What evidence is there supporting the use of methotrexate for neurosarcoidosis and cardiac sarcoidosis?

Comment by InpharmD Researcher

Evidence evaluating the use of methotrexate for the treatment of neurosarcoidosis and cardiac sarcoidosis is scarce, limited to retrospective/observational studies or anecdotal reports. For cardiac sarcoidosis, improvements in cardiac imaging parameters and ejection fraction have been found with use of methotrexate; in neurosarcoidosis, methotrexate use has been associated with reduced relapse rate compared to other immunosuppressive agents (notably, compared to mycophenolate mofetil). Retrospective studies for both sites of disease have shown either similar or improved outcomes when compared to glucocorticoids; the clinical significance of these findings are largely unknown, as randomized controlled trials are seldom performed.

Background

A 2021 review discussing use of non-steroidal treatment in cardiac sarcoidosis describes available studies evaluating the use of various agents. Of 480 cases of cardiac sarcoidosis treated, methotrexate was the synthetic agent used most often (n= 83). It should be noted that studies differed in the criteria used for the diagnosis of cardiac sarcoidosis and typically used methotrexate as an adjunct to low-dose maintenance steroid therapy. Overall, methotrexate generally led to favorable results through improved cardiac imaging and left ventricular ejection fraction, but available data are primarily limited to smaller observational studies and case series/reports (see Table 1). [1]

A 2020 review discusses the management of cardiac sarcoidosis. Once identified, management is primarily based on the use of immunosuppressants, namely corticosteroids. However, there is an overall lack of recommendations or guidelines for optimal treatment. Methotrexate has been utilized both in steroid-saving scenarios and in combination with other steroids, such as prednisone. The benefits are typically observed in small-population studies, or individual case reports, as randomized controlled trials are seldom performed. A prominent study known as the Cardiac Sarcoidosis Multi-Center Randomized Controlled Trial (CHASM CS– RCT) will investigate the efficacy of low- versus standard-dose prednisone in combination with methotrexate and is expected to be completed by December 2024. [2], [3]

The 2021 European Respiratory Society (ERS) guidelines on the treatment of sarcoidosis include recommendations for the management of neurological and cardiac disease. Glucocorticoids are recommended for patients with clinically significant neurosarcoidosis (strong recommendation, very low quality of evidence). In patients with continued disease who have been treated with glucocorticoids, the guidelines recommend adding methotrexate (conditional recommendation, very low quality of evidence). An analysis from a single institution found a significant reduction in the relapse rate of neurosarcoidosis with methotrexate (hazard ratio [HR] 0.47; 95% confidence interval [CI] 0.25 to 0.87; p= 0.02) and hydroxychloroquine (HR 0.37; 95% CI 0.15 to 0.92; p= 0.03), but not with azathioprine (HR 1.88; 95% CI 0.69 to 5.14; p= 0.22) or mycophenolate mofetil (HR 0.58; 95% CI 0.25 to 1.34; p= 0.2). This study also found infliximab significantly lowered relapse rates with sarcoidosis; however, the relapse rate was not significantly lowered with infliximab for neurosarcoidosis. A meta-analysis described the treatment of neurosarcoidosis with methotrexate, azathioprine, and hydroxychloroquine in 27% (144/539) of patients; of the patients who were not switched to third-line therapy, a favorable outcome was observed in 55% (47/85) of patients (95% CI 45 to 66%). A retrospective analysis of patients (Table 3) with neurosarcoidosis treated with either methotrexate (n= 32) or mycophenolate mofetil (n= 14) as part of the regimen showed that methotrexate treatment was associated with a significantly lower yearly relapse rate (0.2 vs. 0.6; p= 0.058) and longer median time to relapse (28 vs. 11 months; p= 0.049). Overall, clinical evidence for the treatment of sarcoidosis is limited since there are no randomized controlled trials, as well as a wide range of outcomes evaluated in retrospective studies of various different medications. [4]

With regards to cardiac sarcoidosis, the ERS guidelines recommend glucocorticoids (with or without other immunosuppressives) for patients with evidence of functional cardiac abnormalities, including heart block, dysrhythmias, or cardiomyopathy (strong recommendation, very low quality of evidence). The most commonly prescribed steroid-sparing immunosuppressive agents reported include methotrexate, azathioprine, mycophenolate mofetil, leflunomide, and cyclophosphamide; evidence to support these therapies is poor and in most studies, patients treated with steroid-spring agents did not have better outcomes than those treated with glucocorticoid monotherapy. However, one single-center study comparing methotrexate plus prednisone compared with prednisone monotherapy suggested improved ejection fraction and brain natriuretic peptide after five years of treatment. [4]

A 2020 review discussed the treatment options for neurosarcoidosis. While corticosteroids are the mainstay of treatment, immunosuppressive agents, including methotrexate, are considered an important treatment option both as a steroid-sparing option and as disease modifiers. Corticosteroids are effective as they reduce inflammation, while immunosuppressants work to prevent recurrence. The author notes that methotrexate has been shown to be better than mycophenolate mofetil in both systemic and neurological disease. The standard dose of methotrexate is 15-20 mg weekly along with folate rescue; the dose can be adjusted according to disease response, corticosteroid dose, and adverse events. [5]

A 2017 review also briefly discusses the role of methotrexate in the treatment of neurosarcoidosis. In patients with partial, or without sustained response to steroids or if long-term treatment is required, methotrexate is a second-line option, along with hydroxychloroquine, azathioprine, and cyclophosphamide. [6]

References: [1] Gallegos C, Oikonomou EK, Grimshaw A, Gulati M, Young BD, Miller EJ. Non-steroidal treatment of cardiac sarcoidosis: A systematic review. Int J Cardiol Heart Vasc. 2021;34:100782. Published 2021 Apr 29. doi:10.1016/j.ijcha.2021.100782
[2] Gilotra N, Okada D, Sharma A, Chrispin J. Management of Cardiac Sarcoidosis in 2020. Arrhythm Electrophysiol Rev. 2020;9(4):182-188. doi:10.15420/aer.2020.09
[3] U.S. National Institute of Health (NIH). ClinicalTrials.gov. Cardiac Sarcoidosis Randomized Trial (CHASM-CS-RCT). Updated October 26, 2021. Accessed April 21, 2023. https://clinicaltrials.gov/ct2/show/NCT03593759
[4] Baughman RP, Valeyre D, Korsten P, et al. ERS clinical practice guidelines on treatment of sarcoidosis. Eur Respir J. 2021;58(6):2004079. Published 2021 Dec 16. doi:10.1183/13993003.04079-2020
[5] Kidd DP. Neurosarcoidosis: clinical manifestations, investigation and treatment. Pract Neurol. 2020;20(3):199-212. doi:10.1136/practneurol-2019-002349
[6] Ibitoye RT, Wilkins A, Scolding NJ. Neurosarcoidosis: a clinical approach to diagnosis and management. J Neurol. 2017;264(5):1023-1028. doi:10.1007/s00415-016-8336-4
Literature Review

A search of the published medical literature revealed 5 studies investigating the researchable question:

What evidence is there supporting the use of methotrexate for neurosarcoidosis and cardiac sarcoidosis?

Please see Tables 1-5 for your response.


 

Cardiac Effects of Methotrexate Given Concurrently With Steroids for Treatment of Cardiac Sarcoidosis

Author, year

Treatments studied Other prior/concurrent treatments Study size (n) Follow-up (months) Cardiac outcomes

Yazaki et al., 2014

MTX

Concurrent: maintenance corticosteroids

7 N/A

PET: Improvement in myocardial perfusion or decreased myocardial uptake of 67Ga was observed in 2 patients (28.6%).

Metabolic: HbA1c decreased from 7.6 ± 1.9 to 7.2 ± 2.0% (p< 0.05) and triglycerides from 226 ± 124 to 167 ± 118 mg/dl (p< 0.05).

Yokomatsu et al., 2018

MTX

Concurrent: prednisolone taper

6 Mean: 17.3

PET: Reduction of 18F-FDG uptake area in all cases (6, 100%) with almost complete disappearance in 3 (50%) cases. SUVmax also decreased in all cases, the average index significantly reduced from 11.70 to 5.08 (p= 0.002).

LVEF: Mean LVEF increased from 41.8% to 45.4%.

Nagai et al., 2014

MTX Concurrent: prednisone 10 (7 steroid-only) 12, 36, 60

LVEF (steroid-only vs MTX group): baseline 52.3 ± 6.07 vs 49.7 ± 6.9 (p= 0.46); 3 years 44.5 ± 13.8 vs 60.7 ± 14.3 (p= 0.04); 5 years 45.7 ± 15.5 vs 53.6 ± 13.3 (p= 0.350).

NT-proBNP (steroid-only vs MTX group): baseline 955.5 ± 551.9 vs 621.4 ± 444.9 pg/mL (p= 0.16); 5 years 2,839.5 ± 3,953.3 vs 494.5 ± 609.8 (p= 0.04).

Ballul et al., 2019

MTX (5, 41.7%)

AZA (5, 41.7%)

CP (2, 16.7%)

Concurrent: corticosteroids 12 (24 steroid-only) Median: 3.6 (range 1 to 15.2)

Clinical relapse (reduced LVEF, 3rd degree atrioventricular block, atrial/ventricular tachycardia, sudden cardiac death): 6.7% (2/12) in the combined non-steroidal/steroidal group versus 45.8% (11/24) in the steroid-only group (p= 0.048).

Chapleon-Abric et al., 2017

CP (20, 57.1%)

MTX (12, 34.3%)

MMF (2, 5.7%)

Cyclosporine A (1, 2.9%-transplant)

Concurrent: corticosteroid taper 59 (35 treated with steroid-sparing) Median: 60

Clinical relapse (reappearance of abnormalities on EKG, echocardiography and one other imaging method on previously healed lesions): The recovery rate was 75% (18/24) for patients who received steroids alone versus 82.9% (29/35) for those who received steroids plus non-steroidal treatment (11/12 cases with MTX and 17/20 cases with CP).

Rosenthal et al., 2019

MTX (25) ± ADA (19, if persistent symptoms or intolerance to MTX)

Concurrent: prednisone taper 28 Mean: 49.2 ± 18

PET: MTX (+prednisone taper) led to reduction (88%) or elimination (60%) of 18F-FDG uptake. ADA resulted in improved (84%) or resolved (63%) 18F-FDG uptake. Radiologic relapse occurred in 8/9 of the patients that stopped immunosuppression, 4/25 of the patients on MTX, and 0/19 patients on ADA.

Arrhythmia: Three out of 19 patients who remained on uninterrupted immunosuppression developed VT, while 3/9 who discontinued immunosuppression developed VT after interruption of therapy.

Sethi et al., 2018

MTX (15, 100%), ADA (added in 8 [53%])

Concurrent: corticosteroids (94%)

15 Median: 24

PET: All 53% patients who were started on ADA had normalization of perfusion defects which were still present when they were on MTX. Stopping MTX early for non-compliance or side effects led to recurrence of perfusion defects in three (20%) patients.

LVEF: The percentage change in LVEF was 45 ± 12.91%, 50.6 ± 11.4% and 50.9 ± 10.7% at the initial and follow-up scans.

ADA: adalimumab; AZA: azathioprine; (NT-pro)-BNP: N-terminal pro-brain natriuretic peptide; CP: cyclophosphamide; CS: cardiac sarcoidosis; EKG: electrocardiogram; MMF: mycophenolate mofetil; MTX: methotrexate; N/A: not available; PET: positron emission tomography; VT, ventricular tachycardia

 

References:
[1] Adapted from: Gallegos C, Oikonomou EK, Grimshaw A, Gulati M, Young BD, Miller EJ. Non-steroidal treatment of cardiac sarcoidosis: A systematic review. Int J Cardiol Heart Vasc. 2021;34:100782. Published 2021 Apr 29. doi:10.1016/j.ijcha.2021.100782

 

Prednisone vs methotrexate in treatment naive cardiac sarcoidosis

Design

Retrospective cohort study

N= 61

Objective

To compare the effects of prednisone monotherapy, methotrexate monotherapy, or a combination of low-dose prednisone and methotrexate on myocardial fluorine-18 fluorodeoxyglucose (FDG) uptake and clinical outcomes in treatment-naive cardiac sarcoidosis (CS) patients

Study Groups

Prednisone (n= 21)

Methotrexate (n= 30)

Low-dose prednisone and methotrexate (n= 10)

Inclusion Criteria

Age ≥ 18 years; diagnosed with CS by clinical consensus in a multidisciplinary team; baseline myocardial FDG uptake available; a minimum of 6 months follow-up; immunosuppressive therapy with oral prednisone and/or methotrexate initiated within 3 months after CS diagnosis

Exclusion Criteria

Patients treated with immunosuppressive therapies in the past 3 months before baseline

Methods

Eligible patients were identified and included in the study. Patients received either prednisone monotherapy, methotrexate monotherapy, or a combination of low-dose prednisone and methotrexate. The prednisone monotherapy dose typically consisted of a starting dose of 40 mg daily for 1 month, followed by a taper to 20 mg daily at 3 months and 10 mg daily at 6 months. The methotrexate monotherapy and combination therapy started with 10 mg weekly and was increased to 15 mg weekly over a 4-week period. Patients receiving methotrexate also received folic acid at a dosage of 5 mg weekly or biweekly. Patients with combination therapy typically received prednisone 20 mg daily for 1 month, followed by prednisone taper to approximately 10 mg daily at 3 months. Subsequent adjustment of doses for all the regimens was made based on findings from clinical follow-up, FDG PET/CT, and side effects.

Duration

Between January 2010 and December 2017

Outcome Measures

Primary: metabolic response based on visual interpretation and quantitative analysis of cardiac FDG PET/CT within 12 months from initiation of treatment

Secondary: treatment patterns, major adverse cardiovascular events (MACE), change in left ventricular ejection fraction (LVEF), biomarkers, and side effects during 24 months after treatment start

Baseline Characteristics

 

Prednisone (n= 21)

Methotrexate (n= 30)

Prednisone + Methotrexate (n= 10)

 

Demographics

Age, years

Male

Caucasian

BMI, kg/m2

Hypertension

Diabetes

Coronary artery disease

 

50.8

81.0%

95.2%

25.4

9.5%

0

 

54.8

70.0%

100%

29.4

46.7%

13.3%

6.7%

 

49.2

80.0%

100%

28.5

30.0%

10.0%

 

Disease severity

Extra-cardiac sarcoidosis

Pulmonary

Neurologi

Liver

Ocular

Isolated cardiac sarcoidosis

 

 

90.5%

4.8%

28.6%

4.8%

4.8%

 

 

80.0%

6.7%

10.0%

10.0%

3.3%

 

 

90.0%

0

20.0%

10.0%

0

 

Cardiac manifestations

NYHA functional class I

NYHA functional class II

NYHA functional class III

Ventricular arrhythmias

Second/third degree AVB

LVEF, %

LGE on CMR

 

47.6%

42.9%

9.5%

28.6%

28.6%

52.0

90.5%

 

40.0%

36.7%

23.3%

10.0%

16.7%

56.0

88.9%

 

30.0%

50.0%

20.0%

0

40.0%

56.0

90.0%

 

Treatment

ICD or pacemaker

Antiarrhythmic treatment

 

76.2%

57.1%

 

43.3%

40.0%

 

60.0%

10.0%

 

Biomarkers

sIL-2R (pg/mL)

NT-proBNP (pg/mL)

 

4,152

217 

 

4,851

184

 

7,325

141

 

ICD, implantable cardioverter defibrillator; AVB, atrioventricular block; LGE, late gadolinium enhancement; CMR, cardiac magnetic resonance imaging

Results

Endpoint

Prednisone (n= 21)

Methotrexate (n= 30)

Prednisone + Methotrexate (n= 10) 

p-Value

Visual PET evaluation

Baseline PET, n

Diffuse

Focal/focal on diffuse

Follow-up PET, n

No uptake

Diffuse

Focal/focal on diffuse

p-value

 

21

3 (14.3%) 

18 (85.7%)

18

4 (22.2%) 

3 (16.7%) 

11 (61.1%)

0.125

 

30

8 (26.7%) 

22 (73.3%)

30

13 (43.3%) 

6 (20.0%) 

11 (36.7%)

<0.001

 

10

2 (20%)

8 (80%)

8

7 (87.5%)

0

1 (12.5%)

0.016

 

 

0.556

--

 

0.019¶§

--

--

--

Quantitative PET evaluation

Baseline PET, n

Myocardial SUVmax

Follow-up PET, n

Myocardial SUVmax

p-value

Changes in SUVmax

PET treatment responder

 

21

7.3 (5.4 to 11.3)  

18

3.7 (2.7 to 5.4)

0.002 

-47 (-70 to -26) 

13 (72.2%)  

 

30

6.5 (4.7 to 9.0)

30

3.4 (1.9 to 4.8)

0.001

-38 (-66 to -12)

20 (66.7%)

 

10

6.3 (4.5 to 9.7)

8

2.2 (1.8 to 2.7)

0.012

-67 (-72 to -56)

7 (87.5%)

 

 

0.465

 

0.093

 

0.248

0.468 

≥1 MACE event, n

7 (33.3%)

6 (20.0%) 1 (10.0%)  0.292 

p< 0.05 (prednisone vs prednisone+methotrexate); §p< 0.05 (methotrexate vs prednisone+methotrexate)

Prior to 2016, initial cardiac sarcoidosis treatment of 27 patients consisted of prednisone (n= 20), methotrexate (n= 3), or combination treatment (= 4). From 2016 onwards, initial therapies in 34 patients were prednisone (n= 1), methotrexate (n= 27), or combination treatment (n= 6). After 24 months of follow-up, 34 patients (55.7%) remained on methotrexate monotherapy or were switched to methotrexate monotherapy.

MACE experiences involved appropriate ICD therapy (n= 10), hospitalization due to heart failure (n= 3), and new third-degree AVB (n= 1). Six out of 10 patients experiencing appropriate ICD therapy during follow-up already showed ventricular arrhythmias at baseline (p< 0.001). 

Median follow-up LVEF measurements did not differ significantly from baseline values in all treatment groups. NT-pro-BNP values were significantly decreased at follow-up only in the combination treatment group. Follow-up sIL-2R values were available for 60 patients at a median interval of 22 months from treatment initiation and were significantly lower than baseline in all groups. 

Between baseline and follow-up FDG PET/CT, BMI increased significantly from 25.6 ± 3.2 to 26.3 ± 3.3 kg/m2 in the prednisone group (p= 0.020). The increase in the other treatment groups was not significant.

Adverse Events

Prednisone group: obstructive sleep apnea (OSA) (n= 1), osteopenia (n= 1), and Achilles tendon rupture (n= 1)

Methotrexate group: abnormal liver function tests (n= 2), hospitalization for infection (n= 1), complex partial seizures (n= 1), OSA (n= 1), erectile dysfunction (n= 1), and hair loss (n= 1)

Prednisone + Methotrexate group: hospitalization for infection (n= 2), new onset diabetes (n= 1) and unacceptable weight gain (n= 1)

Study Author Conclusions

Significant suppression of cardiac FDG uptake occurred in CS patients after 6 months of prednisone, methotrexate, or combination therapy. There were no significant differences in clinical outcomes during follow-up. These results warrant further investigation of methotrexate treatment in CS patients. 

InpharmD Researcher Critique

The study is limited by its retrospective design and small sample size. The comparison of efficacy and safety effects of the different treatment regimens are limited to the study duration period of 6 months, and more long-term effects cannot be determined from the results.



References:
[1] Vis R, Mathijssen H, Keijsers RGM, et al. Prednisone vs methotrexate in treatment naïve cardiac sarcoidosis [published online ahead of print, 2023 Jan 14]. J Nucl Cardiol. 2023;10.1007/s12350-022-03171-6. doi:10.1007/s12350-022-03171-6

 

Treatment of Neurosarcoidosis: A Comparative Study of Methotrexate and Mycophenolate Mofetil

Design

Retrospective, multicenter, observational, cohort study

N= 40

Objective

To compare the efficacy of methotrexate (MTX) and mycophenolate mofetil (MMF) in the prevention of relapses in neurosarcoidosis

Study Groups

Methotrexate (n= 26)

Mycophenolate mofetil (n= 8)

Both treatments (n= 6)

Inclusion Criteria

Definite or probable diagnosis of neurosarcoidosis; treated with a 3-month course of either MTX or MMF (or both with a minimum of 3 months washout)

Exclusion Criteria

No histologically proven sarcoidosis; insufficient data available

Methods

This was a retrospective chart review of patients from four university hospitals in France. Patients were diagnosed with neurosarcoidosis based on: clinical neurologic signs; lumbar puncture analysis and radiologic features; neurologic (definite neurosarcoidosis) or extraneurologic (probable neurosarcoidosis) noncaseating granuloma; and exclusion of differential diagnoses.

Patients who met the inclusion criteria were followed, with the time of first drug administration considered their baseline. Both MTX and MMF were administered with corticosteroids.

Duration

2010 to 2014

Outcome Measures

Incidence of relapse

Baseline Characteristics

 

Methotrexate (n= 26)

Mycophenolate mofetil (n= 8)

Both (n= 6)

 

Median age, years (range)

43.5 (21-77) 48 (17–64) 29 (13-59)  

Female

46.1% 37.5% 16.6%  

African American

23.1%

37.5%

66.6%

 

Other organ involvement

Heart

Lungs

Eyes

Lymph nodes

 

12.5%

69.2%

46.2%

92.3%

 

19.2%

37.5%

37.5%

62.5%

 

50%

83.3%

50%

83.3%

 

Neurologic involvement

Brain

Cranial nerves

Meninges

Spinal cord

Radiculae

Peripheral nerve

 

34.6%

46.1%

38.5%

11.5%

3.8%

7.7%

 

50%

25%

12.5%

25%

12.5%

12.5%

 

66.6%

66.6%

50%

33.3%

16.6%

0

 
Of the patients that received both, three received MTX followed by MMF and three received MMF followed by MTX.

Results

Endpoint

Methotrexate (n= 26)

Mycophenolate mofetil (n= 8)

Both (n= 6)

p-value

Duration of treatment, months (range)

13.5 (3-113) 10.5 (4-47) N/A 0.31

Median dose (range)

20 mg/wk (10-30) 2 g/d (1-3) N/A N/A

Relapse

15/32 (46.8%) 11/14 (78.6%) N/A 0.058

Median time of survival without relapse, months

28 11 N/A 0.0491

Required second-line therapy with infliximab or cyclophosphamide

13/15 (87%) 7/10 (70%) N/A  

Steroid dose

Start of treatment

At time of relapse (or end of follow-up)

 

40 mg/d

12.5 mg/d

 

20 mg/d

5 mg/d

 

 

 

0.08

Adverse Events

11 (34%) of MTX patients experienced an adverse event. The most common were infection (19%) and cytolysis (6%). Of the 6 patients who experienced infection, 2 required discontinuation of MTX.

Only 1 patient experienced an adverse event with MMF; this was not serious and did not require discontinuation.

Study Author Conclusions

Neurosarcoidosis leads to a high rate of relapses, and MTX seems more efficacious at preventing relapses than MMF.

InpharmD Researcher Critique

Limitations of this study include the retrospective, observational nature. There is a potential for selection bias, based on the provider-chosen treatments. It appears MMF was given to patients with more severe disease, possibly resulting in higher relapse rates. The dose of medication was heterogeneous; however, there does not appear to be a correlation between dosage and occurrence of relapse. The concurrent steroid dose was also higher in the MTX group, possibly influencing the relapse rate. 



References:
[1] Bitoun S, Bouvry D, Borie R, et al. Treatment of neurosarcoidosis: A comparative study of methotrexate and mycophenolate mofetil. Neurology. 2016;87(24):2517-2521. doi:10.1212/WNL.0000000000003431

 

Neurosarcoidosis: Longitudinal Experience in a Single-center, Academic Healthcare System

Design

Retrospective, single-center, observational trial

N= 56

Objective

To review the long-term follow-up of a neurosarcoidosis patient population at a tertiary referral center, including a report of the various treatments used and the response to therapy

Study Groups

Neurosarcoidosis (N= 56)

Inclusion Criteria

Patients with at least one ICD-9 or ICD-10 code for sarcoidosis and at least one outpatient visit with a neurologist

Exclusion Criteria

Did not meet the diagnostic criteria for neurosarcoidosis; insufficient data

Methods

This was a retrospective chart review of patients seen at the University of Utah for neurosarcoidosis. Patients were categorized as having either definite, probable, or possible neurosarcoidosis based on available chart information.

A “possible” neurosarcoidosis diagnosis required a clinical presentation and diagnostic evaluation suggestive of neurosarcoidosis without pathologic evidence. A “probable” neurosarcoidosis diagnosis required pathologic confirmation of systemic granulomatous disease consistent with sarcoidosis, and “definite” neurosarcoidosis criteria required nervous system pathology consistent with sarcoidosis.

Duration

July 1, 2010 to August 24, 2018

Outcome Measures

Treatments and their efficacy

Baseline Characteristics

 

Neurosarcoidosis (N= 56)

   

Age at symptom onset, years

49    

Female

63%    

Caucasian

84%    

Diagnosis

Definite

Probable

Possible

 

22%

64%

14%

   

Neruologic involvement

Central nervous system

Peripheral nervous system

 

80%

27%

   

Systemic involvement

Pulmonary

Lymphadenopathy

Joints

Ocular

Cardiac

 

50%

45%

27%

14%

4%

   

Results

Treatments and Outcomes

Improved

Stable

Failed

Prednisone

19 (37%) 13 (26%) 19 (37%)

Methotrexate

5 (19%) 9 (35%) 12 (46%)

Azathioprine

5 (38%) 1 (8%) 7 (54%)

Infliximab

10 (45%) 9 (41%) 3 (14%)

Rituximab

0 2 (50%) 2 (50%)

Mycophenolate mofetil

0 2 (25%) 6 (75%)

Hydroxychloroquine

1 (100%) 0 0

Cyclophosphamide

1 (50%) 0 1 (50%)

Adverse Events

Not reported

Study Author Conclusions

This is a comprehensive characterization of neurosarcoidosis within a single healthcare system at the University of Utah that reports long-term response to treatment and outcomes of patients with neurosarcoidosis. These results suggest the use of infliximab as a first-line therapy for neurosarcoidosis.

InpharmD Researcher Critique

This study was observational and limited by the retrospective design. The incidence of concurrent therapy was not reported; however, it was implied that each treatment was given as monotherapy. Likewise, the sequence of treatments was not reported. 



References:
[1] Lord J, Paz Soldan MM, Galli J, et al. Neurosarcoidosis: Longitudinal experience in a single-center, academic healthcare system. Neurol Neuroimmunol Neuroinflamm. 2020;7(4):e743. Published 2020 May 13. doi:10.1212/NXI.0000000000000743

 

Diagnosis and Management of Neurological Sarcoidosis

Design

Retrospective, single-center, observational cohort study

N= 71

Objective

To report a single center's experience of the manifestations, clinical outcome, and treatment regimens in patients with neurological sarcoidosis

Study Groups

Treated for neurosarcoidosis (n= 48)

Inclusion Criteria

Patients diagnosed with sarcoidosis with neurological symptoms

Exclusion Criteria

External causes of neurological symptoms were present or suspected

Methods

This was a retrospective chart review at the University of Cincinnati of patients diagnosed with sarcoidosis with neurological symptoms. All patients received at least a trial of corticosteroid therapy with an initial dosage of 40 mg/d of prednisone for 1 month. Patients were not considered corticosteroid failures unless they received 20 mg/d or more of prednisone for more than 6 months with worsening symptoms.

Patients who were stable on a regimen of 20 mg/d or more of prednisone but relapsed on lowering the dosage were also considered for alternative therapy. Patients were treated with methotrexate following a previously described protocol whereby patients initially received 10 mg/wk with the final dose ranging from 5 to 15 mg/wk based on tolerance and response.

Patients treated with cyclophosphamide received the drug intravenously 500-700 mg every 2 weeks. The doses were escalated until either neutropenia or other side effects were noted.

Duration

1986 to 1995

Outcome Measures

Treatments and response

Baseline Characteristics

 

Neurosarcoidosis (N= 71)

 

Female

67%  

African American

60%  

Other organ involvement

Lungs

Skin

Eyes

Liver

Heart

 

94%

30%

37%

13%

11%

 
 

Results

Therapy

Alive while receiving therapy at the end of follow-up (treatment success)

Received subsequent therapy (failure/relapse)

Prednisone (n= 48)

14 (29%) 30 (63%)

Methotrexate (n= 28)

17 (61%) 8 (29%)

Cyclophosphamide (n= 10)

8 (80%) 1 (10%)
Methotrexate and cyclophosphamide were each associated with a higher response rate than corticosteroids alone (p< 0.001).

Adverse Events

Three deaths occurred due to neurosarcoidosis in patients receiving prednisone. One death occurred due to neurosarcoidosis while receiving methotrexate. There were no deaths in the cyclophosphamide-treated patients.

One patient experienced methotrexate-induced hepatotoxicity which required discontinuation of the drug. This was the patient who died due to neurosarcoidosis.

Study Author Conclusions

Neurosarcoidosis remains a significant complication for patients with sarcoidosis. Long-term high-dose corticosteroid therapy was tolerated poorly by our patients. The use of immunosuppressive therapy, especially cyclophosphamide, can result in significant improvement in neurological status.

InpharmD Researcher Critique

Limitations of this analysis include the retrospective and observational design. While steroids (prednisone) were used as first-line therapy, the choice of subsequent therapy was either methotrexate or cyclophosphamide. The duration of treatment was not reported, so some patients may have recently started a medication when the follow-up period ended, possibly inflating the success results.



References:
[1] Lower EE, Broderick JP, Brott TG, Baughman RP. Diagnosis and Management of Neurological Sarcoidosis. Arch Intern Med. 1997;157(16):1864–1868. doi:10.1001/archinte.1997.00440370104011