Joint health and pain in the changing hemophilia treatment landscape.

INTRODUCTION: Hemophilia is an inherited bleeding disorder. Bleeding, and in particular joint hemorrhage results in chronic arthropathy and disability. Acute and chronic pain are frequent and limit activity and participation and result in decreased health-related quality of life. Remarkable progress has been made in the diagnosis and treatment of hemophilia but bleeding continues to prove recalcitrant to currently available treatments and joint disease remains problematic. Physiotherapy and pain management are mainstays of current multidisciplinary integrated care of people with hemophilia (PWH). The focus of this review is on preservation of joint health in the era of new and innovative therapies. AREAS COVERED: A search of the PubMed Central was conducted on 1 February 2024 using the MeSH Major Topic terms identified as keywords for the manuscript. This review will highlight what is known and unknown about joint bleeding and arthropathy, including insights on pain as a related complication. EXPERT OPINION: Recent advances in therapeutic interventions aimed at promoting healthy joints in PWH will be discussed, including both the pharmacological treatment landscape and related strategies to promote joint health.

Crohn's disease in hemophilic arthropathy patient: a case report.

Crohn's disease (CD) is an inflammatory bowel disease affecting the digestive tract, the incidence of which is on the rise worldwide. The most common clinical manifestation of hemophilia is arthropathy secondary to recurrent joint effusions and chronic synovitis. This article reports on a rare 25-year-old male patient with both hemophilic arthropathy and Crohn's disease who was at risk for pathogenic gastrointestinal bleeding. After undergoing endoscopic pathologic testing and genetic testing, a multidisciplinary expert work-up of a treatment and nutritional plan was performed. The patient improved clinically and adhered to conservative treatment. This case report is the first report of this rare co-morbidity, demonstrating the highly pathogenic mutation locus and summarizing the clinical experience of early diagnosis and treatment.

Arthrofibrosis of the knee in pediatric orthopedic surgery.

Arthrofibrosis is a challenging complication associated with knee injuries in both children and adults. While much is known about managing arthrofibrosis in adults, it is necessary to understand its unique aspects and management strategies in the pediatric population. This paper provides an overview of arthrofibrosis in pediatric orthopedic surgery, focusing on its causes, implications, classifications, and management. This paper is a comprehensive review of the literature and existing research on arthrofibrosis in pediatric patients. Arthrofibrosis is characterized by excessive collagen production and adhesions, leading to restricted joint motion and pain. It is associated with an immune response and fibrosis within and around the joint. Arthrofibrosis can result from various knee injuries in pediatric patients, including tibial spine fractures, ACL and PCL injuries, and extra-articular procedures. Technical factors at the time of surgery play a role in the development of motion loss and should be addressed to minimize complications. Preventing arthrofibrosis through early physical therapy is recommended. Non-operative management, including dynamic splinting and serial casting, has shown some benefits. New pharmacologic approaches to lysis of adhesions have shown promise. Surgical interventions, consisting of arthroscopic lysis of adhesions (LOA) and manipulation under anesthesia (MUA), can significantly improve motion and functional outcomes. Arthrofibrosis poses unique challenges in pediatric patients, demanding a nuanced approach that includes prevention, early intervention with non-operative means, and improvements in surgical techniques. Modern pharmacological interventions offer promise for the future. Customized interventions and research focused on pediatric patients are critical for optimal outcomes.

La artrofibrosis es una complicación difícil asociada con lesiones de rodilla tanto en niños como en adultos. Si bien se sabe mucho sobre el manejo de la artrofibrosis en adultos, es necesario comprender sus aspectos únicos y estrategias de manejo en la población pediátrica. Este documento proporciona una visión general de la artrofibrosis en la cirugía ortopédica pediátrica, centrándose en sus causas, implicaciones, clasificaciones y manejo. Este documento es una revisión completa de la literatura y la investigación existente sobre artrofibrosis en pacientes pediátricos. La artrofibrosis se caracteriza por una producción excesiva de colágeno y adherencias, lo que conduce a un movimiento articular restringido y dolor. Se asocia con una inmunorrespuesta y fibrosis dentro y alrededor de la articulación. La artrofibrosis puede ser el resultado de varias lesiones de rodilla en pacientes pediátricos, incluyendo fracturas de columna tibial, lesiones de LCA y LCP, y procedimientos extraarticulares. Los factores técnicos en el momento de la cirugía desempeñan un papel en el desarrollo de la pérdida de movimiento y deben abordarse para minimizar las complicaciones. Se recomienda prevenir la artrofibrosis a través de la fisioterapia temprana. La gestión no operativa, incluyendo el empalme dinámico y la fundición en serie, ha mostrado algunos beneficios. Los nuevos enfoques farmacológicos a la lisis de adherencias han demostrado ser prometedores. Las intervenciones quirúrgicas, consistentes en lisis artroscópica de adherencias (LOA) y manipulación bajo anestesia (MUA), pueden mejorar significativamente el movimiento y los resultados funcionales. La artrofibrosis plantea desafíos únicos en los pacientes pediátricos, exigiendo un enfoque matizado que incluye prevención, intervención temprana con medios no operatorios y mejoras en las técnicas quirúrgicas. Las intervenciones farmacológicas modernas ofrecen una promesa para el futuro. Las intervenciones e investigaciones personalizadas centradas en pacientes pediátricos son fundamentales para obtener resultados óptimos.

AAV mediated repression of Neat1 lncRNA combined with F8 gene augmentation mitigates pathological mediators of joint disease in haemophilia.

Haemophilic arthropathy (HA), a common comorbidity in haemophilic patients leads to joint pain, deformity and reduced quality of life. We have recently demonstrated that a long non-coding RNA, Neat1 as a primary regulator of matrix metalloproteinase (MMP) 3 and MMP13 activity, and its induction in the target joint has a deteriorating effect on articular cartilage. In the present study, we administered an Adeno-associated virus (AAV) 5 vector carrying an short hairpin (sh)RNA to Neat1 via intra-articular injection alone or in conjunction with systemic administration of a capsid-modified AAV8 (K31Q) vector carrying F8 gene (F8-BDD-V3) to study its impact on HA. AAV8K31Q-F8 vector administration at low dose, led to an increase in FVIII activity (16%-28%) in treated mice. We further observed a significant knockdown of Neat1 (~40 fold vs. untreated injured joint, p = 0.005) in joint tissue of treated mice and a downregulation of chondrodegenerative enzymes, MMP3, MMP13 and the inflammatory mediator- cPLA2, in mice receiving combination therapy. These data demonstrate that AAV mediated Neat1 knockdown in combination with F8 gene augmentation can potentially impact mediators of haemophilic joint disease.

30-sit-to-stand power is a better tool than isometric knee extensor strength to detect motor impairment in people with haemophilic arthropathy.

INTRODUCTION: Regular assessment of motor impairments is crucial in people with haemophilic arthropathy (PwHA). This study aimed to determine if there are differences in 30-seconds sit-to-stand (30-STS) power and maximal voluntary isometric contraction (MVIC) of the knee extensors between PwHA and healthy control group (CG). The secondary aims were to investigate the correlation between 30-STS power and MVIC of knee extensors with clinical characteristics and to assess their effectiveness in identifying motor impairment in PwHA. METHODS: A cross-sectional study was conducted by collecting data from PwHA (n = 17) and a sedentary CG (n = 15). MVIC (torque) and 30-STS power were normalised to body mass. Correlation analysis and simple linear regression adjusted for age were used to assess the association between tests and clinical variables. Using z-scores derived from the mean and standard deviation of the CG, we compared the MVIC and the 30-STS power in PwHA. RESULTS: PwHA showed lower MVIC and 30-STS power compared to CG (p < .001; large effect size d > .8). Lower 30-STS power was associated with greater joint impairment and greater fear of movement, whereas MVIC showed no association with clinical variables. 30-STS power showed a lower z-score compared to MVIC (p < .001). In addition, 30-STS power detected 47% of PwHA with motor impairment compared to 0% for MVIC (p = .002). CONCLUSIONS: Our results suggest that 30-STS power may be more effective than knee extensors MVIC in detecting motor impairment in PwHA. Consequently, lower limb skeletal muscle power, rather than maximum knee extensor strength, appears to be more affected in PwHA.

Assessing joint health in haemophilia patients: The combined value of physical examination and ultrasound imaging.

INTRODUCTION: Early diagnosis of joint damage is pivotal in haemophilia to prevent the occurrence and progression of haemophilic arthropathy thus providing optimal personalised management. The haemophilia joint health score version 2.1 (HJHS) is based on a physical examination of the mainly affected joints. Musculoskeletal ultrasound has demonstrated the capability to detect early changes in terms of synovitis and osteochondral damage. The haemophilia early detection with ultrasound (HEAD-US) score has been proposed as a simple and reliable evaluation tool. AIM: This study aims to investigate the correlation between the HJHS and the HEAD-US scores performed by two independent operators (physical therapist and musculoskeletal ultrasound expert) for the evaluation of the joint health status of patients with haemophilia. METHODS: Consecutive adult patients independent of the severity degree were included. Elbows, knees and ankles were evaluated by a physical therapist by HJHS and by a musculoskeletal ultrasound expert following the HEAD-US protocol. RESULTS: We observed a good positive correlation between HJHS and HEAD-US (Spearman's rho 0.72). The main discrepancy in conceptually similar domains was found between the HJHS swelling and the HEAD-US synovitis (rho 0.17), as ultrasound was able to detect even mild synovitis when HJHS swelling was scored 0 in up to 40% of cases. CONCLUSIONS: The HJHS and HEAD-US correlate well even when performed by two independent operators. Musculoskeletal ultrasound is particularly useful for the early detection of synovitis. The routine assessment of both scores helps clinicians define the stage and extension of joint involvement and set up a personalised treatment.

Risk Factors for the Development of Arthrofibrosis After Anterior Cruciate Ligament Reconstruction in Children and Adolescents.

BACKGROUND: Arthrofibrosis is a fibrotic joint disorder resulting in restricted joint motion and pain. Risk factors associated with the development of postoperative arthrofibrosis include female sex, type of graft, and quicker time to reconstruction. These patients have typically benefitted from manipulation under anesthesia or arthroscopic lysis of adhesions. The purpose of this study was to retrospectively review the rate of arthrofibrosis in children and adolescents who previously underwent anterior cruciate ligament (ACL) reconstruction. MATERIALS AND METHODS: This was a retrospective chart review examining patients 18 years or younger who underwent ACL reconstruction between 2013 and 2023. Data collected included age, body mass index, reconstruction technique, concomitant meniscal or ligamentous pathology, and need for revision surgery for arthroscopic lysis of adhesions vs manipulation under anesthesia. RESULTS: A total of 461 patients 18 years or younger who underwent ACL reconstruction were included in this study. Eighteen (3.90%) patients required reoperation for the development of arthrofibrosis. Skeletally immature patients were found to have a statistically significant lower rate of arthrofibrosis compared with skeletally mature patients (0% vs 4.80%; P=.0184). Patients with a higher weight and body mass index had an increased rate of arthrofibrosis (P=.0485 and P=.0410, respectively). Graft type did not have a significant impact on arthrofibrosis rates. There were no significant findings in terms of concomitant injuries and rate of arthrofibrosis. CONCLUSION: Arthrofibrosis developed in 3.90% of patients after ACL reconstruction. Skeletal immaturity may be protective against the development of arthrofibrosis. No association was found between graft type or concomitant knee pathology and arthrofibrosis. [Orthopedics. 2024;47(4):e161-e166.].

The efficacy of vitamin E in preventing arthrofibrosis after joint replacement.

BACKGROUND: Arthrofibrosis is a joint disorder characterized by excessive scar formation in the joint tissues. Vitamin E is an antioxidant with potential anti-fibroblastic effect. The aim of this study was to establish an arthrofibrosis rat model after joint replacement and assess the effects of vitamin E supplementation on joint fibrosis. METHODS: We simulated knee replacement in 16 male Sprague-Dawley rats. We immobilized the surgical leg with a suture in full flexion. The control groups were killed at 2 and 12 weeks (n = 5 per group), and the test group was supplemented daily with vitamin E (0.2 mg/mL) in their drinking water for 12 weeks (n = 6). We performed histological staining to investigate the presence and severity of arthrofibrosis. Immunofluorescent staining and α2-macroglobulin (α2M) enzyme-linked immunosorbent assay (ELISA) were used to assess local and systemic inflammation. Static weight bearing (total internal reflection) and range of motion (ROM) were collected for functional assessment. RESULTS: The ROM and weight-bearing symmetry decreased after the procedure and recovered slowly with still significant deficit at the end of the study for both groups. Histological analysis confirmed fibrosis in both lateral and posterior periarticular tissue. Vitamin E supplementation showed a moderate anti-inflammatory effect on the local and systemic levels. The vitamin E group exhibited significant improvement in ROM and weight-bearing symmetry at day 84 compared to the control group. CONCLUSIONS: This model is viable for simulating arthrofibrosis after joint replacement. Vitamin E may benefit postsurgical arthrofibrosis, and further studies are needed for dosing requirements.

Late Presentation of Primary Oxalosis, Microcrystalline Arthropathy, and Tumoral Calcinosis: A Case Report and a Literature Review.

BACKGROUND: Primary hyperoxaluria consists of a group of inherited disorders with enzymatic defects in the glyoxylate pathway, leading to decreased oxalate metabolism. The resulting oxalic deposition is specifically responsible for kidney disease and joint disease. Neonatal oxalosis is the most severe form of primary hyperoxia type 1, with the onset of end-stage renal disease in childhood. CASE PRESENTATION: A 55-year-old hemodialysis man was referred to Nephrology because of inflammatory polyarthralgia and periarticular swelling evolving for six months. He had been on hemodialysis for six years for end-stage chronic renal failure, diagnosed at the same time as primary hyperoxaluria. Radiological investigation showed a rugby jersey appearance on the lumbar spine, budding calcium tone opacities next to large joints and clavicles, vascular calcifications and tumoral calcinosis. The synovial fluid contained a few cells with polymorphic intracellular crystals. We ruled out hyperparathyroidism, hypoparathyroidism, and related phosphocalcic disorders, and we retained arthropathy and tumoral calcinosis secondary to primary hyperoxaliuria. The patient also had congestive heart failure. Despite intensification of hemodialysis, he did not improve and died at the age of 56 in the context of cachexia. CONCLUSION: This rare case documents the possible occurrence of late clinical presentation and long survival in primary oxalosis with extra renal complications.

Ankle arthrodesis and total ankle replacement in patients with congenital bleeding disorders suffering from severe ankle arthropathy.

INTRODUCTION: In patients with congenital bleeding disorders suffering from severe ankle arthropathy, when conservative treatment and joint-preserving surgical techniques fail, there are two possible non-joint-preserving options: ankle arthrodesis (AA) and total ankle replacement (TAR). AREAS COVERED: The scope and aim of this article was to analyze the current role of AA and TAR in patients with congenital bleeding disorders suffering from severe ankle arthropathy. EXPERT OPINION: In patients with congenital bleeding disorders, both TAR and AA provide good results, mainly in terms of pain relief, although they are not exempt from complications (between 0% and 33% in TAR and between 5% and 23.5% in AA). The current controversy about which of the two surgical techniques, TAR or AA, gives better results, the current literature is not able to resolve it in patients with congenital bleeding disorders. While this question is being answered, my opinion regarding patients with congenital bleeding disorders is that the age of the patient must be taken into account. Given known prosthetic survival rates, the older the patient, the more we might be inclined to indicate TAR. Conversely, AA may be more appropriate for relatively young patients.

Manipulation Under Anesthesia is Safe and Effective for Management of Early Postoperative Knee Arthrofibrosis in Adolescent Patients.

BACKGROUND: Management of postoperative knee arthrofibrosis can be challenging and the preferred time for intervention remains controversial. The purpose of this study is to evaluate the safety and efficacy of early (<3 mo postoperatively) manipulation under anesthesia (MUA) for the treatment of knee arthrofibrosis in adolescent patients. We hypothesized that early MUA could restore normal knee motion with a low complication rate and without the need for more invasive intervention. METHODS: In a retrospective review, 57 patients who underwent MUA for postoperative knee arthrofibrosis were identified. The time between the index surgery and MUA as well as changes in range of motion (ROM) before and after MUA were analyzed. Descriptive statistics with median and interquartile range were used to analyze this non-parametric study cohort. Repeated measures ANOVA was performed to assess improvement in ROM over time. A P value <0.05 denoted statistical significance. RESULTS: The median age of the cohort at time of MUA was 14.5 years [interquartile range (IQR) 12.9 to 17.6)]. 54.4% were male. Median time to MUA was 64 days (IQR 52 to 79) after index surgery. ROM before MUA was 90.0 degrees (IQR 75 to 100), which improved to 130 degrees (120 to 135) after MUA. At final median follow-up of 8.9 months (IQR 5.1 to 16.1), mean ROM was 133 degrees (130 to 140). There were no iatrogenic fractures or physeal separations associated with MUA. 12.3% (n=7/57) failed MUA either due to the need for subsequent repeat MUA (n=2), need for lysis of adhesions (n=3) or need for surgery after MUA (n=2). Those who failed early MUA and required subsequent procedures had ROM >120 degrees at final follow-up. CONCLUSIONS: Postoperative knee arthrofibrosis can be safely and effectively treated with early (<3 mo postoperative) MUA. There were no iatrogenic fractures or physeal separations during MUA. Patients who had recurrence of motion deficits after early MUA and required further intervention, regained satisfactory knee motion at final follow-up. Although further research is warranted to better characterize risk factors for knee arthrofibrosis in adolescent patients, early recognition and MUA is a safe and effective treatment for arthrofibrosis to help patients regain full ROM without invasive intervention. LEVEL OF EVIDENCE: Therapeutic Study - Level IV.

Arthroscopic treatment of ankle impingement syndrome / 中华创伤杂志(英文版)

Arthroscopic treatment of ankle impingement syndrome (AIS) is a minimally invasive surgical procedure used to address symptoms caused by impingement in the ankle joint. This syndrome occurs when there is abnormal contact between certain bones or soft tissues in the ankle, leading to pain, swelling, or limited range of motion. Traditionally, open surgery was the standard approach for treating AIS. However, with advancements in technology and surgical techniques, arthroscopic treatment has become a preferred method for many patients and surgeons. With improved visualization and precise treatment of the arthroscopy, patients can experience reduced pain and improved functionality, allowing them to return to their daily activities sooner. In this paper, we reviewed the application and clinical efficacy the of arthroscopic approach for treating AIS, hoping to provide a reference for its future promotion.

Artropatía del apicultor / Beekeeper' arthropathy

En los apicultores se ha descrito una artropatía inflamatoria de etiología desconocida pero relacionada con su actividad profesional. Se expone el caso de un apicultor que tras la picadura de abeja presentó una artritis de la articulación interfalángica del primer dedo de la mano izquierda. Aunque el curso clínico subagudo y los hallazgos de la RMN obligaban a plantear el diagnóstico diferencial con un proceso infeccioso, el resto de pruebas analíticas, de imagen y la evolución, junto al antecedente de episodio similar unos años antes en un dedo de otra mano tras la picadura de abeja, permitió el diagnóstico de esta entidad

An acute inflammatory arthritis of unknown cause has been described in beekeepers in relation to their work with the hives. We present the case of a beekeeper who, after a bee sting, developed arthritis of the interphalangeal joint of the first finger of his left hand. Although the subacute clinical course and the magnetic resonance imaging findings required the differential diagnosis with an infectious process, the rest of the laboratory tests. other imaging studies and the course, together with a history of a similar episode a few years earlier on a finger of the other hand after a bee sting, enabled us to diagnosis this condition

Progressive pseudorheumatoid dysplasia misdiagnosed as ankylosing spondylitis: a case report / 北京大学学报(医学版)

In this study, we reported a case of progressive pseudorheumatoid dysplasia in Peking University Third Hospital. A 56-year-old male patient presented with hip joint pain for more than 40 years and multiple joints pain with limitation of movements of these joints for 28 years. This patient suffered from joint pain and impaired range of motion of the hip, knee, elbow and shoulder gradually, associated with difficulty in walking and inability to take care of himself. He was diagnosed with "femoral head necrosis" or "ankylosing spondylitis" in local hospitals, but the treatment of nonsteroidal antiinflammatory drugs (NSAIDs) and sulfasalazine was not effective. Up to the age of 14, the patient displayed normal physical development, with the highest height was about 158 cm, according to the patient recall. However, his height was 153 cm at present. There was no history of similar illness in any family member. Physical examinations descried limitation of movement of almost all joints. Enlargement and flexion deformity of the proximal interphalangeal (PIP) joints of the hands resulted in the claw hand appearance. Limited abduction and internal and external rotation of the shoulder and hip could be find. He had normal laboratory findings for blood routine test, biochemical indexes and acute phase reactants such as C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR). Furthermore, HLA-B27 and autoimmune antibodies such as rheumatoid factor (RF), anti-cyclic citrullinated peptide (anti-CCP) antibody and antinuclear antibody (ANA) were all negative. X-ray of the hip showed loss of the joint space and irregularities of the femoral head, both femoral head were flattened, it could be see hyperplasia, osteophytes, bilateral femoral neck thicken, neck dry angle turned smaller. The radiological findings of the spinal vertebra indicated kyphosis deformity, narrowing of the intervertebral discs, vertebral syndesmophytes and flattening of the vertebra. However, there was no clues of bone marrow edema in the lumbar MRI. At last, genetic testing for the Wnt1-inducible signaling pathway protein 3 (WISP3) gene was done and indicated compound heterozygous mutations: 756C>G and c.866dupA. These two mutations were derived from the patient's mother and father (the patient's parents each had a heterozygous mutation). Two exons of the WISP3 gene had nucleotide changes leading to amino acid mutations. According to the patient's history, symptoms, physical examinations, radiological findings and genetic testing, the final definitive diagnosis was progressive pseudorheumatic dysplasia.

Artropatía con braquidactilia familiar: una causa infrecuente de deformidad articular en el adolescente / Familial digital arthropathy-brachydactyly: An infrequent cause of joint deformity in adolescents

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Artritis reumatoide: tus articulaciones como escenario de batalla entre tú y tus guerreros / No disponible

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Osteochondroma Arising from Anterior Inferior Iliac Spine as a Cause of Snapping Hip

Snapping hip syndrome is a relatively common problem that can be easily managed with conservative treatment. This syndrome can be divided into external, internal and intra-articular types. Internal snapping hip syndrome is the rarest amongst these and its etiology is not well understood. We report a unique case of osteochondroma arising from the anterior inferior iliac spine (AIIS), which caused the internal snapping hip syndrome with hip pain and restriction of activity. This rare case of snapping hip syndrome from the AIIS was treated surgically and the symptoms completely disappeared after excision of the tumor.

Tumor fibroso solitario de la pleura asociado a síndromes paraneoplásicos / Solitary fibrous tumor of pleura associated with paraneoplastic syndromes

Introduction: solitary fibrous tumors of pleura (SFTP) are occasionally associated with paraneoplastic syndromes such as hypoglycemia and diffuse skeletal hyperostosis. Clinical case: we report a 60 years old male presenting with episodes of disorientation and inappropriate behavior, along with cough and weight loss. On physical examination there was digital clubbing. A chest X ray showed a big tumor in the right hemithorax. Fasting blood glucose was 24 mg/dl and erythrocyte sedimentation rate was elevated. Large bone X rays showed hyperostosis. A needle biopsy of the tumor showed a SFTP. The tumor was excised and the final pathological diagnosis was a benign SFTP. Hypoglycemia subsided after surgery. After four months of follow up, finger clubbing was disappearing.

Introducción: los tumores fibrosos solitarios de la pleura (TFSP) son neoplasias infrecuentes, consideradas lesiones benignas en general, con potencial maligno dependiendo de su histología. Se asocian en raras ocasiones a síndromes paraneoplásicos como la hipoglicemia (Síndrome de Doege-Potter) e hiperostosis ósea difusa (osteoartropatía hipertrófica pulmonar). Caso clínico: consulta un varón de 60 años en el Hospital Clínico de la Pontificia Universidad Católica de Chile por episodios de desorientación y conductas inapropiadas, junto con tos y baja de peso. Además refiere dolor en extremidades y aumento de volumen de falanges distales de manos. Al examen destaca disminución del murmullo pulmonar a derecha e hipocratismo digital. Se estudia con radiografía y tomografía de tórax revelando un gran tumor en hemitórax derecho de 20 por 18 cm con efecto de masa. Exámenes destacan hipoglicemia de 24 mg/dl y VHS elevada. Estudio endocrinológico y neurológico normal, radiografías de huesos largos y manos revelan hiperostosis ósea. Se estudia con biopsia de aguja gruesa, compatible con TFSP. Se realiza resección completa por toracotomía. Biopsia definitiva confirma diagnóstico de TFSP benigno. Paciente evoluciona satisfactoriamente, resolviéndose hipoglicemia y alteraciones conductuales. Se va de alta al octavo día postoperatorio. Seguimiento a los 4 meses sin nuevos episodios de hipoglicemia y resolución progresiva del hipocratismo digital. Radiografías de tórax sin recidiva. Conclusiones: Los TFSP son inhabituales, se pueden presentar con algunos síndromes paraneoplásicos que deben llevar a la sospecha diagnóstica. Su tratamiento consiste en la resección completa del tumor, logrando escasa recidiva y mejoría sintomática.