Evidence of small-fiber neuropathy (SFN) in two patients with unexplained genital sensory loss and sensory urinary cystopathy

AbdelRazek, Mahmoud A.; Chwalisz, Bart K; Oaklander, Anne Louise; Venna, Nagagopal

doi:10.1016/j.jns.2017.07.016

Cited by 3 publications

(5 citation statements)

References 8 publications

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“…In patients with sarcoidosis, the development of systemic inflammation and internal organ dysfunction are observed, which significantly reduces quality of life and worsens patient prognosis. One of the most common complications is considered to be small fiber neuropathy (SFN), a condition that remains significantly understudied [ 1 , 2 , 3 , 4 ]. Quite often, patients with lung sarcoidosis complain of a number of non-specific symptoms, such as weakness, sleep disorders, etc., which can significantly affect the patient’s quality of life, even in the absence of significant deviations from the pulmonary system.…”

Section: Introductionmentioning

confidence: 99%

“…Considering the significant role of genetic predisposition and the possible provocative role of exogenous triggers in the development of this complication, SFN in patients with sarcoidosis can be considered as a part of the autoimmune/inflammatory syndrome that is induced by adjuvants (ASIA) [ 11 ]. Several proven cases SFN have also been observed in patients suffering from the bacterial inflammation, e.g., Lyme disease and leprosy [ 1 , 2 , 3 , 4 ].…”

Section: Introductionmentioning

confidence: 99%

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Small Fiber Neuropathy in Sarcoidosis

et al. 2021

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Sarcoidosis (SC) is a granulomatous disease of an unknown origin. The most common SC-related neurological complication is a small fiber neuropathy (SFN) that is often considered to be the result of chronic inflammation and remains significantly understudied. This study aimed to identify the clinical and histological correlates of small fiber neuropathy in sarcoidosis patients. The study was performed in 2018–2019 yy and included 50 patients with pulmonary sarcoidosis (n = 25) and healthy subjects (n = 25). For the clinical verification of the SFN, the “Small Fiber Neuropathy Screening List” (SFN-SL) was used. A punch biopsy of the skin was performed followed by enzyme immunoassay analysis with PGP 9.5 antibodies. Up to 60% of the sarcoidosis patients reported the presence of at least one complaint, and it was possible that these complaints were associated with SFN. The most frequent complaints included dysfunctions of the cardiovascular and musculoskeletal systems and the gastrointestinal tract. A negative, statistically significant correlation between the intraepidermal nerve fiber density (IEND) and SFN-SL score was revealed. In patients with pulmonary sarcoidosis, small fiber neuropathy might develop as a result of systemic immune-mediated inflammation. The most common symptoms of this complication were dysautonomia and mild sensory dysfunction.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Small Fiber Neuropathy in Sarcoidosis

et al. 2021

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“…In patients with sarcoidosis, the development of systemic in ammation and internal organ dysfunction are observed, which signi cantly reduces the quality of life and worsens the prognosis of the patients. One of the most common complication is considered to be a small ber neuropathy (SFN), that remains signi cantly understudied (1)(2)(3)(4).…”

Section: Introductionmentioning

confidence: 99%

“…Considering the signi cant role of the genetic predisposition and the possible provocative role of exogenous triggers in the development of this complication, SFN in patients with sarcoidosis can be considered as a part of the autoimmune/in ammatory syndrome induced by adjuvants (ASIA) (11). Several cases of the proven SFN were also observed in patients, suffering from the bacterial in ammation, e.g., Lime disease and leprosy (1)(2)(3)(4). To our best knowledge, there is no studies on the detecting the reduction of the small nerve bers in patients with tuberculosis, though this data may contribute to the better understanding of the pathogenesis of different types of in ammation and also allude the new marker of the differential diagnosis between tuberculosis and sarcoidosis.…”

Section: Introductionmentioning

confidence: 99%

Small Fiber Neuropathy in Pulmonary Sarcoidosis and Tuberculosis: Clinical and Histological Correlates

Старшинова

Basantsova

Zinchenko

et al. 2020

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Sarcoidosis (SC) is the granulomatous disease of an unknown origin, where the differential diagnosis with tuberculosis (TB) is challenging and vital for patients’ prognosis. The common neurological complication in SC is a small fiber neuropathy (SFN), that is considered to be the result of the chronic inflammation, and remains significantly understudied. There is no reliable data, whether such complication is observed in TB patients, where the systemic inflammation is also described. Aim: To identify the clinical and histological correlates of the small fiber neuropathy in sarcoidosis and tuberculosis patients.Materials and methods. A study was performed in 2018 – 2019 years and included 71 patients with pulmonary sarcoidosis (n=25), pulmonary tuberculosis (n=21), and healthy subjects (n=25). For the clinical verification of the SFN, the “Small fiber neuropathy screening list” (SFN-SL) was used. A punch biopsy of the skin was performed followed by the enzyme immunoassay analysis with PGP 9.5 antibodies.Results of the study: Up to 60% of sarcoidosis patients and 19% tuberculosis patients report the presence of at least one complaint, which may be associated with SFN. The most frequent complaints included dysfunctions of the cardiovascular, musculoskeletal system and gastrointestinal tract. A negative, statistically significant correlation between the intraepidermal nerve fiber density (IEND) and SFN-SL score was revealed in both groups (Spearman coefficient, r = -0.3508, p = 0.0102, and r = -0.7382, p = 0.0064, respectively). Wherein, the density of small nerve fibers in the patients with pulmonary sarcoidosis was lower, compared to the patients with tuberculosis (Mann-Whitney test, p = 0.0047). Conclusion: In patients with pulmonary sarcoidosis, small fiber neuropathy may develop as a result of systemic immune-mediated inflammation. The most common symptoms of this complication were dysautonomia and mild sensory dysfunction. Wherein, in tuberculosis patients clinical and histological symptoms of the small fiber neuropathy were subsequently less prominent, which may represent the difference between the autoimmune and bacterial inflammation. The validated questionnaires and histologic verification of the diagnosis help to establish the severity of neuropathy of small fibers, to determine the prognosis, to plan the treatment strategн, and also may allude the possibility for the additional criteria of differential diagnosis between two diseases.

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“…У пациентов с саркоидозом органов дыхания наблюдается развитие системных полиорганных нарушений, значительно снижающих качество жизни и ухудшающих прогнозы заболевания. Одним из наиболее важных из них является поражение нервной системы с вовлечением нервных волокон наименьшего диаметра, что носит название нейропатии малых волокон (НМВ) [1][2][3][4].…”

Section: Introductionunclassified

Clinical manifestations of small fiber neuropathy in patients with pulmonary sarcoidosis

2019

MedAlliance

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Саркоидоз легких, являясь иммуноопосредованным заболеванием неизвестной этиологии, приводит к развитию системного поражения внутренних органов. В последние годы большое внимание уделяется развитию такого осложнения, как нейропатия малых волокон (НМВ), которая характеризуется поражением тонких нервных волокон наименьшего диаметра, широко представленных в коже, слизистых оболочках и внутренних органах. К наиболее распространенным клиническим проявлениям НМВ относят болевой синдром и разнообразные нарушения работы внутренних органов, однако данная патология требует дальнейшего изучения в связи со сложностью диагностики и с широким спектром функций, выполняемых нервными волокнами малого диаметра. Цель исследования: выявление клинических проявлений нейропатии малых волокон при саркоидозе в структуре аутоиммунного/провоспалительного синдрома, индуцированного адъювантами (АСИА). Материалы и методы. В 2018–2019 гг. проведено исследование с участием 25 больных, страдающих саркоидозом легких, и 73 здоровых добровольцев. Проводился стандартный комплекс обследований с применением рентгенологических, морфологических, бактериологических и молекулярно-генетических методов. Для клинической верификации НМВ был использован валидизированный «Опросник для выявления нейропатии ма- лых волокон» («Small fiber neuropathy screening list», SFN-SL). Также использовался «исследовательский опросник АСИА» ("ASIA research Questionnaire"), где оценивались потенциальные триггеры и симптомы развития аутоиммунной реакции. Результаты исследования. До 60% пациентов с саркоидозом отмечают наличие хотя бы одной жалобы, которая может быть ассоциирована с НМВ. К наиболее частым жалобам относились нарушения функции сердечно-сосудистой и опорно-двигательной системы, желудочно-кишечного тракта, наблюдалось поражение кожи и офтальмологическая патология. В 20% случаев пациенты отмечали постоянное присутствие и значительную выраженность симптомов. Также была выявлена положительная корреляция между показателями шкалы SFN-SL и частой встречаемостью АСИА-симптомов (коэффициент корреляции Спирмена, p=0,038; r=0,372; 95% CI 0,1976–0,7538), что позволяет предположить иммуноопосредованный генез данного осложнения. Выводы. У пациентов с саркоидозом легких возможно развитие нейропатии малых волокон в результате системного иммуноопосредованного повреждения нервной ткани. К наиболее распространенным симптомам данного осложнения можно отнести нарушения вегетативной регуляции различных внутренних органов и систем. Применение валидизированных опросников помогает установить выраженность и тяжесть нейропатии малых волокон, что может быть показанием к гистологической верификации диагноза и назначению патогенетической терапии.

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Evidence of small-fiber neuropathy (SFN) in two patients with unexplained genital sensory loss and sensory urinary cystopathy

Cited by 3 publications

References 8 publications

Small Fiber Neuropathy in Sarcoidosis

Small Fiber Neuropathy in Sarcoidosis

Small Fiber Neuropathy in Pulmonary Sarcoidosis and Tuberculosis: Clinical and Histological Correlates

Clinical manifestations of small fiber neuropathy in patients with pulmonary sarcoidosis

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