Case Reports

Boskovic, Aleksandra; Stankovic, Ivica; Purkait, Radheshyam; Bhattacharya, Sreyasi; Roy, Birendranath; Bhadra, Ramchandra; Bhatia, Vidyut; Kumar, Pawan; Kodagali, Sheethal S.; Subbarao, S. D.; Hiremagaloor, R.

doi:10.1007/s13312-014-0385-0

Cited by 5 publications

(3 citation statements)

References 9 publications

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“…Peripheral neuropathy and demyelinating lesions are common associations reported by Coronel-Rodríguez et al as being associated with decreased absorption of some dietary components such as tocopherol, cobalamin, or thiamine [2]. In one of our case reports, Boskovic et al describe the case of a 15-year-old female who presented with acute weakness and pricking sensation in her lower limbs on stopping GFD, which she was on since she was diagnosed with CD at nine months of age [19]. Headache, the most common neurological association in CD children, is hypothesized to be due to several pathogenetic mechanisms, as mentioned in Figure 3 [2].…”

Section: Pathophysiologymentioning

confidence: 57%

“…Seven cases of patients with various presentations of CD and gluten sensitivity were identified [18][19][20][21][22][23][24]. In all of the above-mentioned case reports, GFD had a significant impact on improvement in clinical and laboratory findings.…”

Section: Case Reportsmentioning

confidence: 96%

“…In all of the above-mentioned case reports, GFD had a significant impact on improvement in clinical and laboratory findings. In fact, in the case report by Boskovic et al, accidental ingestion of a diet rich in gluten after a period of management with GFD caused the acute episode, which was resolved after resuming strict adherence to GFD [19].…”

Section: Case Reportsmentioning

confidence: 98%

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Pathological Manifestations of Gluten-Related Neuro-Psychiatric Disorders and the Impact of Gluten-Free Diet in a Pediatric Age Group: A Systematic Review

Nagarajappa,

Chavali,

Mylavarapu

2023

Cureus

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Gluten, as a term, causes unease among a vast majority of the population. The reason is the body's inability to process gluten, causing various pathological manifestations. While celiac disease is predominantly a gastrointestinal disease, it also has various extra-intestinal manifestations. Many children receive diagnoses of idiopathic neuropsychiatric symptoms such as epilepsy, attention-deficit hyperactivity disorder (ADHD), restless leg syndrome (RLS), and peripheral neuropathy without ever finding the root cause. A majority of these cases may be associated with celiac disease if only their antibody titers and other appropriate investigations were conducted. The treatment of these manifestations may be eliminated or at least controllable with dietary modification to a gluten-free diet (GFD). In this paper, we will discuss the pathology of celiac disease and the impact of GFD on the neuropsychiatric aspects of this disease, which is of higher prevalence in the pediatric population.A comprehensive literature search was conducted in prominent databases, namely PubMed and Google Scholar, to include studies that provided individual-level data on the neuropathological manifestations and the impact of a GFD on extra-intestinal manifestations of celiac disease. The research protocol was registered in the PROSPERO database (International Prospective Register of Systematic Reviews) with the registration ID: CRD42023415100. Based on the inclusion and exclusion criteria, we included prospective studies, observational studies, and case reports on pediatric patients with biopsy-proven celiac disease, serologically positive celiac disease, celiac disease with neuropsychiatric manifestations, and studies reporting the impact of GFD. After a rigorous quality assessment to remove the risk of bias, we finally included 20 studies to be discussed.In 6 (30%) studies, patients with neuropsychiatric manifestations had positive serology findings and a relatively higher grade of biopsy results. Seven studies discussed the positive impact of GFD. Five of these seven studies reported statistically significant results (p ≤ 0.001). Our study suggests that gluten plays a role in the severity of neuropsychiatric manifestations of celiac disease. Considering the results of our study, we can see that GFD does impact the prognosis of the disease. Neuropsychiatric findings without gastrointestinal manifestations are more common in the pediatric age group. We have clear evidence that several neurological conditions (neuropathy, ADHD, epilepsy, and RLS) have not only a significant association with gluten but can also potentially benefit from GFD. Thus, screening, with a combination of serological, biopsy, and imaging techniques, must be adapted into the guidelines for early detection and induction of GFD. Furthermore, studies should aim at introducing GFD in the pediatric population as a mode of primary prevention. In conclusion, our review underscores the importance of gluten while dealing with idiopathic neurological conditions in children and h...

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Section: Pathophysiologymentioning

confidence: 57%

Section: Case Reportsmentioning

confidence: 96%

Section: Case Reportsmentioning

confidence: 98%

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Pathological Manifestations of Gluten-Related Neuro-Psychiatric Disorders and the Impact of Gluten-Free Diet in a Pediatric Age Group: A Systematic Review

Nagarajappa,

Chavali,

Mylavarapu

2023

Cureus

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Neurological Complications of Gastrointestinal Disease

Shapiro

Blanco

2017

Seminars in Pediatric Neurology

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Acrodystrophic axonal polyneuropathy with celiac disease: a case report

et al. 2021

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Background Patients with celiac disease present with not only gastrointestinal symptoms but also extraintestinal manifestations such as anemia, osteopathy, dermatitis herpetiformis, and celiac neuropathy. Despite a fairly wide range of celiac neuropathies, we report a case of the acrodystrophic variant of celiac polyneuropathy, which has not been previously described. Case presentation A 41-year-old Ukrainian male suffered from symmetric, sensorimotor axonal polyneuropathy and encephalopathy associated with celiac disease, which is characterized by severe trophic disorders in the lower extremities (trophic ulcers, hyperkeratosis, and anhidrosis). Acrodystrophic changes in the lower extremities were due to both neurogenic and direct immunoinflammatory damaging effects. Clinical–electrophysiological dissociation was also noted, which was represented by a gross axonal lesion with the preservation of muscle strength. The absence of enteropathic manifestations was accompanied by the pronounced histological changes in the duodenal mucosa by IIIb stage of Marsh. A gluten-free diet in combination with membrane plasma exchange and intravenous pulse methylprednisolone was prescribed to reduce the severity of sensory disorders and regression of encephalopathy within 7 months. Conclusion Celiac disease may be a potential cause of neuropathy and encephalopathy in adult patients. Further immunosuppressive treatment protocols for both intestinal and extraintestinal manifestations of celiac disease are required.

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Case Reports

Abstract: Polyneuropathy may occur as a complication of celiac disease in childhood.

Cited by 5 publications

References 9 publications

Pathological Manifestations of Gluten-Related Neuro-Psychiatric Disorders and the Impact of Gluten-Free Diet in a Pediatric Age Group: A Systematic Review

Pathological Manifestations of Gluten-Related Neuro-Psychiatric Disorders and the Impact of Gluten-Free Diet in a Pediatric Age Group: A Systematic Review

Neurological Complications of Gastrointestinal Disease

Acrodystrophic axonal polyneuropathy with celiac disease: a case report

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