Diaphragmatic paralysis and respiratory failure as a complication of Lyme disease
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《神经病学神经外科学杂志》
St Richard’s Hospital, Chichester, UK
Correspondence to:
Rachel A Abbott
St Richard’s Hospital, Spitalfield Lane, Chichester, West Sussex PO19 6SE, UK; rachel.abbott@nhs.net
Keywords: Borrelia burgdorferi; diaphragmatic paralysis; Lyme disease; neuroborreliosis
There have been five recorded cases of diaphragmatic paralysis as a complication of neuroborreliosis.1–5 Here we report another case of Lyme meningoradiculitis, caused by an identified tick, leading to bilateral diaphragmatic paralysis with an abbreviated course on treatment.
Case report
A 59 year old female presented with a recent history of abdominal pain and falls because of a weakness in her right leg. She had been complaining of flu-like symptoms with twitches in her back and pain in her right side for a month. She gave a history of recently having been bitten by ticks whilst gardening. There was no history of any recent rash. On the day of presentation, she complained of a mild cough, reduced appetite, abdominal distension, constipation, and dysuria. She was a lifelong smoker but was generally healthy.
At presentation her blood pressure was 206/107 mm Hg. There was some epigastric tenderness. She had bruising on her right leg that she associated with the falls.
The chest radiograph on admission was unremarkable. Abdominal x ray showed dilated loops of small bowel and a loaded colon. Her only blood abnormality was hyponatraemia at 121 mmol/l. She was admitted for further investigations.
On day 3 of admission she became increasingly short of breath and on examination had decreased bibasal air entry. On day 4 her respiratory rate was 25/min and arterial blood gases (ABG) demonstrated hypoxaemia but adequate ventilation with pH 7.51, Po2 6.7 kPa, and Pco2 4.7 kPa. Her chest radiograph showed left basal changes. On day 5 her Pco2 had risen to 6.8; she was admitted to the intensive care unit and non-invasive ventilatory support was commenced. She had a decreased inspiratory pressure and a decreased vital capacity. She was noted to have absent gag reflex and poor swallow and on day 6 was intubated to protect against aspiration pneumonia. The patient remained fully conscious and co-operative, easily triggering the ventilator but requiring significant inspiratory pressure support of 20 cm H2O.
Neurological examination demonstrated right hip and knee extensor weakness (2/5), absent right knee jerk, and a loss of sensation on her left lateral thigh. Because she lived in a known endemic area we thought about Lyme disease, but we also considered differential diagnoses such as Guillain-Barre syndrome, listeriosis, and acute poliomyelitis. We commenced treatment with doxycycline whilst awaiting the results of further investigations. Around this time the patient indicated a small black lesion on her upper abdomen that was removed and on closer examination was identified as tick mouthparts (fig 1).
Figure 1 Photomicrograph of tick recovered from patient, showing tick mouthparts.
Given her smoking history and persistent hyponatraemia, a chest computerised tomography was performed which showed only left lower lobe collapse and a small left pleural effusion. Bronchoscopy was unremarkable. Chest ultrasound screening showed only minimal movement of both diaphragms.
Cerebrospinal fluid analysis (CSF) demonstrated a white cell count 181 cells/mm3 (100% mononuclear), red cell count 22 cells/mm3, glucose 2.3 mmol/l (serum glucose 6.8 mmol/l), and protein 0.96 g/l. Immunological analysis of the CSF was not done because an insufficient sample was obtained. Brain magnetic resonance imaging (MRI) was normal. Spinal MRI and electromyography were not carried out. Immunoglobulin M and immunoglobulin G antibodies to Borrelia burgdorferi were detected in serum, and at this point intravenous ceftriaxone (2 g for 30 days) was commenced.
Over the next 7 days her strength increased and a repeat ultrasound of the diaphragm on day 16 of admission showed marked improvement with the right dome moving normally and some residual left sided weakness associated with overlying lung consolidation.
The patient was weaned from the ventilator and extubated following a total of 22 days of respiratory support. She underwent intensive physiotherapy and has made an uneventful recovery. One year later she complained of mild shortness of breath on lying flat with an exercise tolerance of 1 mile on the flat. On examination she had hyperaesthesia in her right leg with slightly brisk knee reflexes (previously absent right knee jerk). She had persistent bilateral diaphragmatic paralysis demonstrated on chest ultrasound screening. Pulmonary function tests showed a 40% reduction between erect and supine vital capacity measurements.
Discussion
The first case of diaphragmatic paralysis as a complication of Lyme disease was reported in 1986: a 73 year old male, treated with ampicillin and netilmicin, who required ventilation for 3 months and then died after receiving treatment for a pulmonary embolism.1 Another four cases have been reported in patients between the ages of 39 and 68, all of whom were treated with either doxycycline or ceftriaxone and two of whom required ventilation due to respiratory failure.1–5 All patients were well at follow up, although one patient had persistent phrenic paralysis 6 months after treatment.5
In all previous cases of diaphragmatic palsy as a complication of Lyme disease, either the patient reported dyspnoea or hypoxia was noted on ABG. The diagnosis of phrenic nerve palsy was made by the following methods: hemidiaphragm elevation, fluoroscopic screening of diaphragmatic movements, or electrical stimulation of phrenic nerves.1–5 Our patient had a lymphocytic meningitis with sensory and motor neuropathies including bilateral phrenic nerve palsies. Diaphragmatic paralysis due to Lyme disease was diagnosed on the basis of clinical features, chest ultrasonography, the presence of the tick head, and serology indicating a recent infection with B burgdorferi as well as a rapid response to antibiotic therapy.
The clinical diagnosis of Lyme disease may be supported by serologic testing. B burgdorferi antibody tests may be negative in early infection, but patients are usually seropositive at or shortly after presenting with neurological symptoms. In some patients, antibodies against B burgdorferi may be detectable in CSF slightly earlier than serum. Culture and B burgdorferi deoxyribonucleic acid detection using polymerase chain reaction may also be used but were not in our case.
The three patients reported in the literature with respiratory failure caused by neuroborreliosis were ventilated for 3 months, 1 month, and 13 months, respectively, whilst our patient required ventilation for only 22 days.1,2,4 We speculate that early recognition of the possibility of Lyme disease and appropriate treatment shortened our patient’s acute illness.
In conclusion, it is important to consider Lyme disease in the differential diagnosis of acute respiratory failure – with or without erythema migrans.
ACKNOWLEDGEMENTS
We would like to thank Mr P R Randell of the Microbiology Department, St Richard’s Hospital, Chichester, UK and Dr Susan O’Connell of the Department of Microbiology and Public Health Laboratory, Southampton General Hospital, Southampton, UK.
References
Melet M, Gerard A, Voiriot P, et al. Méningoradiculonévrite mortelle au cours d’une maladie de Lyme. Presse Med 1986;5:2075.
Sigler S, Kershaw P, Scheuch R, et al. Rspiratory failure due to Lyme meningoradiculitis. Am J Med 1997;103:544–547.
Faul JL, Ruoss S, Doyle RL, et al. Diaphragmatic paralysis due to Lyme disease. Eur Respir J 1999;13:700–702.
Winterholler M, Erbguth FJ. Tick bite induced respiratory failure. Intensive Care Med 2001;27:1095.
Gomez de la Torre R, Suarez del Villar R, Alvarez Carreno F, et al. Parálisis diafragmática y artromialgia por enfermedad de Lyme. An Med Interna (Madrid) 2003;20:47–9.(R A Abbott, S Hammans, M )
Correspondence to:
Rachel A Abbott
St Richard’s Hospital, Spitalfield Lane, Chichester, West Sussex PO19 6SE, UK; rachel.abbott@nhs.net
Keywords: Borrelia burgdorferi; diaphragmatic paralysis; Lyme disease; neuroborreliosis
There have been five recorded cases of diaphragmatic paralysis as a complication of neuroborreliosis.1–5 Here we report another case of Lyme meningoradiculitis, caused by an identified tick, leading to bilateral diaphragmatic paralysis with an abbreviated course on treatment.
Case report
A 59 year old female presented with a recent history of abdominal pain and falls because of a weakness in her right leg. She had been complaining of flu-like symptoms with twitches in her back and pain in her right side for a month. She gave a history of recently having been bitten by ticks whilst gardening. There was no history of any recent rash. On the day of presentation, she complained of a mild cough, reduced appetite, abdominal distension, constipation, and dysuria. She was a lifelong smoker but was generally healthy.
At presentation her blood pressure was 206/107 mm Hg. There was some epigastric tenderness. She had bruising on her right leg that she associated with the falls.
The chest radiograph on admission was unremarkable. Abdominal x ray showed dilated loops of small bowel and a loaded colon. Her only blood abnormality was hyponatraemia at 121 mmol/l. She was admitted for further investigations.
On day 3 of admission she became increasingly short of breath and on examination had decreased bibasal air entry. On day 4 her respiratory rate was 25/min and arterial blood gases (ABG) demonstrated hypoxaemia but adequate ventilation with pH 7.51, Po2 6.7 kPa, and Pco2 4.7 kPa. Her chest radiograph showed left basal changes. On day 5 her Pco2 had risen to 6.8; she was admitted to the intensive care unit and non-invasive ventilatory support was commenced. She had a decreased inspiratory pressure and a decreased vital capacity. She was noted to have absent gag reflex and poor swallow and on day 6 was intubated to protect against aspiration pneumonia. The patient remained fully conscious and co-operative, easily triggering the ventilator but requiring significant inspiratory pressure support of 20 cm H2O.
Neurological examination demonstrated right hip and knee extensor weakness (2/5), absent right knee jerk, and a loss of sensation on her left lateral thigh. Because she lived in a known endemic area we thought about Lyme disease, but we also considered differential diagnoses such as Guillain-Barre syndrome, listeriosis, and acute poliomyelitis. We commenced treatment with doxycycline whilst awaiting the results of further investigations. Around this time the patient indicated a small black lesion on her upper abdomen that was removed and on closer examination was identified as tick mouthparts (fig 1).
Figure 1 Photomicrograph of tick recovered from patient, showing tick mouthparts.
Given her smoking history and persistent hyponatraemia, a chest computerised tomography was performed which showed only left lower lobe collapse and a small left pleural effusion. Bronchoscopy was unremarkable. Chest ultrasound screening showed only minimal movement of both diaphragms.
Cerebrospinal fluid analysis (CSF) demonstrated a white cell count 181 cells/mm3 (100% mononuclear), red cell count 22 cells/mm3, glucose 2.3 mmol/l (serum glucose 6.8 mmol/l), and protein 0.96 g/l. Immunological analysis of the CSF was not done because an insufficient sample was obtained. Brain magnetic resonance imaging (MRI) was normal. Spinal MRI and electromyography were not carried out. Immunoglobulin M and immunoglobulin G antibodies to Borrelia burgdorferi were detected in serum, and at this point intravenous ceftriaxone (2 g for 30 days) was commenced.
Over the next 7 days her strength increased and a repeat ultrasound of the diaphragm on day 16 of admission showed marked improvement with the right dome moving normally and some residual left sided weakness associated with overlying lung consolidation.
The patient was weaned from the ventilator and extubated following a total of 22 days of respiratory support. She underwent intensive physiotherapy and has made an uneventful recovery. One year later she complained of mild shortness of breath on lying flat with an exercise tolerance of 1 mile on the flat. On examination she had hyperaesthesia in her right leg with slightly brisk knee reflexes (previously absent right knee jerk). She had persistent bilateral diaphragmatic paralysis demonstrated on chest ultrasound screening. Pulmonary function tests showed a 40% reduction between erect and supine vital capacity measurements.
Discussion
The first case of diaphragmatic paralysis as a complication of Lyme disease was reported in 1986: a 73 year old male, treated with ampicillin and netilmicin, who required ventilation for 3 months and then died after receiving treatment for a pulmonary embolism.1 Another four cases have been reported in patients between the ages of 39 and 68, all of whom were treated with either doxycycline or ceftriaxone and two of whom required ventilation due to respiratory failure.1–5 All patients were well at follow up, although one patient had persistent phrenic paralysis 6 months after treatment.5
In all previous cases of diaphragmatic palsy as a complication of Lyme disease, either the patient reported dyspnoea or hypoxia was noted on ABG. The diagnosis of phrenic nerve palsy was made by the following methods: hemidiaphragm elevation, fluoroscopic screening of diaphragmatic movements, or electrical stimulation of phrenic nerves.1–5 Our patient had a lymphocytic meningitis with sensory and motor neuropathies including bilateral phrenic nerve palsies. Diaphragmatic paralysis due to Lyme disease was diagnosed on the basis of clinical features, chest ultrasonography, the presence of the tick head, and serology indicating a recent infection with B burgdorferi as well as a rapid response to antibiotic therapy.
The clinical diagnosis of Lyme disease may be supported by serologic testing. B burgdorferi antibody tests may be negative in early infection, but patients are usually seropositive at or shortly after presenting with neurological symptoms. In some patients, antibodies against B burgdorferi may be detectable in CSF slightly earlier than serum. Culture and B burgdorferi deoxyribonucleic acid detection using polymerase chain reaction may also be used but were not in our case.
The three patients reported in the literature with respiratory failure caused by neuroborreliosis were ventilated for 3 months, 1 month, and 13 months, respectively, whilst our patient required ventilation for only 22 days.1,2,4 We speculate that early recognition of the possibility of Lyme disease and appropriate treatment shortened our patient’s acute illness.
In conclusion, it is important to consider Lyme disease in the differential diagnosis of acute respiratory failure – with or without erythema migrans.
ACKNOWLEDGEMENTS
We would like to thank Mr P R Randell of the Microbiology Department, St Richard’s Hospital, Chichester, UK and Dr Susan O’Connell of the Department of Microbiology and Public Health Laboratory, Southampton General Hospital, Southampton, UK.
References
Melet M, Gerard A, Voiriot P, et al. Méningoradiculonévrite mortelle au cours d’une maladie de Lyme. Presse Med 1986;5:2075.
Sigler S, Kershaw P, Scheuch R, et al. Rspiratory failure due to Lyme meningoradiculitis. Am J Med 1997;103:544–547.
Faul JL, Ruoss S, Doyle RL, et al. Diaphragmatic paralysis due to Lyme disease. Eur Respir J 1999;13:700–702.
Winterholler M, Erbguth FJ. Tick bite induced respiratory failure. Intensive Care Med 2001;27:1095.
Gomez de la Torre R, Suarez del Villar R, Alvarez Carreno F, et al. Parálisis diafragmática y artromialgia por enfermedad de Lyme. An Med Interna (Madrid) 2003;20:47–9.(R A Abbott, S Hammans, M )