Why so low? An unusual case of myositis in a child
Abstract Background Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen in skeletal muscle, though typically asymptomatic and under-recognized. While rare in children, there is a nee...
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| Format: | Article |
| Language: | English |
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BMC
2023-04-01
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| Series: | Pediatric Rheumatology Online Journal |
| Subjects: | |
| Online Access: | https://doi.org/10.1186/s12969-023-00816-9 |
| _version_ | 1797841160524267520 |
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| author | Meagan E. Chriswell Robert C. Fuhlbrigge Mark A. Lovell Matthew Monson Jessica L. Bloom |
| author_facet | Meagan E. Chriswell Robert C. Fuhlbrigge Mark A. Lovell Matthew Monson Jessica L. Bloom |
| author_sort | Meagan E. Chriswell |
| collection | DOAJ |
| description | Abstract Background Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen in skeletal muscle, though typically asymptomatic and under-recognized. While rare in children, there is a need to better characterize the disease and its management. Here we present a 12-year-old female with bilateral calf pain who was ultimately found to have sarcoid myositis. Case Presentation A 12-year-old female presented to rheumatology with significantly elevated inflammatory markers and isolated lower leg pain. MRI of the distal lower extremities demonstrated extensive bilateral myositis with active inflammation, atrophy, and to a lesser extent fasciitis. This distribution of myositis in a child garnered a broad differential requiring a systematic evaluation. Ultimately, muscle biopsy revealed non-caseating granulomatous myositis with perivascular inflammation, extensive muscle fibrosis, and fatty replacement of the muscle with a CD4+ T cell predominant, lymphohistiocytic infiltrate consistent with sarcoidosis. Review of histopathology from age 6 of an extraconal mass resected from her right superior rectus muscle further confirmed the diagnosis. She had no other clinical symptoms or findings of sarcoidosis. The patient improved significantly with methotrexate and prednisone, though flared again after self-discontinuation of medications and was subsequently lost to follow-up. Conclusion This is the second reported case of granulomatous myositis associated with sarcoidosis in a pediatric patient, and the first to present with a chief complaint of leg pain. Increased knowledge of pediatric sarcoid myositis within the medical community will enhance recognition of the disease, improve the evaluation of lower leg myositis, and advance outcomes for this vulnerable population. |
| first_indexed | 2024-04-09T16:26:35Z |
| format | Article |
| id | doaj.art-97e904a8dde54d4c901e7ea5e4949c50 |
| institution | Directory Open Access Journal |
| issn | 1546-0096 |
| language | English |
| last_indexed | 2024-04-09T16:26:35Z |
| publishDate | 2023-04-01 |
| publisher | BMC |
| record_format | Article |
| series | Pediatric Rheumatology Online Journal |
| spelling | doaj.art-97e904a8dde54d4c901e7ea5e4949c502023-04-23T11:11:45ZengBMCPediatric Rheumatology Online Journal1546-00962023-04-012111610.1186/s12969-023-00816-9Why so low? An unusual case of myositis in a childMeagan E. Chriswell0Robert C. Fuhlbrigge1Mark A. Lovell2Matthew Monson3Jessica L. Bloom4School of Medicine, University of Colorado Anschutz Medical CampusDepartment of Pediatrics, Section of Pediatric Rheumatology, University of Colorado Anschutz Medical CampusDepartment of Pathology and Laboratory Services, University of Colorado Anschutz Medical CampusDepartment of Radiology, University of Colorado Anschutz Medical CampusDepartment of Pediatrics, Section of Pediatric Rheumatology, University of Colorado Anschutz Medical CampusAbstract Background Sarcoidosis is characterized by non-caseating epithelioid granulomas in various tissues throughout the body, most commonly the lung. Non-caseating granulomas may be seen in skeletal muscle, though typically asymptomatic and under-recognized. While rare in children, there is a need to better characterize the disease and its management. Here we present a 12-year-old female with bilateral calf pain who was ultimately found to have sarcoid myositis. Case Presentation A 12-year-old female presented to rheumatology with significantly elevated inflammatory markers and isolated lower leg pain. MRI of the distal lower extremities demonstrated extensive bilateral myositis with active inflammation, atrophy, and to a lesser extent fasciitis. This distribution of myositis in a child garnered a broad differential requiring a systematic evaluation. Ultimately, muscle biopsy revealed non-caseating granulomatous myositis with perivascular inflammation, extensive muscle fibrosis, and fatty replacement of the muscle with a CD4+ T cell predominant, lymphohistiocytic infiltrate consistent with sarcoidosis. Review of histopathology from age 6 of an extraconal mass resected from her right superior rectus muscle further confirmed the diagnosis. She had no other clinical symptoms or findings of sarcoidosis. The patient improved significantly with methotrexate and prednisone, though flared again after self-discontinuation of medications and was subsequently lost to follow-up. Conclusion This is the second reported case of granulomatous myositis associated with sarcoidosis in a pediatric patient, and the first to present with a chief complaint of leg pain. Increased knowledge of pediatric sarcoid myositis within the medical community will enhance recognition of the disease, improve the evaluation of lower leg myositis, and advance outcomes for this vulnerable population.https://doi.org/10.1186/s12969-023-00816-9SarcoidosisMyositisGranulomaGranulomatous myositisPediatricMuscle biopsy |
| spellingShingle | Meagan E. Chriswell Robert C. Fuhlbrigge Mark A. Lovell Matthew Monson Jessica L. Bloom Why so low? An unusual case of myositis in a child Pediatric Rheumatology Online Journal Sarcoidosis Myositis Granuloma Granulomatous myositis Pediatric Muscle biopsy |
| title | Why so low? An unusual case of myositis in a child |
| title_full | Why so low? An unusual case of myositis in a child |
| title_fullStr | Why so low? An unusual case of myositis in a child |
| title_full_unstemmed | Why so low? An unusual case of myositis in a child |
| title_short | Why so low? An unusual case of myositis in a child |
| title_sort | why so low an unusual case of myositis in a child |
| topic | Sarcoidosis Myositis Granuloma Granulomatous myositis Pediatric Muscle biopsy |
| url | https://doi.org/10.1186/s12969-023-00816-9 |
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