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Factitious panniculitis mimicking chronic erythema nodosum

Ludmilla Luiza Libanio Guimarães1; Nurimar Conceição Fernandes2; Tullia Cuzzi3,4

DOI: https://doi.org/10.5935/scd1984-8773.2018102666

Received on: 07/07/2015

Approved on: 29/05/2018


This study was performed at the Hospital Universitário Clementino Fraga Filho (HUCFF). Universidade Federal do Rio de Janeiro (UFRJ) - Rio de Janeiro (RJ), Brazil.

Financial support: None

Conflict of interests: None


Abstract

Factitious panniculitis can be triggered by subcutaneous injections of substances linked to esthetic procedures. A case diagnosed and treated as chronic erythema nodosum manifested nodule five years later, with the anatomopathological study suggesting atypical lipomatous tumor. After resection of the lesion, histology showed steatonecrosis, compatible with lobular panniculitis. Magnetic resonance imaging examination revealed the presence of a liquid substance in the gluteal region, favoring the diagnostic suspicion of factitious panniculitis. The patient admitted to having undergone silicone injection in this region in the past, with additional findings arising from the histological review confirming this hypothesis. The late complication linked to injections of cutaneous filling material motivated the present study.


Keywords: Erythema chronicum migrans; Panniculitis; Silicones


INTRODUCTION

Panniculitides, a group of subcutaneous inflammatory conditions of septal or lobular predominance, with or without vasculitis, have multiple etiologies.1

Erythema nodosum (EN) migrans,Vilanova-Pinol Aguadé subacute panniculitis nodosa migrans (SPNM) and chronic EN, variants of classic EN, are now deemed as distinct phases of the same pathological process.1 Described in 1956 by Vilanova and Pinol Aguadé, 2 SPNM is singular due to the fact it has few, unilateral and asymmetrical nodules, which converge to form indurated plates of up to 20cm, with active centrifugal lesions and concomitant central involution, acquiring different shapes and shades. There is predominance in females and it is not very symptomatic, developing during months to years in the knees, buttocks and thighs.1

The objective of the present report is to demonstrate a late complication (10 years after), due to a cutaneous filling, and represented by panniculitis symptoms.

 

METHODS

The authors of the present study report the case of a 50-year-old, mulatto, not very assiduous female patient, who sought care at the Dermatology Clinic of a university medical service in the city of Rio de Janeiro, Brazil, in November 2001. She complained of an erythematous-infiltrated, indurated plaque measuring 25cm in length, with an increase in temperature, located in the anterior aspect of the right thigh (Figure 1), that had emerged four years before. In light of the hypothesis of panniculitis, a biopsy was performed with the anatomopathological study indicating an EN in its chronic phase. The possibly associated conditions (trichomoniasis, onychomycosis, paronychia, hidradenitis and scabies) were adequately treated in ambulatorially during the follow-up. The results of laboratory tests (blood count, biochemistry, thyroid function, serologies for hepatitis B and C, VDRL, anti-HIV, ASLO, alpha -1-antitrypsin, urinary sediment, parasitologic stool) and chest X-ray were normal. Non-hormonal anti-inflammatories, oral potassium iodide saturated solution (reaching only 15 drops / day due to gastrointestinal intolerance), potassium iodide in cream and systemic corticosteroid therapy during periods of more intense inflammation, were administered without significant change in the course of panniculitis.

In March 2006, the patient returned to the clinic reporting the emergence of a hardened painful nodule measuring 7cm on the lateral aspect of the left thigh one year before. Ultrasonographic evaluation described echogenic formations of imprecise limits on the subcutaneous external aspect of the left thigh, left inguinal region and the inner aspect of the right thigh. Subsequent incisional biopsy of the lesion was performed, with the anatomopathology revealing an atypical lipomatous tumor, negative for common germs, fungi and mycobacteria. In February 2012, the histological examination of the lesion’s exeresis (Figure 2) revealed extensive steatonecrosis. Magnetic resonance imaging performed after surgery evidenced liquid infiltration in the subcutaneous area of the gluteal region, in addition to multiple non-specific lymph nodes in the iliac and femoral chains (Figure 3).

 

RESULTS

Taking into account the steatonecrosis (a finding that is present in the histology of some lobular panniculitis), the image of liquid infiltration in the gluteal region, and the clinical history review (information provided by the patient about the silicone injections applied in the gluteal regions at least 10 years before), the hypothesis of factitious panniculitis caused by exogenous substance was raised.

The review of the histological samples added the presence of microcystic spaces in the hypodermis to the finding of steatonecrosis. Some of these spaces were delimited by amphiphilic material, and foam cells, in line with the hypothesis of factitious panniculitis (Figures 4-7). The absence of refraction in polarized light was consistent with the type of material reported by the patient (silicone).

 

DISCUSSION

Factitious or artificial panniculitis are lesions of the subcutaneous tissue that are caused by external factors (mechanical, chemical and physical), and belong in the group of lobular panniculitides without vasculitis.3,4 They can be self-induced, accidental, intentional, a manifestation of psychiatric disorder, or of the iatrogenic injection of substances. Although the etiopathogenic mechanism is unknown, vasoconstriction with ischemia at the sites of injection, inflammatory response to precipitated drugs and trauma by repeated injections have been implicated.3

Fillers can induce a range of adverse reactions — e.g. some may disappear spontaneously while others may lead to complications 30 years after the procedure, 3 at a time when the patient no longer remembers which material was injected.5

When the patient in question admitted to having undergone the filling procedure at a clandestine clinic before the 1990s, the authors of the present study hypothesized that the iatrogenic injection of nonabsorbable material in the past was responsible for the current manifestation. Omission of the procedure, often performed by professionals without the legally required qualification,6 is the probable explanation for the late diagnosis of this type of panniculitis.

Silicone is a polymeric hydrophobic compound that can be found in the forms of oil, gel or solid implant. In its liquid form, it was widely used for cosmetic correction of fine wrinkles, scars and facial tissue volumization of HIV positive patients with lipoatrophy.5,7

The migration of non-biodegradable fillers, such as silicone, is often described to mimic malignant neoplasms and granulomatous diseases.8 It is able to reach sites distant from the treated area, 5,7 causing local inflammatory reaction. The absence of a fibrous capsule involving large volumes allows gravitational migration of the implant towards undesired areas.9 It is believed that the injection of excessive bolus volumes precipitates this process.7 In the studied case, the authors suspected that the filling material previously injected in the buttocks migrated, via the subcutaneous plane, towards the thighs’ topography.

The histological pattern generally observed in factitious panniculitis is that of acute lobular panniculitis associated with steatonecrosis and inflammatory infiltrate with neutrophilic predominance, coursing with granulomatous appearance.3 Chronic lesions are characterized by foamy histiocytes and surrounding fibrosis.3,4 The important dermal involvement also assists in differentiating the shape induced by injected substances from other types of panniculitis.3

The histological findings associated with the use of silicone are varied and correspond more frequently to cutaneous nodules. In general, there is diffuse infiltrate of macrophages and multinucleated giant foreign body cells that delimit empty spaces of varying sizes, in a pattern known as “Swiss cheese.” The presence of cells with reactional cytological atypias — and even of lipoblasts — may justify the differential diagnosis vis a vis neoplastic processes.5,6,10

Due to the fact silicone is a permanent filler, the inflammatory response can occur at any time, constituting a therapeutic challenge.9 The literature describes treatments with local and systemic corticosteroids, 11 minocycline, 5,7,11 5% imiquimod, 5,7,11 isotretinoin and doxycycline.7

Morrondo et al.11 reported a case of factitious panniculitis in gluteal regions caused by silicone and EN (associated with Löfgren’s syndrome) in concomitant pretibial regions with good response to non-steroidal anti-inflammatory.

Intralesional corticosteroids are used in cases secondary to granulomatous reaction, 3,5 with removal of the implants in irresponsive cases, 3,7 which might require extensive resections and complex reconstructions.8

The loss of ambulatorial follow-up precluded therapeutic guidance after the review of this case.

 

CONCLUSION

The histological data, in addition to the previous cutaneous filling history, the migration potential of silicon, the chronic and atypical development, and the evidence of gluteal liquid infiltration, underpinned the conclusion of factitious panniculitis.

Adverse reactions to fillers can arise many years after the procedure has been carried out, and abundant documentation on migration of silicone to distant sites is available. A complete anamnesis, including information from the past and previous aesthetic treatments, should be especially performed in cases of chronic panniculitis.

 

DECLARATION OF PARTICIPATION:

Ludmilla Luiza Libanio Guimarães | ORCID 0000-0002-6543-610X
Diagnostic suspicion, literature research and manuscript write up

Nurimar Conceição Fernandes | ORCID 0000-0002-1819-9630
Patient ambulatorial follow-up, oversight of the manuscript write up and final review

Tullia Cuzzi | ORCID 0000-0002-3331-5290
Histological reports preparation, histological findings analysis and discussion, histological photographic records, critical review of the manuscript

 

REFERENCES

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2. Vilanova X, Pinol Aguadé J. Hypodermite Nodulaire Subaiguë Migratice. Annales de Dermatologie. 1956;83(4):369-401.

3. Sanmartín O, Requena C, Requena L. Factitial Panniculitis. Dermatol Clin. 2008; 26(4):519-27.

4. Jeong KH, Lee MH. Two cases of factitial panniculitis induced by electroacupuncture. Clin Exp Dermatol. 2008;34(5):e170-3.

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8. Hirsch RJ, Stier M. Complications of Soft Tissue Augmentation. J Drugs Dermatol. 2008;7(9):841-45.

9. Gilbert E, Hui A, Meehan S, Waldorf HA. The Basic Science of Dermal Fillers: Past and Present Part II: Adverse Effects. J Drugs Dermatol. 2012;11(9):1069-75.

10. Shvartsbeyn M, Rapkiewicz A. Silicon-associated subcutaneous lesion presenting as a mass: a confounding histopathologic correlation. Hum Patol. 2011;42(9):1364-7.

11. Morrondo CD, Fontana NP, Gontad JML, RNA, González MF, Silva JG et al. Paniculitis facticia y syndrome de Lõfgren inducidos por silicona: a propósito de un caso clínico. Reumatol Clin. 2012;8(6):368-71.


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