CASE REPORTS
Carpal Tunnel Syndrome in Sarcoidosis: A Case Report of a Rare Neurologic Manifestation Ajinkya Sonambekar, MD, MBBS; Nikhil Gupta, MD, MBBS; Akanksha Swadi, MD, MBBS; Laxmikant Ramkumarsingh Tomar, MD, MBBS
Perm J 2016 Fall;20(4):15-168
E-pub: 09/13/2016
http://dx.doi.org/10.7812/TPP/15-168
ABSTRACT Introduction: Sarcoidosis is a multisystemic inflammatory disease with myriad clinical manifestations. Neurologic involvement in sarcoidosis is uncommon. Peripheral neuropathic presentations include mononeuropathy, mononeuritis multiplex, and generalized sensory, motor, autonomic, and sensorimotor polyneuropathies. Case Presentation: We report a case of carpal tunnel syndrome caused by sarcoidosis in a 30-year-old woman. Other causes of carpal tunnel syndrome were ruled out. The patient responded well to the standard line of corticosteroid treatment and wrist splinting. Discussion: Carpal tunnel syndrome caused by sarcoidosis is a rare presentation. The mechanism of neurologic involvement in sarcoidosis is not clear.
of 200 U/L. Fasting blood glucose level was 86 mg/dL, and the thyroid function test had a normal result. Nerve conduction studies showed carpal tunnel syndrome of the right hand with involvement of the sensory component without motor deficit. A chest computed tomography scan was obtained and showed right paratracheal and bilateral hilar lymphadenopathy, which was nonnecrotic without any calcification (Figure 1). A parenchymal lesion showed a reticulonodular pattern and suggested a diagnosis of sarcoidosis. Fine-needle aspiration cytology of the mediastinal lymph node showed noncaseating granulomas without any acidfast bacilli and confirmed the diagnosis of sarcoidosis. Table 1. Case timeline Day 1
INTRODUCTION
Neurologic involvement is reported in 5% to 10% of patients with sarcoidosis.1 Some authors consider neurosarcoidosis a totally different entity. Most often, a presentation of neurosarcoidosis is multiple fluctuating and remitting cranial nerve palsies. Noncranial neuropathy has been reported in 15% to 40% of cases of neurosarcoidosis.2 Scott et al3 reported a case series in which peripheral neuropathy developed in 6% to 18% of patients with neurosarcoidosis.
3
4
CASE PRESENTATION
A 30-year-old woman presented with complaints of tingling and numbness in her right hand. On detailed inquiry, the patient also had a history of malaise, anorexia, weight loss, and cough for 1 month. There was no associated muscular weakness of the right hand, fever, or any other systemic involvement. There was no history of tuberculosis. She was a nonsmoker and a nonalcoholic. Results of the physical examination revealed a positive Tinel sign. There was no skin lesion or lymphadenopathy. The systemic findings were normal. The patient underwent many investigations (Table 1), of which relevant ones included a Mantoux test response of 0 mm at 48 hours and increased angiotensin-converting enzyme levels
6 7 21 33
100 150
Symptoms and treatment Complaints of tingling and numbness in right hand, malaise, anorexia, weight loss, and cough for 1 month Physical examination revealed positive Tinel sign. Systemic findings were normal Mantoux test response was 0 mm at 48 hours and increased angiotensin-converting enzyme level of 200 U/L Blood glucose level and thyroid function test had normal results Nerve conduction studies showed carpal tunnel syndrome of right hand with involvement of sensory component without motor deficit Chest computed tomography scan showed right paratracheal and bilateral hilar lymphadenopathy, which was nonnecrotic without any calcification. Parenchymal lesion showed reticulonodular pattern and suggested sarcoidosis Other causes of carpal tunnel syndrome were ruled out Fine-needle aspiration cytology of mediastinal lymph node showed noncaseating granulomas without any acid-fast bacilli and confirmed the diagnosis of sarcoidosis Treatment started with corticosteroids Carpal tunnel splint was prescribed Symptoms of malaise, cough, anorexia, and weight loss subsided Neuropathic symptoms of tingling and numbness of right hand started subsiding Began tapering corticosteroid Neuropathic symptoms recovered completely Steroid dosage tapering completed and stopped
Ajinkya Sonambekar, MD, MBBS, is a Physician at the University College of Medical Sciences and at Guru Teg Bahadur Hospital in Dilshad Garden, Delhi, India. E-mail: [email protected]. Nikhil Gupta, MD, MBBS, is a Fellow in Clinical Immunology & Rheumatology at the Christian Medical College in Vellore, India. E-mail: [email protected]. Akanksha Swadi, MD, MBBS, is a Radiologist at Byramjee Jeejeebhoy Government Medical College in Pune, India. E-mail: [email protected]. Laxmikant Ramkumarsingh Tomar, MD, MBBS, is a Resident in the Neurology Department at the Govind Ballabh Pant Institute of Post Graduate Medical Education and Research in Delhi, India. E-mail: [email protected].
The Permanente Journal/Perm J 2016 Fall;20(4):15-168
1
CASE REPORTS Carpal Tunnel Syndrome in Sarcoidosis: A Case Report of a Rare Neurologic Manifestation
An association of carpal tunnel syndrome with sarcoidosis is rare, and our case adds to the existing sparse, English-language literature.2,4-6 The etiopathogenesis of carpal tunnel syndrome in sarcoidosis is not clearly defined. Thinking well beyond commonness may help in detecting the underlying cause of carpal tunnel syndrome, or else the diagnosis might be missed. Specific therapy in such cases may help in alleviating patient suffering and working toward the common goal of better patient care. v Disclosure Statement The author(s) have no conflicts of interest to disclose.
Figure 1. Chest computed tomography scan showing right paratracheal and bilateral hilar nonnecrotic, noncalcific lymphadenopathy.
The patient was treated with corticosteroids, after which her symptoms of malaise, cough, anorexia, and weight loss subsided. Other causes of carpal tunnel syndrome were ruled out. A carpal tunnel splint was prescribed for the patient, after which she recovered.
DISCUSSION
This patient, who presented with neurologic complaints, turned out to have a case of neurosarcoidosis with carpal tunnel syndrome. Other common causes of carpal tunnel syndrome were ruled out on investigations. The mechanism of neurologic involvement in sarcoidosis is not clear. Granulomatous inflammation of nerve layers, secondarily caused by vasculitic neuropathy, demyelination, panangiitis, compression by sarcoid tissue, or thick edema under perineural tissue are some of the proposed causes of nerve involvement in sarcoidosis.2 Treatment is mainly corticosteroid based and physical therapy. Our patient responded well to the standard line of treatment.
2
Acknowledgment Kathleen Louden, ELS, of Louden Health Communications provided editorial assistance. How to Cite this Article Sonambekar A, Gupta N, Swadi A, Tomar LR. Carpal tunnel syndrome in sarcoidosis: A case report of a rare neurologic manifestation. Perm J 2016 Fall;20(4):15-168. DOI: http://dx.doi.org/10.7812/TPP/15-158. References
1. Burns TM. Neurosarcoidosis. Arch Neurol 2003 Aug;60(8):1166-8. DOI: http://dx.doi. org/10.1001/archneur.60.8.1166. 2. Nakatani-Enomoto S, Aizawa H, Koyama S, Kuroda K, Kimura T, Kikuchi K. Transient swelling of peripheral nerves in a case of neurosarcoidosis. Intern Med 2004 Nov;43(11):1078-81. DOI: http://dx.doi.org/10.2169/internalmedicine.43.1078. 3. Scott TF. Neurosarcoidosis: progress and clinical aspects. Neurology 1993 Jan;43(1):8-12. 4. Niemer GW, Bolster MB, Buxbaum L, Judson MA. Carpal tunnel syndrome in sarcoidosis. Sarcoidosis Vasc Diffuse Lung Dis 2001 Oct;18(3):296-300. 5. Yanardağ H, Pamuk ON, Kiziltan M, Yildiz H, Demirey S, Karayel T. An increased frequency of carpal tunnel syndrome in sarcoidosis. Results of a study based on nerve conduction study. Acta Medica (Hradec Kralove) 2003;46(4):201-4. 6. Shambaugh GE, Cirksena WJ, Newcomer KL. Carpal tunnel syndrome as manifestation of sarcoidosis. Arch Intern Med 1964 Dec;114:830-3. DOI: http://dx.doi. org/10.1001/archinte.1964.03860120142018.
The Permanente Journal/Perm J 2016 Fall;20(4):15-168
Carpal Tunnel Syndrome in Sarcoidosis: A Case Report of a Rare Neurologic Manifestation Ajinkya Sonambekar, MD, MBBS; Nikhil Gupta, MD, MBBS; Akanksha Swadi, MD, MBBS; Laxmikant Ramkumarsingh Tomar, MD, MBBS
Perm J 2016 Fall;20(4):15-168
E-pub: 09/13/2016
http://dx.doi.org/10.7812/TPP/15-168
ABSTRACT Introduction: Sarcoidosis is a multisystemic inflammatory disease with myriad clinical manifestations. Neurologic involvement in sarcoidosis is uncommon. Peripheral neuropathic presentations include mononeuropathy, mononeuritis multiplex, and generalized sensory, motor, autonomic, and sensorimotor polyneuropathies. Case Presentation: We report a case of carpal tunnel syndrome caused by sarcoidosis in a 30-year-old woman. Other causes of carpal tunnel syndrome were ruled out. The patient responded well to the standard line of corticosteroid treatment and wrist splinting. Discussion: Carpal tunnel syndrome caused by sarcoidosis is a rare presentation. The mechanism of neurologic involvement in sarcoidosis is not clear.
of 200 U/L. Fasting blood glucose level was 86 mg/dL, and the thyroid function test had a normal result. Nerve conduction studies showed carpal tunnel syndrome of the right hand with involvement of the sensory component without motor deficit. A chest computed tomography scan was obtained and showed right paratracheal and bilateral hilar lymphadenopathy, which was nonnecrotic without any calcification (Figure 1). A parenchymal lesion showed a reticulonodular pattern and suggested a diagnosis of sarcoidosis. Fine-needle aspiration cytology of the mediastinal lymph node showed noncaseating granulomas without any acidfast bacilli and confirmed the diagnosis of sarcoidosis. Table 1. Case timeline Day 1
INTRODUCTION
Neurologic involvement is reported in 5% to 10% of patients with sarcoidosis.1 Some authors consider neurosarcoidosis a totally different entity. Most often, a presentation of neurosarcoidosis is multiple fluctuating and remitting cranial nerve palsies. Noncranial neuropathy has been reported in 15% to 40% of cases of neurosarcoidosis.2 Scott et al3 reported a case series in which peripheral neuropathy developed in 6% to 18% of patients with neurosarcoidosis.
3
4
CASE PRESENTATION
A 30-year-old woman presented with complaints of tingling and numbness in her right hand. On detailed inquiry, the patient also had a history of malaise, anorexia, weight loss, and cough for 1 month. There was no associated muscular weakness of the right hand, fever, or any other systemic involvement. There was no history of tuberculosis. She was a nonsmoker and a nonalcoholic. Results of the physical examination revealed a positive Tinel sign. There was no skin lesion or lymphadenopathy. The systemic findings were normal. The patient underwent many investigations (Table 1), of which relevant ones included a Mantoux test response of 0 mm at 48 hours and increased angiotensin-converting enzyme levels
6 7 21 33
100 150
Symptoms and treatment Complaints of tingling and numbness in right hand, malaise, anorexia, weight loss, and cough for 1 month Physical examination revealed positive Tinel sign. Systemic findings were normal Mantoux test response was 0 mm at 48 hours and increased angiotensin-converting enzyme level of 200 U/L Blood glucose level and thyroid function test had normal results Nerve conduction studies showed carpal tunnel syndrome of right hand with involvement of sensory component without motor deficit Chest computed tomography scan showed right paratracheal and bilateral hilar lymphadenopathy, which was nonnecrotic without any calcification. Parenchymal lesion showed reticulonodular pattern and suggested sarcoidosis Other causes of carpal tunnel syndrome were ruled out Fine-needle aspiration cytology of mediastinal lymph node showed noncaseating granulomas without any acid-fast bacilli and confirmed the diagnosis of sarcoidosis Treatment started with corticosteroids Carpal tunnel splint was prescribed Symptoms of malaise, cough, anorexia, and weight loss subsided Neuropathic symptoms of tingling and numbness of right hand started subsiding Began tapering corticosteroid Neuropathic symptoms recovered completely Steroid dosage tapering completed and stopped
Ajinkya Sonambekar, MD, MBBS, is a Physician at the University College of Medical Sciences and at Guru Teg Bahadur Hospital in Dilshad Garden, Delhi, India. E-mail: [email protected]. Nikhil Gupta, MD, MBBS, is a Fellow in Clinical Immunology & Rheumatology at the Christian Medical College in Vellore, India. E-mail: [email protected]. Akanksha Swadi, MD, MBBS, is a Radiologist at Byramjee Jeejeebhoy Government Medical College in Pune, India. E-mail: [email protected]. Laxmikant Ramkumarsingh Tomar, MD, MBBS, is a Resident in the Neurology Department at the Govind Ballabh Pant Institute of Post Graduate Medical Education and Research in Delhi, India. E-mail: [email protected].
The Permanente Journal/Perm J 2016 Fall;20(4):15-168
1
CASE REPORTS Carpal Tunnel Syndrome in Sarcoidosis: A Case Report of a Rare Neurologic Manifestation
An association of carpal tunnel syndrome with sarcoidosis is rare, and our case adds to the existing sparse, English-language literature.2,4-6 The etiopathogenesis of carpal tunnel syndrome in sarcoidosis is not clearly defined. Thinking well beyond commonness may help in detecting the underlying cause of carpal tunnel syndrome, or else the diagnosis might be missed. Specific therapy in such cases may help in alleviating patient suffering and working toward the common goal of better patient care. v Disclosure Statement The author(s) have no conflicts of interest to disclose.
Figure 1. Chest computed tomography scan showing right paratracheal and bilateral hilar nonnecrotic, noncalcific lymphadenopathy.
The patient was treated with corticosteroids, after which her symptoms of malaise, cough, anorexia, and weight loss subsided. Other causes of carpal tunnel syndrome were ruled out. A carpal tunnel splint was prescribed for the patient, after which she recovered.
DISCUSSION
This patient, who presented with neurologic complaints, turned out to have a case of neurosarcoidosis with carpal tunnel syndrome. Other common causes of carpal tunnel syndrome were ruled out on investigations. The mechanism of neurologic involvement in sarcoidosis is not clear. Granulomatous inflammation of nerve layers, secondarily caused by vasculitic neuropathy, demyelination, panangiitis, compression by sarcoid tissue, or thick edema under perineural tissue are some of the proposed causes of nerve involvement in sarcoidosis.2 Treatment is mainly corticosteroid based and physical therapy. Our patient responded well to the standard line of treatment.
2
Acknowledgment Kathleen Louden, ELS, of Louden Health Communications provided editorial assistance. How to Cite this Article Sonambekar A, Gupta N, Swadi A, Tomar LR. Carpal tunnel syndrome in sarcoidosis: A case report of a rare neurologic manifestation. Perm J 2016 Fall;20(4):15-168. DOI: http://dx.doi.org/10.7812/TPP/15-158. References
1. Burns TM. Neurosarcoidosis. Arch Neurol 2003 Aug;60(8):1166-8. DOI: http://dx.doi. org/10.1001/archneur.60.8.1166. 2. Nakatani-Enomoto S, Aizawa H, Koyama S, Kuroda K, Kimura T, Kikuchi K. Transient swelling of peripheral nerves in a case of neurosarcoidosis. Intern Med 2004 Nov;43(11):1078-81. DOI: http://dx.doi.org/10.2169/internalmedicine.43.1078. 3. Scott TF. Neurosarcoidosis: progress and clinical aspects. Neurology 1993 Jan;43(1):8-12. 4. Niemer GW, Bolster MB, Buxbaum L, Judson MA. Carpal tunnel syndrome in sarcoidosis. Sarcoidosis Vasc Diffuse Lung Dis 2001 Oct;18(3):296-300. 5. Yanardağ H, Pamuk ON, Kiziltan M, Yildiz H, Demirey S, Karayel T. An increased frequency of carpal tunnel syndrome in sarcoidosis. Results of a study based on nerve conduction study. Acta Medica (Hradec Kralove) 2003;46(4):201-4. 6. Shambaugh GE, Cirksena WJ, Newcomer KL. Carpal tunnel syndrome as manifestation of sarcoidosis. Arch Intern Med 1964 Dec;114:830-3. DOI: http://dx.doi. org/10.1001/archinte.1964.03860120142018.
The Permanente Journal/Perm J 2016 Fall;20(4):15-168
