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Osteoid osteoma of the rib: A case report
Osteoid
Osteoma
By J.C. Hoeffel,
P. Lascombes,
of the Rib: A Case Report C. Delgoffe,
Vandoeuvre-les-Nancy, l A case of a 6-year-old boy with a painful dorsal scoliosis secondary to an osteoid osteoma in a rib is described. Surgical excision was performed and the scoliosis decreased. Such cases are very rare. Copyright
o 1993 by W.B. Saunders Company
INDEX WORDS:
Osteoid osteoma, rib; scoliosis, dorsal.
0
STEOID OSTEOMA, first defined by Jaffe in 193.5,’ is classified as a neoplasm because of histological features but with inflammatory characteristics because of its clinical presentation and radiographic changes. The occurence of an osteoid osteoma in a rib is extremely rare. CASE REPORT A 6%-year-old boy had a left dorsolumbar scoliosis of 38” since 3 months (Fig 1). The scoliosis was treated by kinesitherapy. For 2 months he had pain in the back mainly on the left side and during the night. He had an asymmetrical thorax with bulging of the left hemithorax. There was a well-defined osteolytic area of the posterior arch of the eighth rib in the curvature of the scoliosis. which looked like an eosinophilic granuloma (Fig 2). There was thickening of adjacent posterior area of this eighth rib medial to the geode and close to the spine. Technetium scan showed increased uptake on the left eighth rib. Computed tomography (CT) showed a geode of the left eighth rib of 0.5 x 0.8 cm with a density of 74 Hounsfield units, and central calcifications on a few scans; there was a break of the cortex on two cuts. There was no soft tissue extension (Fig 3). Surgical excision was performed and a diagnosis of osteoid osteoma made established. The scoliosis has decreased from 3X” to 18” 2 months following surgery.
B. Aymard,
and M.A. Galley
France
area of increased uptake confined to the posterior aspect of the right seventh rib. Lynch and Dorgan’ also reported a case of osteoid osteoma of a rib as a cause of scoliosis in an 11-year-old girl. A YymT~bone scan showed an area of increased uptake to the left lateral aspect of the body of T8. Tomograms of the posterior aspect of the left lower rib demonstrated an expanded lesion in the head of the eighth rib. Ten weeks after the initial presentation, excision of the posterior end of the eighth rib was performed. Intraoperative radiography control was used to identify the correct rib. It was an osteoid osteoma and excision was complete. Following excision of the tumor, the patient’s scoliosis resolved rapidly and there has been no sign of recurrence 18 months after surgery. Nelson et alh described a 15year-old boy with an osteoid osteoma of the sixth rib that contained a single nidus but also showed inflammatory involvement of two adjacent ribs and a focal area of pleural reaction. Abendschein et al’ described a young man of 20 years with a 7-mm geode of the nineth rib with calcification. The technetium scan showed increased
DISCUSSION
Only two cases of rib osteoid osteoma were reported before 1960 in the literature,*,” but other cases have recently been reported (Table 1). Kehl et al4 reported a 7-year-old boy with a .5-month history of dull back pain of spontaneous onset located in the thoracic spine. Radiographs of the spine showed a left thoracic scoliosis. A lytic lesion in the posterosuperior margin of the neck of the right seventh rib was identified. A technetium99m polyphosphate bone scan showed an isolated From the Departments of Radiology and Surgery. Children k Hospital. and the Depatiment of Pathology, Adult? Hospital, Vandoeuvreles-Nancy, France. Address reprint requests to J.C. HoeffeL MD, Department of Radiology. Children’s Hospital, AlEe du Morvan. 54511 Vandoemzreies-Nancy. France. Copyright 0 1993 by W.B. Saunders Company 0022-346819312805-0025$03.OOiO 738
Fig 1. Scoliosis with left concave curvature. The posterior arch of the left eighth rib is dense close to the spine with a geode laterally. JournalofPediafric Surgery, Vol 28, No 5 (May), 1993: pp 738-740
OSTEOID OSTEOMA
OF THE RIB
Fig 3. CT scan shows small destructive lesion on the posterior part of the eighth left rib with considerable new bone formation and with calcifications inside and a break through the cortex. There is no soft tissue extension of the lesion.
locations are much more common than in the rib. Freiberger reported two cases of osteoid osteoma of the spine in two children.“’ Maclellan and Wilson” described three cases of osteoid osteoma of the spine and reviewed 36 welldocumented cases in the literature. Keim and Reina” reported nine lesions in the spine. Nelson and Greer’” reported the value of CT for the diagnosis of an osteoid osteoma of the posterior arch. The average length of time between the onset of symptoms and the diagnosis of osteoid osteoma is of 3 to 5 months in the cases reported.J-’ This delay is due to the lack of dramatic radiographic changes and a lack of awareness by physicians. Back ache in children or young adults is not a common complaint. When it is present and is associated with a scoliosis, an infective or neoplastic lesion of the spine must be considered. In patients with osteoid osteoma. CT demonstrates the radiolucent nidus with a smooth inner surface and in some cases a radiodense center within the nidus can be visualized. In the cases with a radiodense center the calcified density is located in the center of the lucent nidus. It is considered to represent the osteoblastic center of the lesion.
Fig 2. Frontal view of the eighth left rib. Well-defined osteolytic area of the posterior part of the rib. The cortical part of the rib look normal.
uptake on the rib. Histology showed it was an osteoid osteoma. A case of scoliosis secondary to osteoblastoma of the rib has been reported by Wimpee et al.x An X-year-old girl had a progressive painless spinal deformity. She was found to have an atypical scoliosis secondary to a stage three benign osteoblastoma of the rib. Two cases of osteoblastoma of a rib associated with progressive scoliosis have been reported by Fabris et al.” The girls were 11 years and 4 years old. Spinal
Table 1. Costal Osteoid Osteoma and Osteoblastoma Central Diagnosis lnvestlgator
lage)
Nelson et aI6
Osteoid osteoma
Radiolucent LeStOn
(15 yr)
+
Dense Fleck of Bone
Withln
the Center of
the Radlolucency
L
of the Rib
Well-Defined
-
Rim
of Increased
Regresswe
Bone Density
Scoliosls
7
i
Miscellaneous
Adjacent
sclerotic
ribs
and pleural reaction Kehl et al4
Osteoid osteoma
(7 yr 9 mo)
+
+
+
+
Lynch
Osteoid osteoma
(11 yr)
+
7
+
+
+
+ 7
+ 7
+
7
+
i
Adjacent
7
+
+
Nodular aspect 7
+
i
et aI5
Abendschein
et al’
Osteoid osteoma
(20 yr)
Wimpee et ale
Osteoblastoma
(8 yr)
Fabris et aI9
Osteoblastoma
(11 yr)
Osteoblastoma
(4 yr)
sclerotic
ribs
HOEFFEL ET AL
740
The spinal curvature initially visible consists of an antalgic non structural lateral deviation. The associated scoliosis may result from muscle spasm caused by a painful osseous focus located around the spine. The curve usually has a c-shaped configuration with the irritating lesion usually located on the concave side of the curve near the apex. Rotation of the atypical vertebra is often mild. The scoliotic curve is initially functional and will resolve after the painfu1 focus has been removed. In some patients, however, the curve may not be completely corrected because of vertebral structural changes that have occured with time. The differential diagnosis of painful lesions occur-
ing around the ribs should always include tumors of bone. Eosinophilic granuloma, osteoid osteoma, osteoblastoma, and aneurysmal bone cyst of the posterior vertebral elements are the most common lesion found in the first, second and third decades of life. Mehta14 found that the complex of osteoid osteoma and osteoblastoma is the most frequent tumor found when back pain is accompanied by scoliosis in the preadolescent patient.
ACKNOWLEDGMENT The authors Southampton,
thank
Dr P. Guyer.
England
Royal
South
Hants
Hospital.
for his help.
REFERENCES 1. Jaffe
HL: Osteoid osteoma. A benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg 31:709-72X, 1935 2. Jackson IJ: Osteoid osteoma of lamina and its treatment. Am Surg 19:17-23, 1953 3. Mauer I: Osteoid osteoma of the 12th rib. Resection under local anesthesia. A case report. Milit Med 122:194, 1958 4. Kehl DR. Alonso JE, Lovell WW: Scoliosis secondary to an osteoid osteoma of the rib. J Bone Joint Surg (Am) 65:701-703. 1983 5. Lynch MC, Dorgan JC: Osteoid osteoma of a rib as a cause of scoliosis. A case report. Spine 11:480-482, 1986 6. Nelson MC, Brower AC, Ragsdale BD: Case report 448. Skeletal Radio1 16:601-603, 1987 7. Abendschein T, Ulrich C, Kampmann I: Das Osteoidosteom: Bei ungunstiger Lokalisation schwierige Diagnose. Fortschr Rontgenstr 151:229-230. 1989 8. Wimpee MW, Maale GE, Hudkins PG. et al: Case report.
Scoliosis secondary 7:58Y-593. 1987
to osteoblastoma
9. Fabris D, Trainiti osteoblastoma: Report 1983 IO. Freiberger X:232-235,1960
of the rib. J Pediatr
Orthop
G, Di Comun M, et al: Scoliosis due to rib of two cases. J Pediatr Orthop 3:370-375.
RH: Osteoid
osteoma
I I. Maclellan DI. Wilson FC: Osteoid Bone Joint Surg (Am) 49:111-121. 1967
of the spine. osteoma
Radiology
of the spine. J
12. Keim HA, Reina EG: Osteoid-osteoma as a cause scoliosis. J Bone Joint Surg (Am) 57:159-163. 1975
of
13. Nelson OA, Greer RB: Localization of osteoid-osteoma of the spine using computerized tomograph. J Bone Joint Surg (Am) 65:263-265. 1983 14. Mehta MH: Pain provoked scoliosis. Observations evolution of deformity. Clin Orthop 135:5X-65. 1978
on the
Osteoma
By J.C. Hoeffel,
P. Lascombes,
of the Rib: A Case Report C. Delgoffe,
Vandoeuvre-les-Nancy, l A case of a 6-year-old boy with a painful dorsal scoliosis secondary to an osteoid osteoma in a rib is described. Surgical excision was performed and the scoliosis decreased. Such cases are very rare. Copyright
o 1993 by W.B. Saunders Company
INDEX WORDS:
Osteoid osteoma, rib; scoliosis, dorsal.
0
STEOID OSTEOMA, first defined by Jaffe in 193.5,’ is classified as a neoplasm because of histological features but with inflammatory characteristics because of its clinical presentation and radiographic changes. The occurence of an osteoid osteoma in a rib is extremely rare. CASE REPORT A 6%-year-old boy had a left dorsolumbar scoliosis of 38” since 3 months (Fig 1). The scoliosis was treated by kinesitherapy. For 2 months he had pain in the back mainly on the left side and during the night. He had an asymmetrical thorax with bulging of the left hemithorax. There was a well-defined osteolytic area of the posterior arch of the eighth rib in the curvature of the scoliosis. which looked like an eosinophilic granuloma (Fig 2). There was thickening of adjacent posterior area of this eighth rib medial to the geode and close to the spine. Technetium scan showed increased uptake on the left eighth rib. Computed tomography (CT) showed a geode of the left eighth rib of 0.5 x 0.8 cm with a density of 74 Hounsfield units, and central calcifications on a few scans; there was a break of the cortex on two cuts. There was no soft tissue extension (Fig 3). Surgical excision was performed and a diagnosis of osteoid osteoma made established. The scoliosis has decreased from 3X” to 18” 2 months following surgery.
B. Aymard,
and M.A. Galley
France
area of increased uptake confined to the posterior aspect of the right seventh rib. Lynch and Dorgan’ also reported a case of osteoid osteoma of a rib as a cause of scoliosis in an 11-year-old girl. A YymT~bone scan showed an area of increased uptake to the left lateral aspect of the body of T8. Tomograms of the posterior aspect of the left lower rib demonstrated an expanded lesion in the head of the eighth rib. Ten weeks after the initial presentation, excision of the posterior end of the eighth rib was performed. Intraoperative radiography control was used to identify the correct rib. It was an osteoid osteoma and excision was complete. Following excision of the tumor, the patient’s scoliosis resolved rapidly and there has been no sign of recurrence 18 months after surgery. Nelson et alh described a 15year-old boy with an osteoid osteoma of the sixth rib that contained a single nidus but also showed inflammatory involvement of two adjacent ribs and a focal area of pleural reaction. Abendschein et al’ described a young man of 20 years with a 7-mm geode of the nineth rib with calcification. The technetium scan showed increased
DISCUSSION
Only two cases of rib osteoid osteoma were reported before 1960 in the literature,*,” but other cases have recently been reported (Table 1). Kehl et al4 reported a 7-year-old boy with a .5-month history of dull back pain of spontaneous onset located in the thoracic spine. Radiographs of the spine showed a left thoracic scoliosis. A lytic lesion in the posterosuperior margin of the neck of the right seventh rib was identified. A technetium99m polyphosphate bone scan showed an isolated From the Departments of Radiology and Surgery. Children k Hospital. and the Depatiment of Pathology, Adult? Hospital, Vandoeuvreles-Nancy, France. Address reprint requests to J.C. HoeffeL MD, Department of Radiology. Children’s Hospital, AlEe du Morvan. 54511 Vandoemzreies-Nancy. France. Copyright 0 1993 by W.B. Saunders Company 0022-346819312805-0025$03.OOiO 738
Fig 1. Scoliosis with left concave curvature. The posterior arch of the left eighth rib is dense close to the spine with a geode laterally. JournalofPediafric Surgery, Vol 28, No 5 (May), 1993: pp 738-740
OSTEOID OSTEOMA
OF THE RIB
Fig 3. CT scan shows small destructive lesion on the posterior part of the eighth left rib with considerable new bone formation and with calcifications inside and a break through the cortex. There is no soft tissue extension of the lesion.
locations are much more common than in the rib. Freiberger reported two cases of osteoid osteoma of the spine in two children.“’ Maclellan and Wilson” described three cases of osteoid osteoma of the spine and reviewed 36 welldocumented cases in the literature. Keim and Reina” reported nine lesions in the spine. Nelson and Greer’” reported the value of CT for the diagnosis of an osteoid osteoma of the posterior arch. The average length of time between the onset of symptoms and the diagnosis of osteoid osteoma is of 3 to 5 months in the cases reported.J-’ This delay is due to the lack of dramatic radiographic changes and a lack of awareness by physicians. Back ache in children or young adults is not a common complaint. When it is present and is associated with a scoliosis, an infective or neoplastic lesion of the spine must be considered. In patients with osteoid osteoma. CT demonstrates the radiolucent nidus with a smooth inner surface and in some cases a radiodense center within the nidus can be visualized. In the cases with a radiodense center the calcified density is located in the center of the lucent nidus. It is considered to represent the osteoblastic center of the lesion.
Fig 2. Frontal view of the eighth left rib. Well-defined osteolytic area of the posterior part of the rib. The cortical part of the rib look normal.
uptake on the rib. Histology showed it was an osteoid osteoma. A case of scoliosis secondary to osteoblastoma of the rib has been reported by Wimpee et al.x An X-year-old girl had a progressive painless spinal deformity. She was found to have an atypical scoliosis secondary to a stage three benign osteoblastoma of the rib. Two cases of osteoblastoma of a rib associated with progressive scoliosis have been reported by Fabris et al.” The girls were 11 years and 4 years old. Spinal
Table 1. Costal Osteoid Osteoma and Osteoblastoma Central Diagnosis lnvestlgator
lage)
Nelson et aI6
Osteoid osteoma
Radiolucent LeStOn
(15 yr)
+
Dense Fleck of Bone
Withln
the Center of
the Radlolucency
L
of the Rib
Well-Defined
-
Rim
of Increased
Regresswe
Bone Density
Scoliosls
7
i
Miscellaneous
Adjacent
sclerotic
ribs
and pleural reaction Kehl et al4
Osteoid osteoma
(7 yr 9 mo)
+
+
+
+
Lynch
Osteoid osteoma
(11 yr)
+
7
+
+
+
+ 7
+ 7
+
7
+
i
Adjacent
7
+
+
Nodular aspect 7
+
i
et aI5
Abendschein
et al’
Osteoid osteoma
(20 yr)
Wimpee et ale
Osteoblastoma
(8 yr)
Fabris et aI9
Osteoblastoma
(11 yr)
Osteoblastoma
(4 yr)
sclerotic
ribs
HOEFFEL ET AL
740
The spinal curvature initially visible consists of an antalgic non structural lateral deviation. The associated scoliosis may result from muscle spasm caused by a painful osseous focus located around the spine. The curve usually has a c-shaped configuration with the irritating lesion usually located on the concave side of the curve near the apex. Rotation of the atypical vertebra is often mild. The scoliotic curve is initially functional and will resolve after the painfu1 focus has been removed. In some patients, however, the curve may not be completely corrected because of vertebral structural changes that have occured with time. The differential diagnosis of painful lesions occur-
ing around the ribs should always include tumors of bone. Eosinophilic granuloma, osteoid osteoma, osteoblastoma, and aneurysmal bone cyst of the posterior vertebral elements are the most common lesion found in the first, second and third decades of life. Mehta14 found that the complex of osteoid osteoma and osteoblastoma is the most frequent tumor found when back pain is accompanied by scoliosis in the preadolescent patient.
ACKNOWLEDGMENT The authors Southampton,
thank
Dr P. Guyer.
England
Royal
South
Hants
Hospital.
for his help.
REFERENCES 1. Jaffe
HL: Osteoid osteoma. A benign osteoblastic tumor composed of osteoid and atypical bone. Arch Surg 31:709-72X, 1935 2. Jackson IJ: Osteoid osteoma of lamina and its treatment. Am Surg 19:17-23, 1953 3. Mauer I: Osteoid osteoma of the 12th rib. Resection under local anesthesia. A case report. Milit Med 122:194, 1958 4. Kehl DR. Alonso JE, Lovell WW: Scoliosis secondary to an osteoid osteoma of the rib. J Bone Joint Surg (Am) 65:701-703. 1983 5. Lynch MC, Dorgan JC: Osteoid osteoma of a rib as a cause of scoliosis. A case report. Spine 11:480-482, 1986 6. Nelson MC, Brower AC, Ragsdale BD: Case report 448. Skeletal Radio1 16:601-603, 1987 7. Abendschein T, Ulrich C, Kampmann I: Das Osteoidosteom: Bei ungunstiger Lokalisation schwierige Diagnose. Fortschr Rontgenstr 151:229-230. 1989 8. Wimpee MW, Maale GE, Hudkins PG. et al: Case report.
Scoliosis secondary 7:58Y-593. 1987
to osteoblastoma
9. Fabris D, Trainiti osteoblastoma: Report 1983 IO. Freiberger X:232-235,1960
of the rib. J Pediatr
Orthop
G, Di Comun M, et al: Scoliosis due to rib of two cases. J Pediatr Orthop 3:370-375.
RH: Osteoid
osteoma
I I. Maclellan DI. Wilson FC: Osteoid Bone Joint Surg (Am) 49:111-121. 1967
of the spine. osteoma
Radiology
of the spine. J
12. Keim HA, Reina EG: Osteoid-osteoma as a cause scoliosis. J Bone Joint Surg (Am) 57:159-163. 1975
of
13. Nelson OA, Greer RB: Localization of osteoid-osteoma of the spine using computerized tomograph. J Bone Joint Surg (Am) 65:263-265. 1983 14. Mehta MH: Pain provoked scoliosis. Observations evolution of deformity. Clin Orthop 135:5X-65. 1978
on the