Abstract
Autoimmune/inflammatory syndrome induced by adjuvants (ASIA) is associated with a hyperergic response of the immune system. The spectrum of clinical changes in ASIA is extensive and difficult to diagnose. Panniculitis is a heterogeneous group of diseases characterized by lesions of the adipose tissue. Panniculitis may become one of the signs of ASIA progress.
To describe the clinical signs and morphological changes in the subcutaneous fat as a manifestation of ASIA, developing after osteosynthesis with metal structures.
In this case-based review, were searched for all articles published in PubMed and Scopus databases until March, 2021 using the following keywords “panniculitis”, “erythema nodosum”, “Weber-Christian Disease”, “idiopathic lobular panniculitis”, “Autoimmune/inflammatory syndrome induced by adjuvants”, “Shoenfeld’s syndrome”, “metal allergy” “metal implants”, “metal hypersensitivity” and reviewed them. Irrelevant items and duplicates were excluded.
We report a case of an adult female patient presenting with a non-healing uninfected surgical wound following implantation of metal osteosynthesis construction and panniculitis developed after surgery. A comprehensive examination of the patient to check for ASIA was conducted. Considering the medical history of exposure to metals, tissue lesions, arthralgia, myalgia, low-grade fever, the disappearance of symptoms on the background of immunosuppressive therapy suggested the diagnosis of ASIA.
Report affords clinicians and pathologists additional insight into the clinical and morphological picture of ASIA-associated panniculitis which, if remain unidentified, may cause severe complications. We recommend the removal of constructions with adjuvant activity in patients with suspected ASIA-associated manifestations.
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig1_HTML.jpg)
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig2_HTML.jpg)
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig3_HTML.jpg)
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig4_HTML.jpg)
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig5_HTML.png)
![](http://media.springernature.com/m312/springer-static/image/art%3A10.1007%2Fs00296-021-04924-1/MediaObjects/296_2021_4924_Fig6_HTML.png)
Similar content being viewed by others
References
Segura S, Requena L (2008) Anatomy and histology of normal subcutaneous fat, necrosis of adipocytes, and classification of the panniculitides. Dermatol Clin 26:419–424. https://doi.org/10.1016/j.det.2008.05.011
Wick MR (2017) Panniculitis: a summary. Semin Diagn Pathol 34:261–272. https://doi.org/10.1053/j.semdp.2016.12.004
Jara LJ, García-Collinot G, Medina G et al (2017) Severe manifestations of autoimmune syndrome induced by adjuvants (Shoenfeld’s syndrome). Immunol Res 65:8–16. https://doi.org/10.1007/s12026-016-8811-0
Shoenfeld Y, Agmon-Levin N (2011) ‘ASIA’–autoimmune/inflammatory syndrome induced by adjuvants. J Autoimmun 36:4–8. https://doi.org/10.1016/j.jaut.2010.07.003
Alijotas-Reig J, Garcia-Gimenez V, Llurba E, Vilardell-Tarrés M (2012) Autoimmune/inflammatory syndrome (ASIA) induced by biomaterials injection other than silicone medical grade. Lupus 21:1326–1334. https://doi.org/10.1177/0961203312458838
Alijotas-Reig J, Esteve-Valverde E, Gil-Aliberas N, Garcia-Gimenez V (2018) Autoimmune/inflammatory syndrome induced by adjuvants-ASIA-related to biomaterials: analysis of 45 cases and comprehensive review of the literature. Immunol Res 66:120–140. https://doi.org/10.1007/s12026-017-8980-5
Watad A, Quaresma M, Bragazzi NL et al (2018) The autoimmune/inflammatory syndrome induced by adjuvants (ASIA)/Shoenfeld’s syndrome: descriptive analysis of 300 patients from the international ASIA syndrome registry. Clin Rheumatol 37:483–493. https://doi.org/10.1007/s10067-017-3748-9
Israeli E, Agmon-Levin N, Blank M, Shoenfeld Y (2009) Adjuvants and autoimmunity. Lupus 18:1217–1225. https://doi.org/10.1177/0961203309345724
Agmon-Levin N, Shoenfeld Y (2008) Chronic fatigue syndrome with autoantibodies—the result of an augmented adjuvant effect of hepatitis-B vaccine and silicone implant. Autoimmun Rev 8:52–55. https://doi.org/10.1016/j.autrev.2008.07.026
Balbouzis T, Georgiadis T, Grigoris P (2016) Granulomatous lung disease: a novel complication following metallosis from hip arthroplasty. Hip Pelvis 28:249. https://doi.org/10.5371/hp.2016.28.4.249
Chao S-C, Lee Y-P, Lee JY-Y (2010) Eosinophilic cellulitis and panniculitis with generalized vesicular pustular id reaction after a molten aluminum burn. Dermatitis 21:E11–E15. https://doi.org/10.2310/6620.2010.10008
Radenska-Lopovok SG, Egorova ON, Belov BB et al (2019) Idiopathic lobular panniculitis: clinical and morphological correlations. Arkh Patol 81(3):37–44. https://doi.org/10.17116/patol20198103137
Gasparyan AY, Ayvazyan L, Blackmore H, Kitas GD (2011) Writing a narrative biomedical review: considerations for authors, peer reviewers, and editors. Rheumatol Int 31:1409–1417. https://doi.org/10.1007/s00296-011-1999-3
Requena L, Yus ES (2001) Panniculitis. Part II. Mostly lobular panniculitis. J Am Acad Dermatol 45:325–364. https://doi.org/10.1067/mjd.2001.114735
Ameratunga R, Gillis D, Gold M et al (2017) Evidence refuting the existence of autoimmune/autoinflammatory syndrome induced by adjuvants (ASIA). J Allergy Clin Immunol Pract 5:1551-1555.e1. https://doi.org/10.1016/j.jaip.2017.06.033
Alijotas-Reig J (2015) Human adjuvant-related syndrome or autoimmune/inflammatory syndrome induced by adjuvants. Where have we come from? Where are we going? A proposal for new diagnostic criteria. Lupus 24:1012–1018. https://doi.org/10.1177/0961203315579092
Kal BI, Evcin O, Dundar N et al (2008) An unusual case of immediate hypersensitivity reaction associated with an amalgam restoration. Br Dent J 205:547–550. https://doi.org/10.1038/sj.bdj.2008.981
Dietrich K-A, Mazoochian F, Summer B et al (2009) Intolerance reactions to knee arthroplasty in patients with nickel/cobalt allergy and disappearance of symptoms after revision surgery with titanium-based endoprostheses. J Dtsch Dermatol Ges 7:410–412. https://doi.org/10.1111/j.1610-0387.2008.06987.x
Fukahara K, Minami K, Reiss N et al (2003) Systemic allergic reaction to the percutaneous patent foramen ovale occluder. J Thorac Cardiovasc Surg 125:213–214. https://doi.org/10.1067/mtc.2003.125
Loyo E, Jara LJ, López PD, Puig AC (2013) Autoimmunity in connection with a metal implant: a case of autoimmune/autoinflammatory syndrome induced by adjuvants. Auto Immun Highlights 4:33–38. https://doi.org/10.1007/s13317-012-0044-1
Stejskal V (2014) Metals as a common trigger of inflammation resulting in non-specific symptoms: diagnosis and treatment. Isr Med Assoc J 16:753–758
Kręcisz B, Kieć-Świerczyńska M, Chomiczewska-Skóra D (2012) Allergy to orthopedic metal implant—a prospective study. Int J Occup Med Environ Health. https://doi.org/10.2478/s13382-012-0029-3
Kagan P, Halpert G, Amital H et al (2020) Autoimmune/inflammatory syndrome induced by adjuvant associated with a metal implant in the mouth; explantation was followed by recovery. Isr Med Assoc J 9:516–517
Towers WS, Kurtom K (2020) Rare systemic response to titanium spinal fusion implant: case report. Cureus. https://doi.org/10.7759/cureus.7109
Ter Poorten MC, Thiers BH (2002) Panniculitis. Dermatol Clin 20:421–433. https://doi.org/10.1016/s0733-8635(02)00008-6
Requena L, Yus ES (2008) Erythema nodosum. Dermatol Clin 26:425–438. https://doi.org/10.1016/j.det.2008.05.014
Caputo V, Rongioletti F (2018) Panniculitis in the setting of dermato/rheumatologic diseases. G Ital Dermatol Venereol 153:194–207
Llamas Velasco M, Pérez-Gónzalez YC, Kempf W et al (2018) Clues in histopathological diagnosis of panniculitis. Am J Dermatopathol 40:155–167. https://doi.org/10.1097/DAD.0000000000000985
Poorten MAT, Thiers BH (2000) Systemic Weber-Christian disease. J Cutan Med Surg 4:110–112. https://doi.org/10.1177/120347540000400214
Rotondo C, Corrado A, Mansueto N et al (2020) Pfeifer-Weber-Christian disease: a case report and review of literature on visceral involvements and treatment choices. Clin Med Insights Case Rep. https://doi.org/10.1177/1179547620917958
Requena L, Yus ES (2001) Panniculitis. Part I. Mostly septal panniculitis. J Am Acad Dermatol 45:163–186. https://doi.org/10.1067/mjd.2001.114736
Funding
None.
Author information
Authors and Affiliations
Contributions
Substantial contributions to the management of the case and design of this case-based review were made by all the authors. SG: project manager, review and analyzing the literature, editing the manuscript. AO: review and analyzing the literature, writing the manuscript. ON: patient’s supervising. MV: ultrasound examination providing. ID: patient’s treating. All authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical approval
The study was approved by the V.A. Nasonova Research Institute of Rheumatology Ethics Committee, reference number 2 on the 31/01/2013 and number 28 on the 13/11/2014.
Informed consent
Informed consent was obtained from the patient included in the study.
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Radenska-Lopovok, S.G., Kolesnikova, A.O., Egorova, O.N. et al. Panniculitis as a manifestation of metal-associated autoimmune/inflammatory syndrome induced by adjuvants: a case-based review. Rheumatol Int 43, 1547–1553 (2023). https://doi.org/10.1007/s00296-021-04924-1
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-021-04924-1