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Article ; Online: Chronic Nonbacterial Osteomyelitis in Inflammatory Bowel Disease

Ariadni Tzaneti / Elli Athanasopoulou / Smaragdi Fessatou / Lampros Fotis

Life, Vol 13, Iss 12, p

2023 Volume 2347

Abstract: Chronic nonbacterial osteomyelitis (CNO), also known as chronic recurrent multifocal osteomyelitis (CRMO), is a rare autoinflammatory bone disease primarily affecting children and adolescents. This review presents a comprehensive analysis of the ... ...

Abstract	Chronic nonbacterial osteomyelitis (CNO), also known as chronic recurrent multifocal osteomyelitis (CRMO), is a rare autoinflammatory bone disease primarily affecting children and adolescents. This review presents a comprehensive analysis of the intricate relationship between CNO and inflammatory bowel disease (IBD), shedding light on shared pathophysiological mechanisms and clinical management. A thorough literature review was conducted, encompassing 24 case reports involving 40 patients. The demographic distribution of patients revealed a near-equal gender ratio, with a median age of diagnosis at 12 years. The diagnosis patterns showed a higher proportion of CNO as the initial diagnosis, while Crohn’s disease was more prevalent than ulcerative colitis. The time interval between the clinical presentations varied, ranging from simultaneous detection to a substantial 15-year gap. Treatment modalities included nonsteroidal anti-inflammatory drugs (NSAIDs), steroids, aminosalicylates, and biologic agents, such as infliximab, often overlapping in their use and suggesting shared pathophysiological pathways. Both conditions displayed systemic manifestations, and patients often responded well to immunosuppressive medications. The pathophysiology of CNO involves a genetic predisposition, cytokine dysregulation, and osteoclast activation. Dysregulated innate immunity results in immune cell infiltration into bones, causing sterile bone lesions. Notably, emerging evidence hints at a potential link between the microbiome and CNO. In contrast, IBD results from imbalanced mucosal immune responses to the intestinal microbiota. Polymorphisms in the promotor region of IL-10, common cytokines, immune cells, and genetic markers indicate shared immunological and genetic factors between CNO and IBD. Both conditions also involve extraintestinal symptoms. This analysis underscores the need for clinical awareness of the co-occurrence of CNO and IBD, especially among pediatric patients. A deepened understanding of the connections ...
Keywords	CRMO ; Crohn’s disease ; inflammatory bowel disease ; osteomyelitis ; pediatrics ; ulcerative colitis ; Science ; Q
Subject code	610
Language	English
Publishing date	2023-12-01T00:00:00Z
Publisher	MDPI AG
Document type	Article ; Online
Database	BASE - Bielefeld Academic Search Engine (life sciences selection)

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Article: Chronic Nonbacterial Osteomyelitis in Inflammatory Bowel Disease.

Tzaneti, Ariadni / Athanasopoulou, Elli / Fessatou, Smaragdi / Fotis, Lampros

Life (Basel, Switzerland)

2023 Volume 13, Issue 12

Abstract	Chronic nonbacterial osteomyelitis (CNO), also known as chronic recurrent multifocal osteomyelitis (CRMO), is a rare autoinflammatory bone disease primarily affecting children and adolescents. This review presents a comprehensive analysis of the intricate relationship between CNO and inflammatory bowel disease (IBD), shedding light on shared pathophysiological mechanisms and clinical management. A thorough literature review was conducted, encompassing 24 case reports involving 40 patients. The demographic distribution of patients revealed a near-equal gender ratio, with a median age of diagnosis at 12 years. The diagnosis patterns showed a higher proportion of CNO as the initial diagnosis, while Crohn's disease was more prevalent than ulcerative colitis. The time interval between the clinical presentations varied, ranging from simultaneous detection to a substantial 15-year gap. Treatment modalities included nonsteroidal anti-inflammatory drugs (NSAIDs), steroids, aminosalicylates, and biologic agents, such as infliximab, often overlapping in their use and suggesting shared pathophysiological pathways. Both conditions displayed systemic manifestations, and patients often responded well to immunosuppressive medications. The pathophysiology of CNO involves a genetic predisposition, cytokine dysregulation, and osteoclast activation. Dysregulated innate immunity results in immune cell infiltration into bones, causing sterile bone lesions. Notably, emerging evidence hints at a potential link between the microbiome and CNO. In contrast, IBD results from imbalanced mucosal immune responses to the intestinal microbiota. Polymorphisms in the promotor region of IL-10, common cytokines, immune cells, and genetic markers indicate shared immunological and genetic factors between CNO and IBD. Both conditions also involve extraintestinal symptoms. This analysis underscores the need for clinical awareness of the co-occurrence of CNO and IBD, especially among pediatric patients. A deepened understanding of the connections between these seemingly distinct diseases could lead to more effective management and improved patient outcomes.
Language	English
Publishing date	2023-12-15
Publishing country	Switzerland
Document type	Journal Article ; Review
ZDB-ID	2662250-6
ISSN	2075-1729
ISSN	2075-1729
DOI	10.3390/life13122347
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Article ; Online: Arthritis Remission With Partial Cartilage Restoration in a 9 y/o Female With Hip Chondrolysis Following Treatment With Adalimumab.

Fotis, Lampros / Kanellopoulos, Anastasios / Papakonstantinou, Olympia

Modern rheumatology case reports

2022

Abstract: Hip chondrolysis is observed primarily or secondary to other diseases and is a rare but yet debilitating disease, characterized by loss of cartilage of the femoral epiphysis and significant restriction of motion. We present the case of a 9-year-old ... ...

Abstract	Hip chondrolysis is observed primarily or secondary to other diseases and is a rare but yet debilitating disease, characterized by loss of cartilage of the femoral epiphysis and significant restriction of motion. We present the case of a 9-year-old female diagnosed with hip chondrolysis associated with probable juvenile psoriatic arthritis. Avoidance of weight bearing activities and treatment with corticosteroids, methotrexate, and adalimumab followed by aquatic therapy resulted in clinical and radiographic improvement as well as partial cartilage regeneration.
Language	English
Publishing date	2022-03-28
Publishing country	England
Document type	Journal Article
ISSN	2472-5625
ISSN (online)	2472-5625
DOI	10.1093/mrcr/rxac024
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Article ; Online: Tailored treatment strategies and future directions in systemic lupus erythematosus.

Nikolopoulos, Dionysis / Fotis, Lampros / Gioti, Ourania / Fanouriakis, Antonis

Rheumatology international

2022 Volume 42, Issue 8, Page(s) 1307–1319

Abstract: Systemic lupus erythematosus (SLE) represents a diagnostic and therapeutic challenge for physicians due to its protean manifestations and unpredictable course. The disease may manifest as multisystemic or organ-dominant and severity at presentation may ... ...

Abstract	Systemic lupus erythematosus (SLE) represents a diagnostic and therapeutic challenge for physicians due to its protean manifestations and unpredictable course. The disease may manifest as multisystemic or organ-dominant and severity at presentation may vary according to age at onset (childhood-, adult- or late-onset SLE). Different manifestations may respond variably to different immunosuppressive medications and, even within the same organ-system, the severity of inflammation may vary from mild to organ-threatening. Current "state-of-the-art" in SLE treatment aims at remission or low disease activity in all organ systems. Apart from hydroxychloroquine and glucocorticoids (which should be used with caution), the choice of the appropriate immunosuppressive agent should be individualized and depend on the prevailing manifestation, severity stratification and patient childbearing potential. In this review, we provide an overview of therapeutic options for the various organ manifestations and severity patterns of the disease, different phenotypes (such as multisystem versus organ-dominant disease), as well as specific considerations, including lupus with antiphospholipid antibodies, childhood and late-onset disease, as well as treatment options during pregnancy and lactation.
MeSH term(s)	Antibodies, Antiphospholipid ; Female ; Glucocorticoids/therapeutic use ; Humans ; Hydroxychloroquine/therapeutic use ; Immunosuppressive Agents/therapeutic use ; Lupus Erythematosus, Systemic/diagnosis ; Lupus Erythematosus, Systemic/drug therapy
Chemical Substances	Antibodies, Antiphospholipid ; Glucocorticoids ; Immunosuppressive Agents ; Hydroxychloroquine (4QWG6N8QKH)
Language	English
Publishing date	2022-04-21
Publishing country	Germany
Document type	Journal Article ; Review
ZDB-ID	8286-7
ISSN	1437-160X ; 0172-8172
ISSN (online)	1437-160X
ISSN	0172-8172
DOI	10.1007/s00296-022-05133-0
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Article ; Online: Erythromelalgia in an Adolescent Female.

Mamatsi, Eleftheria / Giorgi, Melpomeni / Dinopoulos, Argirios / Papaevangelou, Vasiliki / Fotis, Lampros

Mediterranean journal of rheumatology

2022 Volume 33, Issue 2, Page(s) 256–258

Abstract: Erythromelalgia is a disabling syndrome of paroxysmal vasodilation affecting the feet, hands and face characterised by patient's cooling behaviour to achieve symptom relief. It can be primary or secondary and although a rare disorder it has been ... ...

Abstract	Erythromelalgia is a disabling syndrome of paroxysmal vasodilation affecting the feet, hands and face characterised by patient's cooling behaviour to achieve symptom relief. It can be primary or secondary and although a rare disorder it has been described in children and adolescents. We describe the case of a 14-year-old female diagnosed with primary erythromelalgia successfully treated with aspirin, amitriptyline, and carbamazepine.
Language	English
Publishing date	2022-06-30
Publishing country	Greece
Document type	Case Reports
ZDB-ID	3019943-8
ISSN	2529-198X ; 2459-3516
ISSN (online)	2529-198X
ISSN	2459-3516
DOI	10.31138/mjr.33.2.256
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Article: Janus Kinase Inhibitors and Interstitial Lung Disease Associated With Pediatric Rheumatic Diseases: An Unexplored Field.

Sapountzi, Evdoxia / Fotis, Lampros / Kotanidou, Eleni / Fidani, Liana / Galli-Tsinopoulou, Assimina

Cureus

2023 Volume 15, Issue 12, Page(s) e50928

Abstract: Rheumatic diseases are often complicated by lung disease, commonly presenting as interstitial lung disease (ILD), with potentially detrimental consequences for patient survival. Although less frequent in pediatric patients, pulmonary involvement may be ... ...

Abstract	Rheumatic diseases are often complicated by lung disease, commonly presenting as interstitial lung disease (ILD), with potentially detrimental consequences for patient survival. Although less frequent in pediatric patients, pulmonary involvement may be observed in almost all childhood-onset rheumatic conditions. The development of biological disease-modifying anti-rheumatic drugs has significantly improved clinical outcomes. However, disease remission is not always complete or long-lasting, and treatment may need to be discontinued due to adverse effects. A novel class of drugs, namely Janus kinase inhibitors (JAKis), has been proposed to provide a significant survival benefit for patients with rheumatic diseases. Despite the ample literature on the efficacy and safety of JAKis in rheumatic disease, only a few studies have investigated the effectiveness of these drugs in patients with pulmonary involvement, and only two case reports have presented results in pediatric patients. We provide an overview of the rationale for using JAKis in ILDs associated with rheumatic disease and summarize the main studies evaluating their efficacy in both adult and pediatric patients. The present review highlights the need for controlled long-term studies to assess the efficacy and safety of JAKis in pediatric rheumatic disease complicated by lung disease.
Language	English
Publishing date	2023-12-21
Publishing country	United States
Document type	Journal Article ; Review
ZDB-ID	2747273-5
ISSN	2168-8184
ISSN	2168-8184
DOI	10.7759/cureus.50928
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Article ; Online: Facial necrotic ulcerative lesions in an adolescent female with granulomatosis with polyangiitis (GPA).

Fotis, Lampros / Prountzos, Spyridon / Giannouli, Georgia / Papaevangelou, Vassiliki

Clinical rheumatology

2020 Volume 39, Issue 11, Page(s) 3519–3520

MeSH term(s)	Adolescent ; Antibodies, Antineutrophil Cytoplasmic ; Female ; Granulomatosis with Polyangiitis/complications ; Humans ; Necrosis
Chemical Substances	Antibodies, Antineutrophil Cytoplasmic
Language	English
Publishing date	2020-07-01
Publishing country	Germany
Document type	Editorial
ZDB-ID	604755-5
ISSN	1434-9949 ; 0770-3198
ISSN (online)	1434-9949
ISSN	0770-3198
DOI	10.1007/s10067-020-05264-4
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Article ; Online: Colchicine-Induced Macrophage Activation Syndrome in an Adolescent Female Patient With PSTPIP1-Associated Myeloid-Related Proteinemia Inflammatory Syndrome.

Fotis, Lampros / Kekkou, Kassiani / Papaevangelou, Vassiliki / Fessatou, Smaragdi

Journal of clinical rheumatology : practical reports on rheumatic & musculoskeletal diseases

2020 Volume 27, Issue 8S, Page(s) S409–S410

MeSH term(s)	Adaptor Proteins, Signal Transducing/genetics ; Adolescent ; Colchicine/adverse effects ; Cytoskeletal Proteins/genetics ; Female ; Humans ; Macrophage Activation Syndrome/chemically induced ; Macrophage Activation Syndrome/diagnosis ; Metal Metabolism, Inborn Errors/genetics
Chemical Substances	Adaptor Proteins, Signal Transducing ; Cytoskeletal Proteins ; PSTPIP1 protein, human ; Colchicine (SML2Y3J35T)
Language	English
Publishing date	2020-11-05
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	1283266-2
ISSN	1536-7355 ; 1076-1608
ISSN (online)	1536-7355
ISSN	1076-1608
DOI	10.1097/RHU.0000000000001407
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Article: Whole-body magnetic resonance imaging findings and patterns of chronic nonbacterial osteomyelitis in a series of Greek pediatric patients.

Papakonstantinou, Olympia / Prountzos, Spyridon / Karavasilis, Eustratios / Atsali, Erato / Bizimi, Vasiliki / Alexopoulou, Efthymia / Fotis, Lampros

Acta radiologica open

2022 Volume 11, Issue 6, Page(s) 20584601221106701

Abstract: Background: Chronic nonbacterial osteomyelitis (CNO) is a pediatric autoinflammatory disorder presenting with sterile inflammatory bone lesions. Whole-body MRI (WBMRI) has most recently emerged for disease assessment, but data are limited.: Purpose: ... ...

Abstract	Background: Chronic nonbacterial osteomyelitis (CNO) is a pediatric autoinflammatory disorder presenting with sterile inflammatory bone lesions. Whole-body MRI (WBMRI) has most recently emerged for disease assessment, but data are limited. Purpose: The purpose is to evaluate the imaging findings and patterns of CNO on WBMRI in a series of Greek pediatric patients. Material and methods: Whole-body magnetic resonance imaging studies of all children with documented CNO, performed in a single tertiary center, were retrospectively reviewed. WBMRI included coronal T1 and short-tau inversion recovery (STIR), whole spine sagittal STIR, and ankle/foot sagittal STIR images. High signal intensity lesions on STIR images corresponding to bone marrow edema were recorded. The SPSS v.20 statistical package was used for descriptive statistics. Results: Twenty children were included (mean age: 12, range: 6-16 years) with 1-31 lesions (mean: 11.8) on WBMRI. Two children had unifocal disease localized at the clavicle, three paucifocal (1-4 lesions), and 15 multifocal bone involvement. All but two children presented with ankle pain and exhibited lesions at the bones of the ankle joint (90%) followed by the knee (50%) and pelvis (10%). The tibia was the most frequently affected bone (70%) followed by calcaneus (60%), fibula (50%), femur (45%), talus, and metatarsals (45%). No lesions in the cervical, thoracic, lumbar spine, and mandible were documented. Only small sacral lesions were seen in 25% of patients with the extensive peripheral disease. Bilateral metaphyseal and epiphyseal involvement with transphyseal extension were common, but the periosteal reaction and well-defined lesion margins were rare. Conclusion: Frequent involvement of the foot and ankle and paucity of substantial spinal involvement were seen in Greek pediatric patients with CNO.
Language	English
Publishing date	2022-06-22
Publishing country	England
Document type	Journal Article
ZDB-ID	2818429-4
ISSN	2058-4601
ISSN	2058-4601
DOI	10.1177/20584601221106701
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Article ; Online: Takayasu arteritis in an adolescent with Crohn's disease.

Fotis, Lampros / Kourti, Afroditi / Prountzos, Spyridon / Alexopoulou, Efthymia / Papaevangelou, Vasiliki / Fessatou, Smaragdi

Rheumatology international

2021 Volume 42, Issue 3, Page(s) 563–570

Abstract: Crohn's disease (CD) and Takayasu arteritis (TA) are two distinct clinical entities. Τhe likelihood of both diseases coexisting is low, and although CD co-occurs with all types of vasculitis, TA is the most common subtype. Herein, the case of a 15-year- ... ...

Abstract	Crohn's disease (CD) and Takayasu arteritis (TA) are two distinct clinical entities. Τhe likelihood of both diseases coexisting is low, and although CD co-occurs with all types of vasculitis, TA is the most common subtype. Herein, the case of a 15-year-old female, diagnosed with TA following an initial diagnosis of CD, is reported. A review of the literature, including a systemic review of the case reports and case series of children and adolescents up to the age of 21, with both CD and TA, follows the case description. In total, 28 cases of TA and CD were retrieved. The median age of patients was 14.8 years, they were mostly females (72%) and the median time between the two diagnoses was 3.7 years. In the majority of cases, CD was diagnosed first and TA followed. Computed tomography angiography and magnetic resonance angiography were the preferred imaging modalities to assist diagnosis.
MeSH term(s)	Adolescent ; Child ; Crohn Disease/complications ; Crohn Disease/diagnostic imaging ; Crohn Disease/pathology ; Female ; Humans ; Magnetic Resonance Imaging ; Takayasu Arteritis/complications ; Takayasu Arteritis/diagnostic imaging ; Takayasu Arteritis/pathology ; Tomography, X-Ray Computed
Language	English
Publishing date	2021-04-27
Publishing country	Germany
Document type	Case Reports ; Journal Article ; Review
ZDB-ID	8286-7
ISSN	1437-160X ; 0172-8172
ISSN (online)	1437-160X
ISSN	0172-8172
DOI	10.1007/s00296-021-04869-5
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