Literature DB >> 32667754

Vertebra Plana in Children May Result from Etiologies Other Than Eosinophilic Granuloma.

Fady Baky1, Todd A Milbrandt1, Carola Arndt1, Matthew T Houdek1, A Noelle Larson1.   

Abstract

BACKGROUND: Vertebra plana in children is a diagnostic dilemma for orthopaedic surgeons. This radiographic finding sometimes has been said to be pathognomonic for eosinophilic granuloma (Langerhans cell histiocytosis); however, vertebra plana may also be caused by a range of other conditions. We sought to determine whether vertebra plana can be associated with malignancies other than eosinophilic granuloma. QUESTIONS/PURPOSES: (1) To report the underlying diagnoses for children with vertebra plana and determine how frequently these patients were found to have eosinophilic granuloma as opposed to an underlying malignant process, (2) to evaluate the occurrence of nondiagnostic results on biopsy, and (3) to determine whether the presenting characteristics of spinal lesions were associated with the ultimate clinical diagnosis.
METHODS: As part of a retrospective review, our institutional electronic medical record was searched for all patients younger than 18 years between 1976 and 2017 whose clinical record included the term vertebra plana. Patients with trauma were excluded. Twenty-seven patients met the inclusion criteria (mean [range] age 9 years [0 to 18]; 12 girls). To address our first research purpose about the underlying diagnoses of patients with vertebra plana, we reviewed the final clinical diagnosis. To address our second research purpose about the utility of biopsy, we reviewed which patients underwent a biopsy and whether it had been diagnostic. To address our third research purpose about the radiographic criteria, we classified the radiographs and compared this to the clinical diagnosis. Vertebral collapse was described as less than 50% collapse, 50% to 100% collapse, symmetrical, and asymmetrical. The location of each lesion was noted.
RESULTS: Twelve of 27 patients had a diagnosis of eosinophilic granuloma. Six of 27 had other neoplastic etiologies, including acute lymphoblastic leukemia, primary germ cell tumor, giant cell tumor, rhabdomyosarcoma and teratoma. Seventeen of 27 patients underwent biopsy to confirm the diagnosis; six biopsies were consistent with eosinophilic granuloma, six for other etiologies, and five were nondiagnostic. With the limited patient numbers available, there was no difference in the frequency of less than 50% loss of height or 50% to 100%, or symmetric and asymmetric loss of height, and location of the lesion among patients with eosinophilic granuloma and those with other diagnoses.
CONCLUSIONS: Eosinophilic granuloma or Langerhans cell histiocytosis is a common cause of vertebra plana, but other causes must be considered in children presenting with this radiographic finding. Six of 27 of patients presenting to our center with vertebra plana had an underlying neoplasm other than eosinophilic granuloma. With the limited patient numbers available, pain, spinal location, and the degree and symmetry of collapse were not associated with a diagnosis of eosinophilic granuloma in this series. Thus, patients presenting with vertebral plana and back pain need a comprehensive work-up and potentially tissue biopsy to determine diagnosis and appropriate treatment. LEVEL OF EVIDENCE: Level IV, diagnostic study.

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Year:  2020        PMID: 32667754      PMCID: PMC7491903          DOI: 10.1097/CORR.0000000000001409

Source DB:  PubMed          Journal:  Clin Orthop Relat Res        ISSN: 0009-921X            Impact factor:   4.755


  28 in total

Review 1.  Infection and tumors of the spine in children.

Authors:  John P Dormans; Leslie Moroz
Journal:  J Bone Joint Surg Am       Date:  2007-02       Impact factor: 5.284

2.  Eosinophilic granuloma in the pediatric spine.

Authors:  Jeremy D W Greenlee; Albert J Fenoy; Kathleen A Donovan; Arnold H Menezes
Journal:  Pediatr Neurosurg       Date:  2007       Impact factor: 1.162

3.  Vertebra plana-like lesions in children: case report with special emphasis on the differential diagnosis and indications for biopsy.

Authors:  J O'Donnell; L Brown; H Herkowitz
Journal:  J Spinal Disord       Date:  1991-12

4.  Osteochondritis (Calvé's disease) of a vertebral body--a rare form of vertebra plana.

Authors:  Tamas Shisha; Sandor Kiss; Peter Pal Varga; Laszlo Bucsi; Karoly Pap; George Szoke
Journal:  Eur Spine J       Date:  2005-08-13       Impact factor: 3.134

Review 5.  Vertebra plana caused by primary Ewing sarcoma: case report and review of the literature.

Authors:  Panayiotis J Papagelopoulos; Bradford L Currier; Evanthia Galanis; Mark J Grubb; Douglas J Pritchard; Michael J Ebersold
Journal:  J Spinal Disord Tech       Date:  2002-06

6.  Langerhans cell histiocytosis of spine: a comparative study of clinical, imaging features, and diagnosis in children, adolescents, and adults.

Authors:  Wending D Huang; Xinghai H Yang; Zhipeng P Wu; Quan Huang; Jianru R Xiao; Mosong S Yang; Zhenhua H Zhou; Wangjun J Yan; Dianwen W Song; Tielong L Liu; Ningyang Y Jia
Journal:  Spine J       Date:  2013-04-18       Impact factor: 4.166

7.  Imaging of chronic recurrent multifocal osteomyelitis of childhood first presenting with isolated primary spinal involvement.

Authors:  S E Anderson; P Heini; M J Sauvain; E Stauffer; L Geiger; J O Johnston; A Roggo; D Kalbermatten; L S Steinbach
Journal:  Skeletal Radiol       Date:  2003-02-21       Impact factor: 2.199

8.  Chronic recurrent multifocal osteomyelitis. Association with vertebra plana.

Authors:  L Yu; J R Kasser; E O'Rourke; H Kozakewich
Journal:  J Bone Joint Surg Am       Date:  1989-01       Impact factor: 5.284

Review 9.  Langerhans cell histiocytosis of the spine in children. Long-term follow-up.

Authors:  Sumeet Garg; Samir Mehta; John P Dormans
Journal:  J Bone Joint Surg Am       Date:  2004-08       Impact factor: 5.284

10.  Multifocal Skeletal Tuberculosis Mimicking Langerhans Cell Histiocytosis in a Child: a Case Report With a Long-Term Follow-Up.

Authors:  Hamidreza Haghighatkhah; Yousef Jafroodi; Morteza Sanei Taheri; Ramin Pourghorban; Afarin Sadeghian Dehkordy
Journal:  Iran Red Crescent Med J       Date:  2015-12-01       Impact factor: 0.611

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