Coexisting Sacroiliac Arthritis and Chronic Nonbacterial Osteomyelitis in an Adolescent with Ehlers-Danlos Syndrome: A Case Report and Treatment Success

BACKGROUND Chronic nonbacterial osteomyelitis (CNO) is a multifocal autoinflammatory bone disease mainly affecting children and adolescents. Sacroiliitis is an inflammation of the sacroiliac joint, diagnosed with the use of musculoskeletal MRI due to its ability to visualize active inflammatory lesions. Ehlers-Danlos syndrome (EDS) is non-inflammatory hereditary disorder of connective tissue. Here, we report the case of a 17.5-year-old female patient with classical EDS and long-term course of the CNO with coexistence of sacroiliac arthritis. CASE REPORT On admission, a patient with CNO reported pain in the scapula, thoracic spine, shoulders, and iliac region, with morning stiffness present for 5 months. Physical examination revealed knee and elbow joint hyperextension, hypermobility of the phalanges, increased range of motion of the hip joints, and the presence of reticular rash on the face. In the laboratory blood tests, minor leukocytosis was reported. During hospitalization, a whole-body MRI was performed, detecting bone marrow edema in the Th3, Th4, and Th7 vertebral bodies and the head of seventh rib on the left side, as well as bilaterally in the sacroiliac joints. The patient was diagnosed with sacroiliitis and EDS and successfully treated with risedronate sodium, methotrexate with folic acid, sulfasalazine, and meloxicam, achieving CNO remission and reduced severity of axial skeleton pain. CONCLUSIONS The coexistence of these 3 diseases - CNO, sacroiliac arthritis, and EDS - in the same patient is rare and requires interphysician collaboration to determine the correct diagnosis and subsequently arrange multi-speciality therapeutic management to achieve remission.

Authors

Institutions

Publication Details

Journal
American Journal of Case Reports
Published
2024-08-08
DOI
https://doi.org/10.12659/ajcr.943579
Citations
1
Primary Topic
Osteomyelitis and Bone Disorders Research
Type
article
Field-Weighted Citation Impact
0.47
Controls
|||
ALL TIME
JAN
FEB
MAR
APR
MAY
JUN
JUL
AUG
SEP
OCT
article

Coexisting Sacroiliac Arthritis and Chronic Nonbacterial Osteomyelitis in an Adolescent with Ehlers-Danlos Syndrome: A Case Report and Treatment Success

Zbigniew Żuber, Elżbieta Mężyk, Wadim Wojciechowski, Joanna Ożga et al.
1 citations
American Journal of Case Reports
Osteomyelitis and Bone Disorders Research
0.47
article

Coexisting Sacroiliac Arthritis and Chronic Nonbacterial Osteomyelitis in an Adolescent with Ehlers-Danlos Syndrome: A Case Report and Treatment Success

Zbigniew Żuber, Elżbieta Mężyk, Wadim Wojciechowski, Joanna Ożga, Wojciech Kmiecik
article en
1 citations

Abstract

BACKGROUND Chronic nonbacterial osteomyelitis (CNO) is a multifocal autoinflammatory bone disease mainly affecting children and adolescents. Sacroiliitis is an inflammation of the sacroiliac joint, diagnosed with the use of musculoskeletal MRI due to its ability to visualize active inflammatory lesions. Ehlers-Danlos syndrome (EDS) is non-inflammatory hereditary disorder of connective tissue. Here, we report the case of a 17.5-year-old female patient with classical EDS and long-term course of the CNO with coexistence of sacroiliac arthritis. CASE REPORT On admission, a patient with CNO reported pain in the scapula, thoracic spine, shoulders, and iliac region, with morning stiffness present for 5 months. Physical examination revealed knee and elbow joint hyperextension, hypermobility of the phalanges, increased range of motion of the hip joints, and the presence of reticular rash on the face. In the laboratory blood tests, minor leukocytosis was reported. During hospitalization, a whole-body MRI was performed, detecting bone marrow edema in the Th3, Th4, and Th7 vertebral bodies and the head of seventh rib on the left side, as well as bilaterally in the sacroiliac joints. The patient was diagnosed with sacroiliitis and EDS and successfully treated with risedronate sodium, methotrexate with folic acid, sulfasalazine, and meloxicam, achieving CNO remission and reduced severity of axial skeleton pain. CONCLUSIONS The coexistence of these 3 diseases - CNO, sacroiliac arthritis, and EDS - in the same patient is rare and requires interphysician collaboration to determine the correct diagnosis and subsequently arrange multi-speciality therapeutic management to achieve remission.

American Journal of Case ReportsVol. 25
Jagiellonian University (PL), Andrzej Frycz Modrzewski Krakow University (PL)
Good health and well-being
Openalex Percentile: Top 33%
Osteomyelitis and Bone Disorders Research
0.47
AI Navigator

Ask Laika to Summarize, Analyze, and Connect papers live on the map.

Summarize Papers & Methodologies

Extract key findings, datasets, and comparative methods across publications.

Benchmark Rankings & Visual Analytics

Rank top research institutions, authors, funders, topics, and journals by Field-Weighted Citation Impact (FWCI) and paper volume with instant charts.

Connect Distant Disciplines

Bridge topological clusters on the map to find hidden collaborative intersections.