Langerhans Cell Histiocytosis of the Atlas in the Pediatric Spine: Total Reconstitution of the Bone Lesion After Nonoperative Treatment

Puigdevall, Miguel; Bosio, S; Hokama, Jorge; Maenza, Ruben

doi:10.2106/jbjs.g.01352

Cited by 9 publications

(11 citation statements)

References 17 publications

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“…Without a normal vertebral body, a lateral mass in the C1 vertebra is most commonly involved in cases of EG. [3,10–12] Unlike the subaxial cervical spine, the atlantooccipital and atlantoaxial joints are adjacent to the C1 lateral mass and C2 vertebral body, and the stability of this region is maintained with the help of ligaments. The atlantooccipital joint contributes to the 23° to 24.5° flexion/extension, and the atlantoaxial joint provides 25° to 30° rotation and 10.1° to 22.4° flexion/extension.…”

Section: Discussionmentioning

confidence: 99%

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The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint

et al. 2017

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Rationale:Solitary eosinophilic granuloma (EG), the most benign, common form of Langerhans cell histiocytosis, has a self-limiting process and is associated with a good prognosis. Immobilization is recommended as the first treatment strategy for solitary EG, although the treatment protocols are still controversial. Radiotherapy and surgery are secondary treatment choices. Lesions of the upper cervical spine react differently to treatment because of their specific anatomical and motor features.Patient concerns:We discuss the case of a 29-year-old man with axis EG who underwent immobilization, radiotherapy, and finally surgery.Diagnosis:Eosinophilic granuloma (EG).Interventions:An initial conservative protocol, including immobilization with a collar and radiotherapy, effectively relieved his neck pain, whereas torticollis secondary to atlantoaxial subluxation was not improved. Therefore, he underwent tumor resection through the anterior approach and spinal reconstruction, fixation, and fusion through the posterior approach.Outcomes:The deformity was well corrected and follow-up was satisfactory.Lessons:Upon review of the literature, we found that EG lesions affecting the axis have more risk factors for instability or deformity, and they have particular anatomical and motor characteristics; thus, they require more consideration and attention in terms of treatment, prognosis, and follow-up.

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Section: Discussionmentioning

confidence: 99%

“…Neck pain with or without a torticollis deformity was the most common complaint when the C1 vertebral body was affected. [3,10–12] Atlantoaxial subluxation or instability is rarely observed.…”

Section: Discussionmentioning

confidence: 99%

The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint

et al. 2017

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“…Spinal LCH is self-limiting and the prognosis is usually good. A variety of treatment modalities for spinal LCH have been reported, including conservative management, intralesional steroid injection, radiation therapy, chemotherapy, and curettage with or without reconstructive surgery (Puigdevall et al 2008; Garg et al 2003; Bertram et al 2002). Mild, isolated involvement of the spine without neurological deficits or spinal instability can be managed with conservative methods such as simple observation, nonsteroidal anti-inflammatory drugs, immobilization, or casting with or without initial bed rest.…”

Section: Discussionmentioning

confidence: 99%

Langerhans cell histiocytosis in the adult lumbar spine: case report

et al. 2016

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IntroductionLangerhans cell histiocytosis (LCH) occurs rarely in the spine of adults. The radiological findings usually resemble vertebral tumors. Etiology of LCH has not been clearly established yet. Therapeutic approaches are still controversial. We describe a case of LCH in an adult spine.Case descriptionA patient who presented with low back pain had an osteolytic lesion in the L1 vertebral body without neurological deficits, and fluoroscopy-guided needle biopsy of the L1 vertebral body was performed. The immunohistochemical diagnosis confirmed LCH. The patient was successfully treated with conservative methods.DiscussionThe choice of appropriate therapy is very important, with treatment options varying from watch-and to aggressive treatment.ConclusionLCH is considered as a pediatric disease that is extremely rare in the spine of adults and should be include in the differential diagnosis of osteolytic vertebral lesions. Conservative treatment is best choice for a patient with LCH without neurological deficit or spinal instability.

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“…Recent studies have reported that as many as 39% LCH cases were adults and that spine lesion is less common in the adult group (3-10%) [9]. More than half of the cervical LCH lesions affected the C3-5 vertebrae [15], and there were very few published cases describing Langerhans cell histiocytosis of the atlas. To the best of our knowledge, only 17 LCH cases involving the atlas have been reported in the English literature [4,6,10,13,15,16,19].…”

Section: Historical Reviewmentioning

confidence: 99%

“…More than half of the cervical LCH lesions affected the C3-5 vertebrae [15], and there were very few published cases describing Langerhans cell histiocytosis of the atlas. To the best of our knowledge, only 17 LCH cases involving the atlas have been reported in the English literature [4,6,10,13,15,16,19]. Most cases had multiple vertebrae [4,6,10] and/or extraspinal involvement [19].…”

Section: Historical Reviewmentioning

confidence: 99%

Langerhans cell histiocytosis of the atlas in an adult

et al. 2009

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Langerhans cell histiocytosis (LCH), formerly known as histiocytosis X, is a rare disorder (approximately 1:1,500,000 inhabitants) characterized by clonal proliferation and excess accumulation of pathologic Langerhans cells causing local or systemic effects. The exact etiology of LCH is still unknown. LCH could affect patients of any age, although most present when they are children. The most frequent sites of the bony lesions are the skull, femur, mandible, pelvis and spine. A variety of treatment modalities has been reported, but there was no evidence suggesting that any one treatment was more advantageous than another. We present an adult with LCH of the atlas. A 26-year-old young man presented with a 2-month history of neck pain and stiffness. CT revealed osteolytic lesion in the left lateral mass of atlas with compression fracture. Histopathological diagnosis was Langerhans cell histiocytosis by percutaneous needle biopsy under CT guidance. The patient underwent conservative treatment, including Halo-vest immobilization and radiotherapy. At 7-year follow-up, the patient was asymptomatic except for mild motion restriction of the neck. CT revealed a significant reconstruction of the C1 lateral mass.

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Langerhans Cell Histiocytosis of the Atlas in the Pediatric Spine: Total Reconstitution of the Bone Lesion After Nonoperative Treatment

Cited by 9 publications

References 17 publications

The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint

The outcome of eosinophilic granuloma involving unilateral atlantoaxial joint

Langerhans cell histiocytosis in the adult lumbar spine: case report

Langerhans cell histiocytosis of the atlas in an adult

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