Lisa D. Hobson-Webb, MD1 Stephen N. Donahue, MD1 Richard D. Bey, MD2 1 Department of Neurology, Duke University Medical Center, Durham, North Carolina 2 NovantHealth Winston Neurology, Winston-Salem, North Carolina
FIGURE 1. Serial measurements of grip strength recorded by a digital dynamometer. Day 1 measures began approximately 10 days after a dose of IVIG at 1 g/kg, beginning in the eighth month of treatment. Left grip strength is seen to fluctuate with IVIG infusions, but shows gradually increasing improvement.
a low amplitude right ulnar compound muscle action potential (CMAP) with mild diffuse slowing and absent F-waves. The right radial CMAP was not tested, as it had previously been documented as absent. The right median CMAP was normal, but the amplitude was reduced by 41% compared with the left (9.3 mV right, 5.5 mV left). Please see Supplementary Table S1 (available online) for details. On EMG (Supplementary Table S2), spontaneous activity and reinnervation changes were found in the right first dorsal interosseus, flexor digitorum profundus-4, 5, and extensor digitorum with chronic reinnervation changes of the triceps brachii. Cervical paraspinal, left upper extremity, and right lower extremity EMG was normal. Nerve ultrasonography of the right median, ulnar, and radial nerves was unhelpful, as they were normal other than focal enlargement of the right radial nerve (Supplementary Fig. S1) between the spiral groove and elbow (14 mm2). Motor neuron disease and MMN were considered. A motor neuropathy panel (GM1, D1b, GD1a, and MAG antibodies) was negative. An empiric trial of intravenous immunoglobulin (IVIG), 2 g/kg, was administered 2 weeks after initial evaluation. Within a week, he noted the return of finger extension and abduction and regained functional use of the right hand. Examination demonstrated 5/5 right triceps, 4/5 wrist extension, 3/5 finger extension, 3/5 finger abduction, 3/5 thumb abduction and opposition, 4/5 grip, and 5/5 deep finger flexion. He has continued to improve (Figure 1) while receiving IVIG 1 g/kg every 3 weeks over the past 6 months. This report demonstrates a 29-year lapse in stepwise progression of MMN. Despite the absence of antibodies and clear conduction block, the lack of sensory involvement and isolated enlargement of the radial nerve at a site not prone to compression helped suggest a diagnosis of MMN. The dramatic improvement in radial innervated muscles after IVIG was surprising given the duration of deficits and presumed axonal loss. It seems that his initial radial neuropathy was not compressive, but represented the first symptoms of MMN, diagnosed 30 years after onset. Letters to the Editor
1. Dimachkie MM, Barohn RJ, Katz J. Multifocal motor neuropathy, multifocal acquired demyelinating sensory and motor neuropathy, and other chronic acquired demyelinating polyneuropathy variants. Neurol Clin 2013;31:533–555. 2. Ulane CM, Brannagan TH. Chronic inflammatory demyelinating polyradiculoneuropathy, multifocal motor neuropathy and related disorders. In: Donofrio P, editor. Textbook of peripheral neuropathy. New York: DemosMedical; 2012. p 197–198. 3. Beekman R, van den Berg LH, Franssen H, Visser LH, van Asseldonk JT, Wokke JH. Ultrasonography shows extensive nerve enlargements in multifocal motor neuropathy. Neurology 2005;65:305–307.
Published online 12 November 2015 in Wiley Online Library (wileyonlinelibrary.com). DOI 10.1002/mus.24968
--------------------------------------------------------PRIMARY MYOSITIS AND ABSCESS OF ILIACUS MUSCLE DUE TO Brucella Paravertebral infections such as psoas abscesses are frequent in brucellosis.1 Most of these infections are secondary to a contiguous focus in the vertebral column or the sacroiliac joint.1,2 On the other hand, infection of the iliacus muscle alone with initial sparing of the psoas muscle and the vertebral column is rare.2 Turan et al.2 described the first case of iliacus muscle abscess and olecranon bursitis due to brucellosis in a young man who was treated with antibiotics alone and had successful outcome. Here, we report a case of primary Brucella iliacus muscle abscess and myositis that was complicated by sacroiliitis and was treated successfully with antibiotics alone. The patient is a previously healthy 22-year-old woman, who had a 2-week history of fever, chills, and low back pain upon presentation. The pain radiated to the left leg, was severe, and increased with walking. During this period, she was prescribed ciprofloxacin for suspected urinary tract infection but without benefit. Her physical exam showed normal vital signs and tenderness over the left paraspinal area, left flank, and left iliac fossa. Her white blood cell count was 6.3 3 109/L; erythrocytic sedimentation rate (ESR) was 55 mm/1st hour (normal, < 20 mm/1st hour); and C-reactive protein (CRP) was 56 mg/L (normal, < 5 mg/dl). A pelvis MRI showed myositis and abscess in the left iliacus muscle without involvement of the psoas muscle or the sacroiliac joint (Fig. 1A). Upon further inquiry, the patient reported raw milk intake 2 months before the onset of symptoms. Subsequent testing showed a Brucella standard tube agglutination titer of 1:640, and Brucella IgM and IgG (enzyme-linked immunosorbent assay) 23.8 U/ ml (normal,
FIGURE 1. Serial measurements of grip strength recorded by a digital dynamometer. Day 1 measures began approximately 10 days after a dose of IVIG at 1 g/kg, beginning in the eighth month of treatment. Left grip strength is seen to fluctuate with IVIG infusions, but shows gradually increasing improvement.
a low amplitude right ulnar compound muscle action potential (CMAP) with mild diffuse slowing and absent F-waves. The right radial CMAP was not tested, as it had previously been documented as absent. The right median CMAP was normal, but the amplitude was reduced by 41% compared with the left (9.3 mV right, 5.5 mV left). Please see Supplementary Table S1 (available online) for details. On EMG (Supplementary Table S2), spontaneous activity and reinnervation changes were found in the right first dorsal interosseus, flexor digitorum profundus-4, 5, and extensor digitorum with chronic reinnervation changes of the triceps brachii. Cervical paraspinal, left upper extremity, and right lower extremity EMG was normal. Nerve ultrasonography of the right median, ulnar, and radial nerves was unhelpful, as they were normal other than focal enlargement of the right radial nerve (Supplementary Fig. S1) between the spiral groove and elbow (14 mm2). Motor neuron disease and MMN were considered. A motor neuropathy panel (GM1, D1b, GD1a, and MAG antibodies) was negative. An empiric trial of intravenous immunoglobulin (IVIG), 2 g/kg, was administered 2 weeks after initial evaluation. Within a week, he noted the return of finger extension and abduction and regained functional use of the right hand. Examination demonstrated 5/5 right triceps, 4/5 wrist extension, 3/5 finger extension, 3/5 finger abduction, 3/5 thumb abduction and opposition, 4/5 grip, and 5/5 deep finger flexion. He has continued to improve (Figure 1) while receiving IVIG 1 g/kg every 3 weeks over the past 6 months. This report demonstrates a 29-year lapse in stepwise progression of MMN. Despite the absence of antibodies and clear conduction block, the lack of sensory involvement and isolated enlargement of the radial nerve at a site not prone to compression helped suggest a diagnosis of MMN. The dramatic improvement in radial innervated muscles after IVIG was surprising given the duration of deficits and presumed axonal loss. It seems that his initial radial neuropathy was not compressive, but represented the first symptoms of MMN, diagnosed 30 years after onset. Letters to the Editor
1. Dimachkie MM, Barohn RJ, Katz J. Multifocal motor neuropathy, multifocal acquired demyelinating sensory and motor neuropathy, and other chronic acquired demyelinating polyneuropathy variants. Neurol Clin 2013;31:533–555. 2. Ulane CM, Brannagan TH. Chronic inflammatory demyelinating polyradiculoneuropathy, multifocal motor neuropathy and related disorders. In: Donofrio P, editor. Textbook of peripheral neuropathy. New York: DemosMedical; 2012. p 197–198. 3. Beekman R, van den Berg LH, Franssen H, Visser LH, van Asseldonk JT, Wokke JH. Ultrasonography shows extensive nerve enlargements in multifocal motor neuropathy. Neurology 2005;65:305–307.
Published online 12 November 2015 in Wiley Online Library (wileyonlinelibrary.com). DOI 10.1002/mus.24968
--------------------------------------------------------PRIMARY MYOSITIS AND ABSCESS OF ILIACUS MUSCLE DUE TO Brucella Paravertebral infections such as psoas abscesses are frequent in brucellosis.1 Most of these infections are secondary to a contiguous focus in the vertebral column or the sacroiliac joint.1,2 On the other hand, infection of the iliacus muscle alone with initial sparing of the psoas muscle and the vertebral column is rare.2 Turan et al.2 described the first case of iliacus muscle abscess and olecranon bursitis due to brucellosis in a young man who was treated with antibiotics alone and had successful outcome. Here, we report a case of primary Brucella iliacus muscle abscess and myositis that was complicated by sacroiliitis and was treated successfully with antibiotics alone. The patient is a previously healthy 22-year-old woman, who had a 2-week history of fever, chills, and low back pain upon presentation. The pain radiated to the left leg, was severe, and increased with walking. During this period, she was prescribed ciprofloxacin for suspected urinary tract infection but without benefit. Her physical exam showed normal vital signs and tenderness over the left paraspinal area, left flank, and left iliac fossa. Her white blood cell count was 6.3 3 109/L; erythrocytic sedimentation rate (ESR) was 55 mm/1st hour (normal, < 20 mm/1st hour); and C-reactive protein (CRP) was 56 mg/L (normal, < 5 mg/dl). A pelvis MRI showed myositis and abscess in the left iliacus muscle without involvement of the psoas muscle or the sacroiliac joint (Fig. 1A). Upon further inquiry, the patient reported raw milk intake 2 months before the onset of symptoms. Subsequent testing showed a Brucella standard tube agglutination titer of 1:640, and Brucella IgM and IgG (enzyme-linked immunosorbent assay) 23.8 U/ ml (normal,
