Possible association between Sever’s disease and attention deficit hyperactivity disorder. a prospective observational study

Abstract

Background

Sever’s disease is among the most common causes of foot pain in childhood.The etiology of Sever’s disease is still under debate.The objective of this prospective study is to investigate the relationship between Sever’s disease and attention deficit hyperactivity disorder(ADHD).

Methods

The study was prospectively conducted for 1 year between 2023–2024.Of 238 children admitted to our clinic with foot pain, 88 children diagnosed with Sever’s disease were included in the study.A control group including 90 individuals was employed.Patients diagnosed with Sever’s disease in the orthopedics outpatient clinic and healthy control group were evaluated by a pediatric psychiatrist in terms of ADHD based on the DSM-4 diagnostic criteria.

Results

Fifty-seven patients were male, and 31 were female.While 45 male patients were diagnosed with ADHD, 12 of them did not have ADHD.While 17 female patients were diagnosed with ADHD, 14 did not have ADHD. ADHD was detected in 12 (13.3%) of 90 patients in the control group, comprising 9 boys and 3 girls. The prevalence of ADHD in the Sever’s disease cohort was 62 (70.5%) out of 88, significantly higher than the control group’s 12 (13.3%) out of 90 patients (p < 0.01). The high number of the patients diagnosed with ADHD among the Sever’s patients who were not engaged in sports was found to be statistically significant(p < 0.01).The diagnosis of ADHD was found to be statistically more significant in boys with Sever’s disease compared to girls(p < 0.018).

Conclusion

Children with Sever’s disease can be the candidates for attention deficit hyperactivity disorder.It is thought that evaluating children, especially male children, diagnosed with calcaneal apophysitis within the framework of attention deficit hyperactivity disorder will be valuable.

Introduction

Calcaneal apophysitis, also known as Sever’s disease, is among the common causes of foot pain in childhood [1]. In the literature, it is known as overuse injury that is common in young athletes. Symptoms often become worse by increased sportive activity [2]. Sever’s disease is more commonly seen in girls aged 7–9 and boys aged 8–15. Its incidence peaks between the ages of 10 and 12 [3]. Although many theories have been put forward to reveal the underlying risk factors and etiology, the common ground of many of them is that it is apophysis damage caused by repetitive micro traumas due to physical activity [3–5]. The most important treatment method is to restrict activity [6]. Many patients with calcaneal apophysitis, who are not active athletes, visit our clinic. Most families state that their children are very active. ADHD, which has an incidence of 3–5% in school age, is characterized by neurobehavioral disorder, attention deficit and hyperactivity [7, 8]. This study aims to evaluate whether there is a relationship between ADHD and Sever’s disease.

Materials and methods

Our study was prospectively conducted for one year between January 2023 and January 2024 after obtaining approval of the local ethics committee. Patients admitted to our clinic with hind foot pain and diagnosed with calcaneal apophysitis were included in the study. A control group including 90 individuals was employed. Written consents of the families were also obtained.

Study population

The diagnosis of Sever’s disease was established based on localized pain in the calcaneal apophysis which worsened with activity in a skeletally immature patient. Patient’s feeling of pain in calcaneal apophysis with pressure in detailed physical examination is typical in the diagnosis of Sever’s disease [9–11]. In all patients, ankle radiographs were used to confirm the clinical status and to exclude other ankle pathologies. Irregular ossification or damage, thinning and disintegration of the calcaneal apophysis and soft tissue swelling are among the typical radiographic findings. After the diagnosis was made, the patients were referred to the Child and adolescent psychiatry clinic.

Psychiatric assessment

Parents of the children with this disease mentioned that their children were very active. For this reason, patients diagnosed with Sever’s disease and their parents were evaluated by a pediatric psychiatrist. For diagnosis, ADHD was evaluated based on DSM-IV-TR diagnostic criteria. With this evaluation, pediatric patients were predominantly categorized as predominant intention deficit, predominant hyperactivity and combined type accompanied by both conditions.

Moreover, as the demographic data of the patients, age, gender, predominant type, duration of symptoms and their participation in sportive activities were examined. Children with participation in sports were required to be a member of a school team or a professional team that exercised at least twice a week.

Statistical analysis

Whether the numerical variables showed normal distribution was tested with Shapiro-Wilk test. In addition, Mann-Whitney U test was used to compare non-normally distributed variables between two groups. Relationships between categorical variables were tested with Chi-square and Bonferroni test. SPSS 22.0 Windows version package program was used in the analyses. P < 0.05 was considered to be significant.

Results

Eighty-eight patients diagnosed with Sever’s disease and whose consents were obtained included in the study among 238 pediatric patients admitted to our clinic with foot pain. Fifty-seven of the children included in the study were male, while 31 were female. Foot pain was on the right in 32 patients, left in 27 patients and bilateral in 29 patients. Only 18 of these patients were engaged in sports. While 62 of the patients were diagnosed with ADHD by a pediatric adolescent and mental health specialist, 27 of the patients had combined type, 7 had predominant attention deficit and 28 patients had predominant hyperactivity (Table 1).

Table 1.

Demographic data of the patients

		n	%
Gender	Male	57	64.8
	Female	31	35.2
ADHD	Yes	62	70.5
	No	26	29.5
Subtype	Combined type	27	43.5
	Predominant attention deficit	7	11.3
	Predominant hyperactivity	28	4.2
Subthreshold ADHD	Yes	10	38.5
	No	16	61.5
Side	Right	32	36.4
	Left	27	30.7
	Bilateral	29	33.0
Sportive activity	Yes	18	20.5
	No	70	79.5

ADHD was detected in 12 (13.3%) of 90 patients in the control group, comprising 9 boys and 3 girls. The mean age of the children was calculated as 10.24 ± 1.36. In the control group, the prevalence of ADHD was higher in males (75%). The prevalence of ADHD in the Sever’s disease cohort was 62 (70.5%) out of 88, which is significantly higher than the control group’s 12 (13.3%) of 90 patients (p < 0.01).

While the mean age of the patients was calculated as 9.86 ± 1.98, the mean symptom duration at the time of admission was 6.26 ± 2.47 months. The statistical analysis revealed that ADHD was significantly higher in boys in Sever’s disease than girls (p < 0.018) (Table 2).

Table 2.

Relationship between ADHD, sportive activity and gender of the patients

		ADHD
		Yes	No
		n(%)	n(%)	p
Gender	Male	45 (72.6)	12 (46.2)	0.018*
	Female	17 (27.4)	14 (53.8)
Sportive activity	Yes	5 (8.1)	13 (50)	0.001*
	No	57 (91.9)	13 (50)

Furthermore, 5 (8.1%) of the children with Sever’s disease who were diagnosed with ADHD did sports, while 57 (91.9%) did not do sports, 13 (50%) of the children who were not diagnosed with ADHD did sports, while 13 (50%) did not do sports (Table 2).

Discussion

Calcaneal apophysitis is among the most common causes of foot pain in childhood and adolescence and accounts for 8% of all pediatric overuse injuries [12, 13]. Children who engage in sports that require jumping, running and plantar flexion activation (such as football, gymnastics, dance, athletics and basketball) have been found to be at the highest risk in terms of developing calcaneal apophysitis [12–15]. Athletic activity, spending too much time standing and excessive mobility have been identified as important risk factors for this disease [16].

In their study conducted on calcaneal apophysitis, Ceylan H. H. et al. found that 59 of 74 patients were male, the mean age was 10.77 (6.87–15.73), and symptoms were bilateral in 46 (62.16%) patients [17]. In their study, Alicia M. James reported that 72 (58%) of 133 patients were male, the mean age was 10.88, and the symptoms were bilateral in 106 patients [16]. In accordance with the literature, 57 (64.8%) of 88 patients were male in our study. In addition, the mean age was 9.86 ± 1.98 years, and symptoms were bilateral in 29 (33%) patients.

In the literature, there are many studies suggesting that Sever’s disease is an overuse injury [18–20]. In a study conducted by Nicolò Martinelli on 430 athletic children, it was revealed that Sever’s disease was more common in children with sportive activity than the normal population [20]. Similarly, Patrick Belikan et al. mentioned the frequency of Sever’s disease in young athletes [6]. The literature reports that Sever’s disease can be an overuse injury and is the biggest risk factor in the etiology of sportive activity. Hyperactivity may not always be associated with sports, but it can be associated with ADHD. Our study aimed to bring a new etiological perspective to the etiology of Sever’s disease.

The incidence of ADHD in the general population is between 3 and 5% [7, 21]. Recent studies have revealed that acute injuries of children may be associated with ADHD [22–24]. In the study conducted by Shen J. et al., it was reported that patients diagnosed with ADHD in the United States between 2011 and 2020 had twice the increase in emergency admission and health expenditures compared to the normal population [25]. Similarly, in a study conducted by Ziv-Baran T. et al., a 1.2-fold increase was found in the probability of foot fracture in children diagnosed with ADHD compared to the normal population [26]. Children diagnosed with ADHD are prone to chronic recurrent injuries as well as being exposed to acute traumatic injuries. In our study, the incidence of ADHD was found to be significantly higher compared to the healthy control group.

In the literature, there is a study comparing behavioral disorder in children with Osgood-Schlatter disease, which is one of the osteochondroses [27]. Our study is the first study investigating the coexistence of Sever’s disease and behavioral disorder which is also an osteochondrosis. It is thought that there is an overuse injury with an insidious onset of pain in the etiopathogenesis of Sever’s disease, and complaints increase as a result of stress and pressure in the calcaneal apophysis [9, 28]. Patients diagnosed with Sever’s disease were included in our study, and 70.5% of these patients were diagnosed with ADHD.

It is thought that attention deficit hyperactivity disorder may play a role in the etiopathogenesis of Sever’s disease since children with combined type and predominant hyperactivity are physically very active.

The study has some limitations. The number of the patients participating in the study is low. An assessment of the prevalence of Sever’s disease among subgroups of ADHD patients has not been conducted; further studies may be undertaken with larger patient cohorts. The strengths of our study are that it is prospective and the diagnosis of ADHD is established by a child and adolescent psychiatry.

Conclusion

Children with Sever’s disease can be the candidates for attention deficit hyperactivity disorder. As a result, it can be stated that it would be beneficial to evaluate it in terms of attention deficit and hyperactivity disorder in patients diagnosed with calcaneal apophysitis who are not engaged in sports and especially male children.

Author contributions

All authors contributed to the study conception and design. Nevzat Gonder.: study design, performed measurements, manuscript preparation; Ibrahim Halil Demir: study design; Volkan Ozel.: study design, manuscript preparation; Kubra ılicepınar.: performed measurements; Musa Alperen Bilgin.: study design, manuscript preparation.

Funding

There is no funding source.

Data availability

No datasets were generated or analysed during the current study.

Declarations

Ethics approval

This study was performed in line with the principles of the Declaration of Helsinki. Approval was granted by the Local Ethics Committee (Date 2023/No. 2023-2ÖNP-0156).

Competing interests

The authors declare no competing interests.