Landing Task in Male Collegiate Soccer Players With Chronic Ankle Instability.

Background: Chronic ankle instability (CAI) is commonly encountered in soccer players. The feelings of instability and anxiety caused by CAI can lead to poor performance, such as difficulty in sharp change of direction during soccer play. The single-leg drop landing (SLDL) task is often used to evaluate dynamic postural stability. Purpose/Hypothesis: The purpose of this study was to clarify whether dynamic stability measured during SLDL is altered in male collegiate soccer players with CAI. The hypothesis was that athletes with CAI would show poor dynamic postural stability. Study Design: Controlled laboratory study. Methods: A total of 103 male collegiate soccer players were recruited, and their limbs were classified based on the new international CAI criteria. All players performed three 5-second SLDL trials on a force plate. The main outcome measures included time to stabilization of the horizontal ground-reaction force (GRF); peak GRF in the vertical, horizontal, and sagittal directions; and trajectory length of the center of pressure during SLDL. Results: Data from 59 CAI limbs and 147 non-CAI limbs were collected in this study. Time to stabilization of horizontal GRF was significantly longer in the CAI limbs (P < .001), and the peak GRFs in all directions were significantly lower in the CAI limbs (vertical, P < .001; horizontal, P < .001; sagittal, P = .001). Additionally, the trajectory length of the center of pressure was significantly greater in the CAI limbs (P = .004). Conclusion: Soccer players with CAI had decreased dynamic postural stability that led them to land softly when performing the SLDL task. Measurement of dynamic postural stability may be useful in the evaluation of CAI. Clinical Relevance: Our findings may be useful for strategies of daily training or as an evaluation tool.

Kinematic Differences Between the Dominant and Nondominant Legs During a Single-Leg Drop Vertical Jump in Female Soccer Players.

BACKGROUND: In soccer, the roles of the dominant (kicking) and nondominant (supporting) legs are different. The kinematic differences between the actions of the dominant and nondominant legs in female soccer players are not clear. PURPOSE: To clarify the kinematic differences between dominant and nondominant legs during a single-leg drop vertical jump (DVJ) in female soccer players. STUDY DESIGN: Controlled laboratory study. METHODS: A total of 64 female high school and college soccer players were included in this study. Participants performed a single-leg DVJ test utilizing video motion capture with artificial intelligence during the preseason period. This study assessed the knee flexion angles, knee valgus angles, hip flexion angles, and lower leg anterior inclination angle at 3 time points (initial contact, maximum flexion of the knee, and toe-off) and compared them between the dominant and nondominant legs. These angles were calculated from motion capture data and analyzed in 3 dimensions. A paired t test was used to analyze the differences between legs, and the significance level was set at P < .05. RESULTS: The knee valgus angle at initial contact was greater in the nondominant leg (mean ± SD, 0.8°± 5.2°) than the dominant leg (-0.9°± 4.9°) (P < .01). There were no differences between legs for any other angles at any of the time points. CONCLUSION: The kinematics of the dominant and nondominant legs of female soccer players in a single-leg DVJ differ in knee valgus angle. CLINICAL RELEVANCE: Leg dominance is associated with the risk of sports injuries. Kinematic differences between the dominant and nondominant legs may be a noteworthy factor in elucidating the mechanisms and risk of sports injury associated with leg dominance.

Inconvenience and adaptation in Japanese adult achondroplasia and hypochondroplasia: A cross-sectional study.

The health-related quality of life is reduced in patients with achondroplasia (ACH) and hypochondroplasia (HCH); however, the detailed inconveniences in the daily living and individual adaptations have not been elucidated. This study aimed to evaluate the inconvenience and adaptation in patients with ACH/HCH. A cross-sectional study was conducted in patients with ACH/HCH aged 20 yr or older. Questionnaires were sent to 567 patients (described 86) with a medical history at the co-authors' institutions or who were registered at the patients' association with ACH in Japan. The questionnaire included a free description format for the inconveniences and adaptations in daily living; a content analysis was performed. The recorded inconveniences included 148 physical, 84 mental, and 52 social problems. Patients who underwent spine surgery had significantly more recorded physical problems than those who did not (p < 0.05). Pain and numbness were significantly higher in patients aged ≥ 50 yr (p < 0.05). The 160 and 1 adaptations were for physical and social problems, respectively. No patient adaptation was found for mental health problems. Individual adaptations by ACH/HCH patients can improve only some aspects of physical and social problems. Multilateral social support is needed to resolve patients' issues.

Body knowledge in children with spina bifida.

BACKGROUND: Patients with spina bifida suffer from motor paralysis and sensory disturbance, secondary deformation of the lower extremities, and development of decubitus ulcers. A deep understanding of one's body, such as identifying the names, functions, relationships, homology (e.g. upper and lower limbs) and relative position of body parts, may prevent secondary disorders. The awareness of the body and its characteristics in children with spina bifida has not been fully elucidated. This study aimed to investigate how children with spina bifida perceive their bodies, especially their paralyzed parts and related or homologous ones. METHODS: Participants included 36 children with spina bifida and 14 control children, aged 5-11 years. They were asked to draw self-portraits, and to answer questions about the names of body parts. The number of drawn body parts in the pictures and the correct response rates to the questions were quantified and compared between children with spina bifida and control children. RESULTS: Children with spina bifida drew fewer hands, legs, and feet than control children. In the verbal tests, children with spina bifida had a lower correct response rate than control children on questions concerning the upper limbs, hands, trunk, and feet. CONCLUSIONS: Children with spina bifida have diminished visuospatial and lexical-semantic body knowledge of the paralyzed parts and related organs.

Visual attention to their own paralytic limbs in children with spina bifida: Measurement of gaze direction using eye tracking.

BACKGROUND: Patients with spina bifida experience sensory and motor paralysis and complications in the form of deformation and skin problems of the lower limbs. Enhancing their awareness of the paralysed lesions could be helpful for these patients to prevent secondary disorders. This study sought to investigate to what extent children with spina bifida are visually aware of their body parts and, in particular, to their paralysed lesions. METHODS: Participants included children with spina bifida (n = 10) and typically developing control children (n = 10) between the ages of 5 and 11 years. All participants were shown still images of themselves on a monitor after which their gaze direction was measured using an eye tracker, with or without providing an instruction to direct their attention to their limbs. The gaze direction data were analyzed and compared between children with spina bifida and the control children. RESULTS: Children with spina bifida paid visual attention to their bodies in a manner similar to that of control children. Prompts to direct their attention to their limbs were effective in both groups; however, the degree of efficacy in children with spina bifida was inferior to that in control children. CONCLUSIONS: Promotion of visual awareness to their paralytic parts could be a reasonable and effective method for children with spina bifida to improve their understanding and recognition of their body and prevent associated skin problems.

Health-related Quality of Life in Adult Patients with Multiple Epiphyseal Dysplasia and Spondyloepiphyseal Dysplasia.

OBJECTIVES: Multiple epiphyseal dysplasia (MED) and spondyloepiphyseal dysplasia (SED) are skeletal dysplasias associated with premature osteoarthritis and short stature. Patients with SED often have spinal and ocular problems. Few reports have focused on the health-related quality of life (HRQoL) of patients with skeletal dysplasias associated with premature osteoarthritis. The purpose of this study was to evaluate the HRQoL of adult patients with MED and SED. METHODS: Questionnaires covering demographics, medical history (cataract, retinal detachment, and osteoarthritis), surgical history (osteotomy and arthroplasty), and the Short Form-36 (SF-36) health survey were sent to all patients with MED and SED with medical records at the investigators' institutions. Among the 27 patients who completed the questionnaire, patients aged 20 years or older were included in this cohort. RESULTS: The subjects were 18 affected individuals. The physical component summary score (PCS) was significantly lower in the MED and SED groups than in the normal population and tended to deteriorate with age. Conversely, there was a positive correlation between the mental component summary score and age. The role/social component summary score was not correlated with age. MED patients with osteoarthritis had a low PCS. PCS was particularly low in two SED patients with a medical history of cataract, whereas there was no association with a history of retinal detachment or osteoarthritis. CONCLUSIONS: The physical domain of HRQoL in MED and SED patients significantly deteriorated at a young age. Appropriate medical management of these skeletal dysplasias is required not only for orthopedic functions but also for ocular problems.

How Children with Congenital Limb Deficiencies Visually Attend to Their Limbs and Prostheses: Eye Tracking of Displayed Still Images and Visuospatial Body Knowledge.

Purpose: This study aimed to clarify how children with congenital limb deficiencies visually attend to their bodies, particularly their limbs and prostheses.Methods: Participants included children with and without congenital limb deficiencies. They were shown photographs of themselves and their visual attention was measured using an eye tracker.Results: Six children with lower limb deficiencies (age [mean ± SD]: 8.8 ± 2.9; 2 girls and 4 boys), six children with upper limb deficiencies (age: 7.0 ± 2.3; 2 girls and 4 boys), and ten control children (age: 7.7 ± 1.9; 5 girls and 5 boys) were included. Children with congenital upper/lower limb deficiencies looked at their upper/lower limbs as often or more than the control children. Prompts to direct their visual attention to their limbs had limited efficacy.Conclusions: To improve the body knowledge of limbs, approaches other than visual recognition prompting, such as improving linguistic understanding, might be considered.

Rehabilitation Approach for a Child with Cerebral Palsy and Upper Limb Deficiency.

BACKGROUND: Congenital limb deficiency is a rare and intractable anomaly of the limbs; however, prostheses can partially complement the motor function and appearance of the missing limbs. The first prosthesis is usually prescribed for children with upper limb deficiencies at approximately 6-8 months of age. In affected children with additional problems associated with motor function, such as limb paralysis, the age for initiating prosthetic therapy and the benefit of prostheses in promoting and expanding their motor function and activities is unknown. CASE: In this case presentation, we describe a 25-month-old boy with cerebral palsy and left unilateral congenital upper limb deficiency caused by congenital constriction band syndrome. The patient could stand with assistance and crawl on his hands and knees. However, he was unable to walk with assistance or to stand on his own. A forearm prosthesis with a passive hand was prescribed and issued, and rehabilitation therapy for wearing and using the prosthesis was performed. At 34 months of age, the patient was able to walk forward using a walker with the prosthesis. Without the prosthesis, he still could not walk using a walker. The upper limb prosthesis also improved other movements such as sitting, standing, and tasks performed on a desk or on the floor. DISCUSSION: The prosthesis was apparently effective in improving motor function. Prosthesis prescription should be considered at an appropriate and early age considering individual developmental stages and needs, regardless of the existence of additional problems associated with motor function.

Treatment approaches for congenital transverse limb deficiency: Data analysis from an epidemiological national survey in Japan.

BACKGROUND: Congenital limb deficiency is a rare anomaly that impairs limb function. Transverse deficiency accounts for approximately half of congenital limb deficiency cases. In Japan, there have been no detailed data of clinical features, especially treatment approaches, of this disorder. The present study aimed to investigate the status of treatment approaches of congenital transverse limb deficiency in Japan. METHODS: From the national epidemiological survey of congenital limb deficiency undertaken in Japan in 2016, all the data of 200 patients with congenital transverse limb deficiencies were extracted. These data were analysed to reveal the treatment approaches of congenital transverse limb deficiency and its basic clinical features. RESULTS: Surgical treatments and prosthetic or orthotic intervention had been implemented or planned for about one-quarter of patients, respectively. In the upper limb deficiencies, prosthetic or orthotic intervention was likely chosen in cases of deficiency at the metacarpal or proximal to metacarpal level. Surgical treatment was chosen only in cases of deficiency at the carpal or distal to carpal level. Although the number of patients with transverse lower limb deficiencies was small, prosthetic or orthotic intervention was likely chosen in proximal deficiencies, and surgical treatment was likely chosen in distal deficiencies. CONCLUSIONS: Herein, we revealed the status of treatment approaches for congenital transverse limb deficiency in Japan. Approximately half of the patients had no history of-and no plans for-surgical, prosthetic, or orthotic interventions. Further treatment advances may enable patients with congenital limb deficiencies to increase their participation in daily activities. STUDY DESIGN: Cross-sectional survey.

Reconstruction for Severe Extracorporeal Membrane Oxygenation-induced Ischemic Lower Limb Injury Complicated by Osteomyelitis.

Extracorporeal membrane oxygenation (ECMO) is a well-established mechanical circulatory support system used in patients with life-threatening cardiopulmonary conditions. However, severe complications associated with vascular access require consideration. We report a patient with fatal ventricular arrhythmia who was successfully resuscitated with ECMO but who developed severe lower limb ischemia, which resulted in compartment syndrome. Even with emergent fasciotomy, tissue necrosis developed in wide areas of the limb, with subsequent tibial osteomyelitis. After extensive debridement and tibial sequestrectomy, the soft tissue and bone defect were simultaneously reconstructed with free tissue transfer of the latissimus dorsi muscle and scapular tip composite flap. The limb was successfully salvaged with satisfactory functional outcomes without major complications. This report discusses limb reconstruction for ECMO-induced compartment syndrome and illustrates the importance of appropriate selection of reconstruction methods among various composite flaps.

Effect of prostheses on children with congenital upper limb deficiencies.

BACKGROUND: Individual weaknesses in motor skills are a characteristic of children with congenital upper limb deficiencies. These weaknesses increase with age. In Japan, however, prosthetic prescription and subsequent rehabilitation approaches for children with upper limb deficiencies are insufficient and often delayed. This study aimed to elucidate whether rehabilitation approaches, including prostheses prescription and occupational therapy, improve these children's adaptive behaviors, especially their motor skills. METHODS: The study included nine children, aged 0-6 years, with unilateral transradial or transcarpal upper limb deficiencies. We measured their adaptive behaviors and motor skills at the beginning of prosthetic therapy and after 1.5 years, using the Vineland Adaptive Behavior Scales - Second Edition. RESULTS: The score for the motor skills domain was significantly lower than the median score of the domains at the beginning of prosthetic therapy. The motor skill weaknesses significantly improved after 1.5 years of prosthetic therapy. CONCLUSIONS: Although children with congenital upper limb deficiencies have individual weaknesses in their motor skill behavior, it was shown that these weaknesses can be improved through rehabilitation approaches, including occupational and prosthetic therapies. Issuing the appropriate prostheses and implementing the appropriate training to use the prostheses for congenital upper limb deficiencies are reasonable and meaningful interventions to improve quality of life.

Body Knowledge in Children with Congenital Upper Limb Deficiency.

BACKGROUND: It is advantageous to effectively develop motor functions and a deep understanding of one's body (for example, relative positions, relationships, names, and functions of body parts). It has been reported that lexical-semantic knowledge of the defective body part is diminished in children with congenital lower limb deficiencies, and the features of body knowledge in children with congenital upper limb deficiencies (ULDs) have not been clarified. This study aimed to explore how children with ULDs perceive their bodies. METHODS: Participants included six children with congenital ULDs and 14 control children, aged 5-11 years. They drew self-portraits and answered questions about the names of body parts. RESULTS: Children with ULDs were significantly more likely to omit hands in their self-portraits than the control children. In the verbal tests, children with ULDs had a lower rate of correct responses concerning upper limbs, arms, hands, legs, and feet than the control children. CONCLUSION: Children with ULDs have diminished visuospatial body knowledge of the hands, as well as diminished lexical-semantic body knowledge of both the upper and lower limbs.