Clivus chordoma and torticollis in children, plus chemotherapy.

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Search completed for: 

Search required by: 14 March 2012 

Search completed on: 12 March 2012 

Search completed by: Richard Bridgen 

Search details 

Clivus chordoma and torticollis in children plus chemotherapy. 

Resources searched 

NHS Evidence; TRIP Database; Cochrane Library; AMED; BNI; CINAHL; EMBASE; HMIC; 

MEDLINE; PsychINFO; Google Scholar 

Database search terms : clivus adj2 chordoma; CHORDOMA; CRANIAL FOSSA, 

POSTERIOR; clivus; “posterior cranial fossa”; cancer*; exp NEOPLASMS; neoplasm*; 

tumor*; tumour*; chordoma*; torticol*; TORTICOLLIS; “cervical dystonia”; wryneck; torsion 

adj2 neck; chordocarcinoma; notochordoma; chordoepithelioma; “sphenoid bone”; 

SPHENOID BONE; “basilar occipital bone”; “occipital bone”; OCCIPITAL BONE; sphenooccipital; 

sphenooccipital; child*; exp CHILD; adolescent*; exp ADOLESCENT; juvenile*; 

teenager*; “young person*”; pediatric*; paediatric*; exp PEDIATRICS; chemotherapy*; exp 

CHEMOTHERAPY; exp DRUG THERAPY 

Google search string : "~clivus" (chordoma OR "posterior cranial fossa") (~torticollis OR 

"cervical dystonia" OR wryneck) 

Summary 

There is some research on clivus chordoma and torticollis in children but nothing with the 

addition of chemotherapy to the search beyond a tantalising result found in Google Scholar 

(see item 12). 

Guidelines 

None found 

Evidence-based reviews 

1

None found 

Published research 

1. Clival osteomyelitis mimics central skull base neoplasm in a healthy 7-yearold 

boy 

Author(s): Liang M., Chang F., Wong T. 

Citation: Neuro-Oncology, June 2010, vol./is. 12/6(ii133), 1522-8517 (June 2010) 

Publication Date: June 2010 

Abstract: A 7-year-old boy presented with severe painful torticollis, headache and neck 

stiffness for 20 days. Progressive neck pain with fever happened 2 days before admission. 

Elevated white blood cell and inflammatory reactive protein were also noted. CT scan of 

brain and cervical spine revealed an osteolytic lesion with soft tissue mass over clivus, 

suspected to be leukemia, eosinophilic granuloma, Ewing sarcoma or chordoma. MRI of 

brain disclosed an enhancing margin lesion over clivus with a few fluid accumulations over 

retropharyneal space. Initially, central skull base neoplasm with related meningitits was 

impressed; however, the diagnosis changed to clival osteomyelitis after fine needle CTguided 

biopsy through pterygo-mandibular fossa. The pathology of the specimens showed 

lymphoid cells and histiocytes including CD20(+) B-lymphoid cell, CD3(+) T-lymphoid cells, 

CD138(+) plasma cells, neurtrophils and eosinophils, and negative for malignant cells. 

Antibiotic treatment using clindamycin and claforan started and continued for 4 weeks. The 

symtoms of neck pain and stiffness dramatically decreased after 3 days of treatment and 

mostly subsided in one week. The follow-up images after 3 months showed complete 

resolution of inflammation without recurrence. Clival osteomyelitis could present clival 

syndrome and mimic central skull base neoplasm. This problem mainly happens in old age 

and immuno-compromised patients, or results from spread of infection through otitis media, 

sphenoid sinusitis, or fossa navicularis magna. The possible cause of clival osteomyelitis in 

this healthy 7-year-old boy might attribute to innate fossa navicularis magna with lymphoid 

tissue residing. 

Source: EMBASE 

Full Text: 

Available in fulltext at EBSCOhost 

Available in fulltext at National Library of Medicine 

Available in print at a non-ULHT hospital library. Click and complete an online form to 

request this article/an article from this journal if fulltext is not available. 

2. Intracranial tumors in first year of life: The CHEO experience 

Author(s): Mehrotra N., Shamji M.F., Vassilyadi M., Ventureyra E.C.G. 

Citation: Child's Nervous System, December 2009, vol./is. 25/12(1563-1569), 0256-7040 

(December 2009) 

Publication Date: December 2009 

Abstract: Introduction: One seventh of pediatric brain tumors are diagnosed in the first 

year of life. With more widespread and accessible neuroimaging, these lesions are being 

diagnosed earlier, but there remains scant literature about their natural history. Methods: A 

retrospective review was performed of brain tumor patients presenting to the Children"s 

Hospital of Eastern Ontario (CHEO) through the last 34 years. Patients presenting in the 

first year of life, including symptoms, management features, and functional outcome, were 

analyzed using ANOVA and chi 2 statistics. Results: Eighteen cases of brain tumors in the 

first year of life were identified: 12 suptratentorial, eight with benign histology, and six 

infratentorial all with malignant histology. Median age of presentation differed by lesion 

location (p=0.05) and glial tumors were most common. Raised intracranial pressure was 

more than twice as prevalent with posterior fossa lesions (p

cerebrospinal fluid diversion was more frequently necessary among infratentorial lesions 

(p=0.02), and adjuvant therapy was more utilized for infratentorial lesions (p

this series (6.6 years) is consistent with a decreasing trend over the past 70 years. 

Headaches, ataxia and torticollis emerge as significant symptoms worthy of further 

investigation whilst abdominal pain and constipation might herald the presence of a 

posterior fossa tumour on rare occasions. 

Source: MEDLINE 

Full Text: 



5. Torticollis secondary to posterior fossa tumors. 

Author(s): Turgut M 

Citation: Journal of Pediatric Orthopedics, May 1998, vol./is. 18/3(415), 0271-6798;0271- 

6798 (1998 May-Jun) 

Publication Date: May 1998 


Full Text: 

Available in fulltext at Ovid 

6. Cervical dystonia associated with tumors of the posterior fossa. 

Author(s): Krauss JK, Seeger W, Jankovic J 

Citation: Movement Disorders, May 1997, vol./is. 12/3(443-7), 0885-3185;0885-3185 

(1997 May) 

Publication Date: May 1997 

Abstract: Cervical dystonia was associated with posterior fossa tumors in three patients. 

The onset of dystonia paralleled the appearance of other focal neurologic signs. All patients 

had extraaxial tumors located in the cerebellopontine angle that were removed via 

suboccipital approaches. The tumors were identified as schwannomas arising from the 

glossopharyngeal nerve and from the vagus/accessory nerves; and a meningioma. 

Postoperatively, the cervical dystonia improved markedly during a period of 8 years in one 

patient, and it remitted completely within 1 year in another patient. In the third patient, 

cervical dystonia persisted. The combination of the clinical findings and the temporal 

relationship of their appearance suggest a causal association between the posterior fossa 

tumors and cervical dystonia in three cases. Possible pathogenic mechanisms are 

reviewed. 


7. Torticollis secondary to posterior fossa tumors. 

Author(s): Gupta AK, Roy DR, Conlan ES, Crawford AH 

Citation: Journal of Pediatric Orthopedics, July 1996, vol./is. 16/4(505-7), 0271-6798;0271- 

6798 (1996 Jul-Aug) 

Publication Date: July 1996 

Abstract: Torticollis in childhood may be a sign of many disorders. Five cases, with 

torticollis as the initial sign of a posterior fossa tumor, are presented. The diagnosis and 

treatment of the tumor was considerably delayed in all patients because posterior fossa 

tumor was not considered in the initial differential diagnosis. In two patients, operative 

procedures on the sternocleidomastoid muscle were performed before discovering the 

underlying causative tumors. Four of the five patients also had other associated symptoms 

such as headache, nausea, and vomiting. It is stressed that in acquired torticollis, posterior 

fossa tumor be considered in the differential diagnosis. 


Full Text: 

4

Available in fulltext at Ovid 

8. Photophobia, epiphora, and torticollis: a masquerade syndrome. 

Author(s): Marmor MA, Beauchamp GR, Maddox SF 

Citation: Journal of Pediatric Ophthalmology & Strabismus, July 1990, vol./is. 27/4(202-4), 

0191-3913;0191-3913 (1990 Jul-Aug) 

Publication Date: July 1990 

Abstract: Three young children presented with photophobia, epiphora, and torticollis as the 

initial manifestation of a posterior fossa tumor. In each case there was a delay in treatment 

due to the presumptive diagnosis of a local ocular inflammatory condition. We recommend 

that children with unexplained photophobia, epiphora, and torticollis undergo an imaging 

technique to evaluate the posterior fossa. 


Full Text: 



9. Metastasizing chordoma in early childhood: a pathological and 

immunohistochemical study with review of the literature. 

Author(s): Sibley RK, Day DL, Dehner LP, Trueworthy RC 

Citation: Pediatric Pathology, 1987, vol./is. 7/3(287-301), 0277-0938;0277-0938 (1987) 

Publication Date: 1987 

Abstract: A 2 1/2-year-old female with a sphenooccipital-vertebral chordoma presented 

with neck pain, torticollis, fever, a lytic lesion of C2 vertebra, and bilateral nodular infiltrates 

in the lung. The lung biopsy revealed multiple tumor emboli by an enigmatic epithelioidappearing 

neoplasm with immunohistochemical staining for vimentin, cytokeratin, and 

epithelial membrane antigen. A thorough roentgenographic evaluation disclosed a 

destructive, prepontine mass in the region of the clivus, erosion of the odontoid process, 

and compression of the cervical spinal cord. The patient died after a clinical course of 3 

months. We identified 16 additional cases of metastasizing chordomas in the pediatric-age 

population; this case is the first to our knowledge with pathologically documented 

pulmonary metastasis at presentation. 


Full Text: 



10. Benign paroxysmal vertigo in childhood. 

Author(s): Eeg-Olofsson O, Odkvist L, Lindskog U, Andersson B 

Citation: Acta Oto-Laryngologica, March 1982, vol./is. 93/3-4(283-9), 0001-6489;0001- 

6489 (1982 Mar-Apr) 

Publication Date: March 1982 

Abstract: Fifteen children with a syndrome called benign paroxysmal vertigo are 

presented. This syndrome is characterized by attacks of vertigo of sudden onset lasting a 

few minutes and in extreme cases several hours or even up to 2 days. Additional 

symptoms are pallor, sweating, vomiting and nystagmus. Consciousness is not impaired. 

The attacks usually occur in the first 4 years of life. They are recurrent, the appearance 

varying from several times a week to once a year, and they may cease spontaneously after 

only months but usually years. The most important differential diagnoses are epilepsy and 

posterior fossa tumour. The etiology is unknown, but there may be a vascular disturbance 

affecting the posterior cerebral circulation with secondary disturbances of the vestibular 

nuclei. 

5


Full Text: 



11. Malignant intracranial chordoma and sarcoma of the clivus in infancy. 

Author(s): Nolte K 

Citation: Pediatric Radiology, February 1979, vol./is. 8/1(1-6), 0301-0449;0301-0449 (1979 

Feb 26) 

Publication Date: February 1979 

Abstract: Neuroradiological and clinical findings suggesting chordoma of the clivus are 

described in three children. They presented with torticollis as the primary and predominant 

symptom. Progressive deterioration with development of slurred speech, signs of 

intracranial pressure and further cranial nerve involvement led to more extensive 

neuroradiological investigation. The clinical and neuroradiological findings in these three 

children were similar; the prognosis was unfavourable for two of the children. The 

histological diagnosis in two cases was malignant chordoma; in the third case it was 

undifferentiated sarcoma of the clivus. 


Full Text: 



Google Scholar 

From 1 st 50 results… 

12. The role of chemotherapy in pediatric clival chordomas 

G Dhall, M Traverso, JL Finlay, L Shane… - Journal of neuro- …, 2011 - Springer 

... and outcome of six cases of clival chordoma in children Pt no. Age at Dx Sex 

Clinical features Surgery Pathology Initial treatment Response Time to relapse 

(months) Treatment at relapse Complications Survival 1 2 years F Ataxia, torticollis 

and head tilt Subtotal resection ... 

Cited by 3 - Related articles - Lancashire Teaching Hospitals - Find@The Christie - All 

4 versions 

13. Craniovertebral junction neoplasms in the pediatric population 

AH Menezes - Child's Nervous System, 2008 - Springer 

... Tumors arising in this location are summarized in Table 1. In those children with 

torticollis and radiographic evidence of rotary subluxation or atlantoaxial dislocation, 

the need for stabilization and fusion is made at the ... Chordoma ... Six of these were 

chordomas affecting the clivus. ... 

Cited by 19 - Related articles - Find@The Christie - All 2 versions 

14. Basilar invagination, Chiari malformation, syringomyelia: A review 

A Goel - Neurology India, 2009 - Medknow 

... [10] Short neck, low hairline, web-shaped neck muscles, torticollis, reduction in the 

range of neck 

movements, and several such physical variations have been ... The angle of the clivus 

and the 

posterior cranial fossa volume were essentially unaffected in these patiens. ... 

Cited by 3 - Related articles - Lancashire Teaching Hospitals - All 6 versions 

15. Primary bone tumors of the spine in children 

AJ Fenoy, JDW Greenlee, AH Menezes… - Journal of …, 2006 - thejns.org 

... Three patients exhibited torticollis. A summary of presentations is provided inTable 

1. ... Neurosurgery 17:317–319, 1985. 26. Handa J, Suzuki F, Nioka H, Koyama T: 

6

Clivus chordoma in childhood. Surg Neurol 28:58–62, 1987. 27. ... 

Cited by 20 - Related articles - BL Direct - All 3 versions 

16. Differential Diagnosis of Clival Tumors 

VC TRAYNELIS… - … of the skull base and spine, 2003 - books.google.com 

... be held in a stiff and fixed position, sometimes resembling torticollis. ... 26 

Demographics, Presentation, and Diagnosis TABLE 4-1. Clival Pathology Congenital 

... disease Osteogenesis imperfecta Eosinophilic granuloma Fibrous dysplasia 

Chordoma Chondroma Chondrosarcoma ... 

Related articles 

17. Common Presentation of Clival Tumors 

VC TRAYNELIS… - … of the skull base and spine, 2003 - books.google.com 

... The head may be held in a stiff and fixed position, sometimes resembling torticollis. 

... Although these tumors usually are associated with a history of slowly progressive 

symptoms, Simonsen reported a patient with a clivus chordoma that presented 

acutely with a fatal subarachnoid ... 

Related articles 

18. Meningioma at the Craniocervical Junction 

L Ibañez-Valdés, J Moré-Rodriguez… - The Internet Journal of …, 2010 - ispub.com ... 

acquired abnormalities are: injury or disease that may involve occipital bone (foramen 

magnum/clivus), the first ... disease of the cervical spine, slowly growing CCJ tumours 

(neurofibroma, shwannomas, meningioma, and chordoma) that can ... Torticollis was 

seen in the very young. ... 

Related articles - Cached - All 2 versions 

19. Surgical treatment of clival chordomas: the transsphenoidal approach revisited 

G Maira, R Pallini, C Anile, E Fernandez… - Journal of …, 1996 - thejns.org 

... CT) scan showing the relationship of a lower clival chordoma with the right occipital 

condyle. B: Postoperative nonenhanced axial CT scan showing the right dorsal 

condylectomy for the extreme-lateral transcondylar approach. This patient suffered 

from spasmodic torticollis. ... 

Cited by 64 - Related articles - BL Direct - All 5 versions 

7

Search strategy 

No. Database Search term Hits 

1 MEDLINE (clivus adj2 chordoma*).ti,ab 68 

2 MEDLINE CHORDOMA/ AND CRANIAL FOSSA, POSTERIOR/ 180 

3 MEDLINE clivus.ti,ab 1209 

4 MEDLINE CRANIAL FOSSA, POSTERIOR/ 3701 

5 MEDLINE 3 OR 4 4528 

6 MEDLINE cancer*.ti,ab 895034 

7 MEDLINE NEOPLASMS/ 240680 

8 MEDLINE neoplasm*.ti,ab 87962 

9 MEDLINE (tumor* OR tumour*).ti,ab 1008113 

10 MEDLINE chordoma*.ti,ab 2413 

11 MEDLINE CHORDOMA/ 2395 

12 MEDLINE 6 OR 7 OR 8 OR 9 OR 10 OR 11 1695273 

13 MEDLINE 5 AND 12 1669 

14 MEDLINE torticollis.ti,ab 2371 

15 MEDLINE TORTICOLLIS/ 2797 

16 MEDLINE "cervical dystonia".ti,ab 812 

17 MEDLINE wryneck.ti,ab 24 

18 MEDLINE chordocarcinoma*.ti,ab 1 

19 MEDLINE notochordoma*.ti,ab 0 

20 MEDLINE chordoepithelioma*.ti,ab 0 

21 MEDLINE (torsion adj2 neck).ti,ab 51 

22 MEDLINE 14 OR 15 OR 16 OR 17 OR 21 3908 


24 MEDLINE 1 OR 2 221 


26 MEDLINE "sphenoid bone".ti,ab 514 

27 MEDLINE SPHENOID BONE/ 3047 

28 MEDLINE "basilar occipital bone".ti,ab 0 

29 MEDLINE "occipital bone".ti,ab 668 

30 MEDLINE OCCIPITAL BONE/ 2612 

8


31 MEDLINE spheno-occipital.ti,ab 134 

32 MEDLINE sphenooccipital.ti,ab 34 

33 MEDLINE 26 OR 27 3280 

34 MEDLINE 29 OR 30 2955 


36 MEDLINE 31 OR 32 OR 35 333 



39 EMBASE (clivus adj2 chordoma*).ti,ab 79 

40 EMBASE CHORDOMA/ AND CRANIAL FOSSA, POSTERIOR/ 66 

41 EMBASE clivus.ti,ab 1367 

42 EMBASE CRANIAL FOSSA, POSTERIOR/ 4771 

43 EMBASE 41 OR 42 5985 

44 EMBASE cancer*.ti,ab 1074749 

45 EMBASE NEOPLASMS/ 173371 

46 EMBASE neoplasm*.ti,ab 97265 

47 EMBASE (tumor* OR tumour*).ti,ab 1140585 

48 EMBASE chordoma*.ti,ab 2677 

49 EMBASE CHORDOMA/ 3103 

50 EMBASE 44 OR 45 OR 46 OR 47 OR 48 OR 49 1922572 

51 EMBASE 43 AND 50 1930 

52 EMBASE torticollis.ti,ab 2710 

53 EMBASE TORTICOLLIS/ 3164 

54 EMBASE "cervical dystonia".ti,ab 1154 

55 EMBASE wryneck.ti,ab 27 

56 EMBASE chordocarcinoma*.ti,ab 1 

57 EMBASE notochordoma*.ti,ab 0 

58 EMBASE chordoepithelioma*.ti,ab 0 

59 EMBASE (torsion adj2 neck).ti,ab 64 

60 EMBASE 52 OR 53 OR 54 OR 55 OR 59 4940 

61 EMBASE 51 AND 60 6 

62 EMBASE 39 OR 40 136 

9



64 EMBASE "sphenoid bone".ti,ab 556 

65 EMBASE SPHENOID BONE/ 2558 

66 EMBASE "basilar occipital bone".ti,ab 0 

67 EMBASE "occipital bone".ti,ab 716 

68 EMBASE OCCIPITAL BONE/ 2215 

69 EMBASE spheno-occipital.ti,ab 120 

70 EMBASE sphenooccipital.ti,ab 38 

71 EMBASE 64 OR 65 2772 

72 EMBASE 67 OR 68 2495 

73 EMBASE 71 AND 72 177 

74 EMBASE 69 OR 70 OR 73 290 

75 EMBASE 50 AND 74 77 


77 EMBASE 39 OR 40 OR 51 1930 


79 EMBASE 39 OR 40 OR 51 OR 75 1988 

80 EMBASE child*.ti,ab 988171 

81 EMBASE exp CHILD/ 1526385 

82 EMBASE adolescent*.ti,ab 152789 

83 EMBASE exp ADOLESCENT/ 1137780 

84 EMBASE juvenile*.ti,ab 55757 

85 EMBASE teenager*.ti,ab 10669 

86 EMBASE youth*.ti,ab 35617 

87 EMBASE "young person*".ti,ab 2701 

88 EMBASE paediatric*.ti,ab 47025 

89 EMBASE exp PEDIATRICS/ 62743 

90 EMBASE pediatric*.ti,ab 193955 

91 EMBASE 

80 OR 81 OR 82 OR 83 OR 84 OR 85 OR 86 OR 87 OR 88 

OR 89 OR 90 

2484811 

92 EMBASE 79 AND 91 793 

93 EMBASE chemotherap*.ti,ab 288989 

94 EMBASE exp CHEMOTHERAPY/ 279430 

10


95 EMBASE 93 OR 94 431550 

96 EMBASE 92 AND 95 173 


98 EMBASE torticol*.ti,ab 2764 

99 EMBASE 60 OR 98 4960 

100 EMBASE 79 AND 99 6 

101 MEDLINE chemotherap*.ti,ab 232438 

102 MEDLINE exp DRUG THERAPY/ 963089 

103 MEDLINE 101 OR 102 1107149 

104 MEDLINE 1 OR 2 OR 13 OR 37 1733 

105 MEDLINE 103 AND 104 103 

106 MEDLINE torticol*.ti,ab 2409 

107 MEDLINE 22 OR 106 3932 

108 MEDLINE 105 AND 107 0 

109 MEDLINE exp NEOPLASMS/ 2295723 

110 MEDLINE 6 OR 8 OR 9 OR 10 OR 11 OR 109 2644233 

111 MEDLINE 5 OR 36 4813 

112 MEDLINE 110 AND 111 2765 

113 MEDLINE 107 AND 112 10 

114 EMBASE exp NEOPLASM/ 2737958 

115 EMBASE 50 OR 114 3070243 

116 EMBASE 43 AND 115 2959 

117 EMBASE 62 OR 77 OR 116 2959 

118 EMBASE 99 AND 117 7 

11

Clivus chordoma and torticollis in children, plus chemotherapy.

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