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Case reports / Journal of Clinical Neuroscience 14 (2007) 997–1000

stretch a degenerated ligamentum flavum to cause intraligamentous hemorrhage. Minor trauma or laceration of the
ligamentum flavum is often caused by lumbar punctures
for epidural or intrathecal injections; however, intraligamentous hematoma has not been reported. The patients
in our study had not undergone such procedures, so direct
trauma theory cannot fully explain the mechanism of
hematoma formation in the ligamentum flavum. Nonetheless, back injury and degenerate ligamentum flavum with
poor vascularization may be important factors that increase the risk of hematoma of the ligamentum flavum.2
MRI is the most effective and useful investigational technique for detecting epidural mass lesions and provides
information about their anatomic position in relation to
the ligamentum flavum. T1-weighted images can show a
well-circumscribed lesion that is slightly or moderately
hyperintense relative to the ligamentum flavum in the
extradural space. T2-weighted images can reveal heteregenous intensity and areas of increased intensity within the
hematoma.1–5,10,11,13 In our patients, T1-weighted images
showed moderately high iso-intensity, and a hyperintense
area was present in the mass in the T2-weighted images.
Neurologic symptoms and findings of ligamentum flavum
hematoma are similar to those of other spinal canal disorders such as herniated nucleus pulposis, neoplasm, infection,
trauma, and spontaneous epidural hematoma.1,2,11 For this
reason, differential clinical diagnosis is difficult.
Epidural hematoma can be acute or chronic, and both
neurological findings and prognosis are usually worse than
with ligamentum flavum hematoma.1–5,10,11,13
Flavum hematomas are typically treated surgically
with extented interlaminar fenestration, excision of the
ligamentum flavum, and removal of the hematoma. For
each of the reported patients, including those in this study,

997

surgery produced excellent results. No recurrence has been
reported during the follow-up period.

References
1. Cruz-Conde R, Berjano P, Buitron Z. Ligamentum flavum hematoma
presenting as progressive root compression in the lumbar spine. Spine
1995;20:1506–9.
2. Hirakawa K, Hanakita J, Suwa H, et al. A post-traumatic
ligamentum flavum progressive hematoma: a case report. Spine
2000;25:1182–4.
3. Minamide A, Yoshida M, Tamaki T, et al. Ligamentum flavum
hematoma in the lumbar spine. Orthop Sci 1999;4:376–9.
4. Sweasey TA, Coester HC, Rawal H, et al. Ligamentum flavum
hematoma. Report of two cases. J Neurosurg 1992;76:534–7.
5. Yuceer N, Baskaya MK, Smith P, et al. Hematoma of the ligamentum flavum in the lumbar spine: case report. Surg Neurol 2000;53:
598–600.
6. Mizuno J, Nakagawa H, Inoue T, et al. Ligamentum flavum
hematoma in the lumbar spine. Case report. Neurol Med Chir
(Tokyo) 2005;45:212–5.
7. Baker JK, Hanson GW. Cyst of the ligamentum flavum. Spine
1994;19:1092–4.
8. Vernet O, Fankhauser H, Schynder P, et al. Cysts of the ligamentum
flavum: report of six cases. Neurosurgery 1991;29:277–83.
9. Yamaguchi S, Hida K, Akino M, et al. Ligamentum flavum
hematoma in the lumbar spine. Neurol Med Chir (Tokyo) 2005;45:
272–6.
10. Maezawa Y, Baba H, Uchida K, et al. Ligamentum flavum hematoma in the thoracic spine. Clin Imaging 2001;25:265–7.
11. Holtas S, Heiling M, Lonntoft M. Spontaneous spinal epidural
hematoma: findings at MR imaging and clinical correlation. Radiology 1996;199:409–13.
12. Jamjoom ZA. Acute spontaneous spinal epidural hematoma: the
influence of magnetic resonance imaging on diagnosis and treatment.
Surg Neurol 1996;6:345–9.
13. Major O, Sipos L, Czirjak S, et al. Spontaneous spinal epidural
haematomas. Acta Neurochir (Wien) 1991;111:40–2.

doi:10.1016/j.jocn.2006.05.015

Lupus erythematosus profundus (lupus panniculitis) induced
by interferon-b in a multiple sclerosis patientq
Takahisa Gono a, Masayuki Matsuda a,*, Yasuhiro Shimojima a, Kazuma Kaneko a,
Hiroshi Murata b, Shu-ichi Ikeda a
a

Department of Internal Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto 390-8621, Japan
b
Department of Dermatology, Shinshu University School of Medicine, Matsumoto, Japan
Received 16 March 2006; accepted 26 July 2006

q

Sources of support in the form of grants: the Neuroimmunological
Disease Research Committee of the Intractable Disease Division, the
Ministry of Public Health, Labor and Welfare, Japan.
*
Corresponding author. Tel.: +81 263 37 2673; fax: +81 263 37 3427.
E-mail address: matsuda@hsp.md.shinshu-u.ac.jp (M. Matsuda).

998

Case reports / Journal of Clinical Neuroscience 14 (2007) 997–1000

Abstract
We report a patient with multiple sclerosis (MS) who developed subcutaneous nodules on the face, shoulders and extremities while
being treated with interferon (IFN)-b-1b. These nodules fluctuated in parallel with myelopathy, and were diagnosed as lupus erythematosus profundus (LEP) based on histopathological findings. The patient showed no relapse of either neurological symptoms or subcutaneous nodules after cessation of IFN-b-1b. This agent can cause induration and necrosis in the sites of injection but also systemic skin
lesions such as LEP ascribable to its immunomodulatory effects.
2006 Elsevier Ltd. All rights reserved.
Keywords: Interferon-b; Lupus erythematosus profundus; Multiple sclerosis

1. Introduction
Interferon (IFN)-a and b are multifunctional cytokines
produced by leukocytes and fibroblasts, respectively. Both
are used clinically in the treatment of viral hepatitis, immune-mediated disorders and hematological malignancies,
especially multiple myeloma. IFN-a has been reported to
sometimes cause or exacerbate autoimmune inflammatory
disorders such as systemic lupus erythematosus (SLE)
and rheumatoid arthritis.1 Here, we report a patient with
multiple sclerosis (MS) who developed lupus erythematosus profundus (LEP) in conjunction with frequent attacks
of myelopathy while being treated with IFN-b-1b. This
skin manifestation has been described also as lupus panniculitis because the subcutaneous adipose tissue is mainly affected. No relapse of either myelopathy or LEP was seen
after cessation of IFN-b-1b, and we postulate that this
agent might have contributed to the development of LEP
with neurological exacerbations in our patient.
2. Case report
A 19-year-old woman experienced frequent attacks of
retrobulbar optic neuritis and transverse myelopathy with
no significant previous or family history. She was diagnosed
as having a relapse-remitting type of opticospinal MS based
on elevated levels of total protein and IgG in the cerebrospinal fluid (CSF) and abnormal signals in the thoracic cord
suggestive of demyelination on MRI. After commencement
of IFN-b-1b at a dose of 8 MIU subcutaneously every other
day at age 29 the patient showed a decrease in the frequency
of relapse. At age 33 she suddenly developed painful nodules on the face, both shoulders and upper limbs with no
precipitating cause. This symptom persisted, and the patient
was admitted to our hospital two months later because of
acute-onset paraparesis with urinary incontinence. Physical
examination showed multiple subcutaneous nodules with
slight redness and tenderness in the above-mentioned regions (Fig. 1A,B) in addition to bilateral hypoesthesia and
hypoalgesia below the Th3 level and muscle weakness in
both legs. Deep tendon reflexes were highly exaggerated in
both lower extremities with a positive Babinski’s sign. The
upper extremities showed no muscle weakness or sensory
disturbance. Laboratory tests demonstrated no abnormal

Fig. 1. Subcutaneous nodules with slight redness (in circles) were seen on
the left shoulder (A) and the left forearm (B). Biopsy from the nodule on the
left shoulder demonstrated marked infiltration of mononuclear cells in the
subcutaneous fat tissue suggestive of lobular panniculitis (C, bar = 100 lm).

findings in serum other than a slight increase in C-reactive
protein (1.32 mg/dL, normal <0.1 mg/dL) and positive results for the anti-nuclear (·160) and anti-SS-A antibodies.
The anti-double stranded DNA antibody was negative,
and serum levels of complements were within normal limits.
The neutralizing antibody for IFN-b-1b in serum was undetectable. CSF showed normal levels of total protein and
IgG with no oligoclonal IgG bands. MRI demonstrated
abnormal intensities but no obvious enhancement with gadolinium in the upper thoracic cord. Skin biopsy from the
subcutaneous nodule in the left shoulder showed severe
infiltration of lymphocytes in the dermis and fat tissue suggestive of lobular panniculitis (Fig. 1C).
Myelopathy and skin nodules quickly improved after
methylprednisolone pulse therapy at a dose of 1000 mg/day
for 3 days, but 1 month later these symptoms

Case reports / Journal of Clinical Neuroscience 14 (2007) 997–1000
2005
January

March

February

Methylprednisolone pulse therapy

999

April

20 mg/day

Oral prednisolone

Treatment

30 mg/day

Interferon β -1b

LEP
EDSS

6
4
2

Fig. 2. Clinical course of the patient. Neurological symptoms due to myelopathy fluctuated in parallel with lupus erythematosus profundus (LEP). Soon
after cessation of interferon-b-1b both neurological symptoms and LEP improved. Change in LEP is subjectively demonstrated on the basis of palpation
and inspection. EDSS: expanded disability status scale.14

simultaneously reworsened. Despite readministration of
methylprednisolone followed by oral prednisolone, both neurological and skin symptoms fluctuated (Fig. 2). Soon after
cessation of IFN-b-1b the subcutaneous nodules disappeared,
and the patient has remained in good neurological condition
without relapse of myelopathy for 1 year to date. The anti-nuclear and anti-SS-A antibodies are still detectable in serum.
3. Discussion
IFN-b-1b is widely used for MS, particularly in the relapse-remitting type as in our patient, in order to prevent
worsening of neurological symptoms. This agent often produces induration with or without skin ulcers in the site of
injection as an adverse effect.2 Our patient showed multiple
subcutaneous nodules with a histopathology of lobular panniculitis irrespective of the injection site of IFN-b-1b and no
involvement of visceral organs such as kidneys, leading to a
clinical diagnosis of LEP, which is an autoimmune disease
mainly affecting the skin. LEP is similar to SLE with regard
to the gross appearance of skin manifestations and involvement of IFN in the pathogenesis.3 Because skin symptoms
quickly improved after cessation of IFN-b-1b, this agent is
considered to have caused LEP in our patient. There are several recent reports showing association of autoimmune diseases, including SLE, after long-term use of IFN-b for
MS.4–6 Considering that the anti-nuclear and anti-SS-A
antibodies were positive at the onset of skin symptoms,
IFN-b-1b may have altered the immune status and caused
LEP in our patient, although this agent has not been shown
to increase the frequency of autoantibodies in MS.7–9
The most interesting point in our patient is that LEP fluctuated in parallel with upper thoracic myelopathy. This
neurological manifestation was clinically indistinguishable
from relapse of MS as previously seen, but our patient
showed neither abnormal findings in CSF nor new obvious
lesions on MRI. Although the precise etiology of myelopathy in our patient is unclear, there are two possibilities
worth consideration. One is mild exacerbations of MS,

which were undetectable in both the CSF study and MRI.
Our patient initially showed a decrease in the frequency
and severity of relapse in response to IFN-b-1b, but the
therapeutic efficacy may have diminished after the longterm use of this agent. The other is involvement of autoantibodies causing neurological symptoms. It is well known
that neurological manifestations as seen in MS occasionally
develop also in other autoimmune disorders, especially in
SLE and Sjo¨gren’s syndrome, and autoantibodies such as
the anti-SS-A antibody are considered to play an important
role in the pathogenesis.10–12 Considering that our patient
showed myelopathy as seen before the use of IFN-b-1b in
conjunction with development of LEP, the pathognomonic
autoantibodies may have easily migrated into the spinal
cord across the blood-brain barrier impaired by MS, resulting in frequent exacerbations of neurological symptoms.13
In conclusion, we described LEP due to IFN-b-1b in a
patient being treated for MS. This agent can cause induration and necrosis in the sites of injection but also systemic
skin lesions such as LEP ascribable to its immunomodulatory effects. When systemic skin lesions develop during
treatment with IFN-b-1b, autoimmune diseases due to
alteration of the immune status are possible causes, and
in such a case cessation of this agent should be considered
as a potent therapeutic option.
References
1. Vial T, Descotes J. Immune-mediated side-effects of cytokines in
humans. Toxicology 1995;105:31–57.
2. Walther EU, Hohlfeld R. Multiple sclerosis. Side effects of
interferon beta therapy and their management. Neurology 1999;53:
1622–7.
3. Blanco P, Palucka AK, Gill M, et al. Induction of dendritic cell
differentiation by IFN-alpha in systemic lupus erythematosus. Science
2001;294:1540–3.
4. Nousari HC, Kimyai-Asadi A, Tausk FA. Subacute cutaneous lupus
erythematosus associated with interferon beta-1a. Lancet
1998;352:1825–6.
5. Schmutz J, Barbaud A, Trechot P. Subacute cutaneous lupus erythematosus and interferon beta-1a. Ann Dermatol Venereol 2000;127:237.

1000

Case reports / Journal of Clinical Neuroscience 14 (2007) 1000–1003

6. Crispin JC, Diaz-Jouanen E. Systemic lupus erythematosus induced
by therapy with interferon-beta in a patient with multiple sclerosis.
Lupus 2005;14:495–6.
7. Polman CH, Kappos L, Dahlke F, et al. Interferon beta-1b
treatment does not induce autoantibodies. Neurology 2005;64:
996–1000.
8. Kivisakk P, Lundahl J, von Heigl Z, et al. No evidence for increased
frequency of autoantibodies during interferon-beta 1b treatment of
multiple sclerosis. Acta Neurol Scand 1998;97:320–3.
9. Colosimo C, Pozzilli C, Frontoni M, et al. No increase of serum
autoantibodies during therapy with recombinant human interferonbeta 1a in relapsing-remitting multiple sclerosis. Acta Neurol Scand
1997;96:372–4.
10. Hirohata S, Kosaka M. Association of anti-Sm antibodies with
organic brain syndrome secondary to systemic lupus erythematosus.
Lancet 1994;343:796.

11. Swaak AJ, Huysen V, Nossent JC, et al. Antinuclear antibody
profiles in relation to specific disease manifestations of systemic lupus
erythematosus. Clin Rheumatol 1990;9:82–94.
12. Silva LM, Garcia AB, Donadi EA. Increased lymphocyte death by
neglect-apoptosis is associated with lymphopenia and autoantibodies
in lupus patients presenting with neuropsychiatric manifestations. J
Neurol 2002;249:1048–54.
13. McLean BN, Miller D, Thompson EJ. Oligoclonal banding of IgG in
CSF, blood-brain barrier function, and MRI findings in patients with
sarcoidosis, systemic lupus erythematosus, and Behcet’s disease
involving the nervous system. J Neurol Neurosurg Psychiatry
1995;58:548–54.
14. Kurtzke JF. Rating neurological impairment in multiple sclerosis: an
expanded disability status scale (EDSS). Neurology (Cleveland)
1983;33:1444–52.

doi:10.1016/j.jocn.2006.07.011

Intracranial mucocele in pregnancy
Murat Cosar a, Mustafa Aziz Hatiboglu

b,*

, Emine Cosar c, A. Celal Iplıkcıoglu

b

a

c

Department of Neurosurgery, Afyon Kocatepe University Medicine Faculty, Afyon, Turkey
b
Department of Neurosurgery, Okmeydani Training Hospital, Istanbul, Turkey
Department of Obstetrics and Gynecology, Afyon Kocatepe University Medicine Faculty, Afyon, Turkey
Received 25 January 2006; accepted 2 August 2006

Abstract
Intracranial mucocele is rare. They are slow-growing lesions and usually form as a result of an obstruction of the paranasal sinuses,
particularly by osteoma, fibrosis, trauma, previous surgery or inflammation. The effect of pregnancy in the development of intracranial
mucocele is unclear. We report a pregnant patient who was admitted to our clinic with severe headache and diplopia. A giant mucocele in
the right frontal lobe was detected on magnetic resonance imaging. The patient underwent surgery with satisfactory outcome.
2006 Elsevier Ltd. All rights reserved.
Keywords: Frontal lobe; Intracranial extension; Mucocele; Surgery

1. Introduction
Mucoceles are slow-growing and locally aggressive lesions which form due to the accumulation of mucous secretions in a paranasal sinus secondary to obstruction caused
by inflammation, fibrosis, trauma, previous surgery, anatomical abnormality or by a mass lesion such as an osteoma. Most mucoceles arise in the frontal and ethmoidal
sinuses, but they rarely extend into the intracranial cavity.
We report a pregnant patient with a giant mucocele in the

*
Corresponding author. Present address: Turkali Mah. Sehit Asim Cad.
Sakarya Apt. 104/2, 80690, Besiktas, Istanbul, Turkey. Tel.: +90 532 761
22 90; fax: +90 212 2217800.
E-mail address: azizhatiboglu@yahoo.com (M.A. Hatiboglu).

frontal region. The possible role of pregnancy in the development of intracranial mucocele is also discussed.
2. Case report
A 35-year-old woman in the 7th month of pregnancy
was admitted to our clinic with a history of severe headache and diplopia of 1-month duration. Also, the patient
had complained of nasal obstruction and rhinitis over
the previous few months. Neurological examination revealed a sixth nerve palsy of the right eye and bilateral
papilledema. The patient had no history of previous surgery or trauma.
MRI showed a mass lesion measuring 4 · 4 · 7 cm
which was hypointense on T1-weighted and hyperintense


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