1 Introduction

The literature review indicates that lumbar spondylolysis and spondylolisthesis should be at the top of the differential diagnosis in children, adolescents, and young athletes suffering from low back pain. Various pathologies are included in the differential diagnosis, although the diagnostic investigation in most children and adolescents with low back pain may end with no definitive result. Some of them may represent serious abdominal, pelvic, and spinal pathology and spinal inflammatory arthritis such as rheumatoid arthritis (RA) and spondyloarthropathies (SpA).^1,2 In addition, inflammatory bowel diseases (IBD), including celiac disease (CD), in children, adolescents, and adults, can affect bone mineral metabolism through a pathogenetic process involving chronic inflammation and malabsorption or may be associated with arthritis classified as a subtype of seronegative SpA, with axial, peripheral or combined joint manifestations. Subsequently, CD may occasionally be symptomatic with pain referred to the lower back, either due to metabolic bone disease or inflammatory arthritis involving the lumbar spine and/or the sacroiliac joints.^3–5

The primary aim of this paper is to report a patient with spondylolytic spondylolisthesis who had been previously diagnosed with CD and to present her clinical and imaging findings. This article also provides a literature review for children and adolescents suffering from spondylolysis, spondylolisthesis, or both who were also diagnosed with CD, RA, or different types of SpA.

2 Case report

A 15-year-old female presented with an approximately 12-month history of mild low back pain; the latter most commonly occurred during sports or exercise and had worsened during the last few months. Her history indicated no acute traumatic injury or intense participation in athletic activities. She reported suffering from a CD.

3 Clinical findings

The physical examination revealed localized pain over the lower lumbar spine but no abnormal clinical signs from the hips and sacroiliac joints, radicular pain, or neurological deficit.

4 Diagnostic assessment

Plain radiographs of the pelvis and lumbar spine revealed spondylolysis with spondylolisthesis of the fifth lumbar (L5) over the first sacral (S1) vertebra, grade 2 per the Meyerding classification,⁶ an incomplete fusion of the posterior arch of L5, and a potential thoracic scoliotic curve (Fig. 1a). Further evaluation with magnetic resonance imaging (MRI) demonstrated low signal intensity on T1-weighted images and high signal intensity on T2-weighted SPectral Attenuated Inversion Recovery (SPAIR) sequences, indicative of bone marrow edema, in response to the bilateral fracture completely extending through the L5 pars interarticularis. On the supine MRI views, the amount of translation of L5 detected on the neutral-lateral standing radiograph was significantly reduced (Fig. 1b). The clinical and imaging findings indicated the diagnosis of acquired spondylolytic spondylolisthesis.

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Fig. 1a The frontal radiograph shows incomplete closure of the posterior neural arch of L5. The left lamina grows obliquely upward, and the right lamina grows obliquely downward. Although both laminae reach the midline, they cannot meet each other. A potential thoracic scoliotic curve and a Risser stage 4 sign are also evident. The lateral radiograph shows lucency in the region of the pars interarticularis of L5 associated with spondylolisthesis of L5 over S1.

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Fig. 1b T1-weighted parasagittal image revealed cortical disruption, a bilateral defect of the pars interarticularis, and reactive marrow changes in the posterior elements of L5, while T2-weighted SPAIR images indicated high signal intensity on both sides of the bilateral L5 pars defect.

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5 Literature review

The terms used for searching in all possible combinations were spondylolysis/spondylolisthesis, celiac/coeliac disease, and RA/SpA, as well as all the types of inflammatory diseases that SpA comprise, including ankylosing spondylitis (AS), psoriatic arthritis/spondylitis (PsA), reactive arthritis (ReA), arthritis/spondylitis associated with IBD and undifferentiated spondyloarthropathy (uSpA). Reports on patients older than 19 years and postsurgical, pathologic, and degenerative lumbar spondylolisthesis were excluded. The databases examined revealed no available studies or case reports.

6 Discussion

In primary care, low back pain is a common symptom among children and adolescents, while cases without evidence of a clear cause are hardly managed clinical problems.⁷

Both spondylolysis and spondylolisthesis may be asymptomatic and found incidentally on radiographs. In addition, diagnosis is often delayed or missed due to non-typical clinical presentation, especially in patients treated by non-orthopaedic health or spine care providers.⁸ Spondylolysis may be seen in up to 47 % of athletes presenting with back pain. Bilateral spondylolysis is more prone to be associated with symptoms than unilateral. Timely diagnosis facilitates early conservative treatment in children and adolescents with symptomatic spondylolysis and low-grade spondylolisthesis.^9,10

The imaging examination of the presented patient revealed three findings, which are also briefly discussed.a)Spina bifida occulta (SBO) is the most common lumbosacral dysplasia among young patients with low back pain. A very high rate of lumbar spondylolysis has been reported in spines with SBO in adults and pediatric patients.¹¹b)The relationship between scoliosis and spondylolisthesis is complex but well-documented. It has been stated that lumbar scoliosis, less than 15° Cobb in young men with spondylolysis or spondylolisthesis is not idiopathic.¹²c)In segmental instability, there is a reduction of the anterior slippage during supine imaging due to the elimination of the load. Axial-loaded MRI demonstrates a significantly higher degree of anterior translation, which is strongly correlated with that observed on upright radiographs than conventional MRI.¹³

Celiac disease is a complex immune response to gluten proteins from wheat, rye, and barley, but not oats.¹⁴ It is most likely related to abnormal mucosal permeability to unidentified provoking antigens, which may vary widely between individuals. Although it is classically considered a disease of early childhood, it can arise at any age with a broad spectrum of symptoms. Triggers of CD in adulthood are unknown.¹⁵ Clinical presentation of CD is highly variable, including classical or non-classical gastrointestinal symptoms, extraintestinal manifestations, and even subclinical or symptomless cases.¹⁶ Periodic diarrhea and malabsorption of various nutrients is a common presentation,¹⁷ unlike metabolic bone disease, which may present as rickets, osteomalacia, osteopenia, osteoporosis, and even pathological fractures.¹⁸ It must be differentiated from irritable bowel syndrome and non-celiac gluten sensitivity, which does not have the pathological findings seen in CD.¹⁹

Various common pathogenetic aspects have been previously reported, as well as an increased risk of co-occurrence and associated disease course of CD with RA and SpA. The involvement of the gut in the pathogenesis of RA and SpA, as in CD, by permitting the entrance of external factors, is based on previous evidence that ReA, formerly Reiter syndrome, is induced by specific urogenital or enterogenic bacteria. It is also evident, that the gut is implicated in IBD (Crohn's disease and ulcerative colitis) and even in RA. Furthermore, CD in adults is frequently associated with a high incidence of imaging sacroiliac changes, as in other chronic intestinal diseases. In addition, some authors have described a clinical presentation of CD with varying distribution of arthritis prominent at diagnosis or similar to seronegative SpA. In these patients, an associated autoimmune rheumatologic disease may be evident. Clinicians also need to be aware of the possible coexistence of CD in any juvenile seronegative SpA or other autoimmune diseases (mainly type 1 diabetes mellitus or thyroid disorders), which has important implications for the correct management of these patients. Finally, CD is more common in patients with juvenile idiopathic arthritis (JIA) than in the general population, indicating that CD screening is invaluable in all JIA children, especially those with a family history of autoimmunity.²⁰

In conclusion, this is the first reported patient with spondylolytic spondylolisthesis previously diagnosed with CD. Patients suffering from CD, as well as those with other inflammatory intestinal diseases, spinal inflammatory arthritis, or abdominal and pelvic pathologies, may present with a chief complaint of pain referred to the abdomen and/or the lower back. In all these patients, a lumbar spine coincidental pathology to the primary one may exist, and the potential diagnosis of spondylolysis, spondylolisthesis, or both should always require orthopaedic consultation. This consideration is much more valuable in children and adolescents with CD than adults since musculoskeletal manifestations such as axial arthritis or metabolic bone disease, which may be a source of low back pain, are not so frequent. Finally, the potential etiopathology of spondylolytic spondylolisthesis developing due to CD in the presented patient is lacking. Therefore, it is still only a hypothesis that the impaired mineralization of bone structures could facilitate isthmic lysis in CD patients, indicating a potential pathogenic relation between spondylolytic spondylolisthesis and CD.

Financial support and sponsorship

None.

Informed consent

Informed consent for publication of the case details has been obtained from the patient.

Funding statement

The author received no financial support for this study.

Ethical review committee statement

Approval is not required by our institution.

CRediT authorship contribution statement

N.K. Sferopoulos: Conceptualization, Researching, Writing – original draft, Writing – review & editing.