Multiple panniculitis presenting as orbital cellulitis: a rare case report

Abstract

Introduction:

Orbital cellulitis is an infectious inflammatory condition affecting structures surrounding the eye, posterior to the orbital septum. Panniculitis is an inflammatory disorder of subcutaneous fat, which may or may not be associated with vasculitis. Orbital cellulitis may be mimicked by non-infectious inflammatory conditions such as panniculitis, and these presentations are rarely reported in the literature.

Case summary:

A 65-year-old woman presented with pain and swelling in the left eye for 1.5 months, associated with redness and watering with no discharge. She also had multiple localized swellings on various body parts, including the right lateral flank and bilateral arms. On examination, approximately six lumps were found, with the largest located at the right lateral flank. Ocular examination of the left eye showed conjunctival congestion and edema and proptosis measuring 24 mm, with no ophthalmoplegia, restriction of eye movements, or visual disturbance. Initially, she was thought to have orbital cellulitis and was treated with topical and systemic antibiotics. Later, a biopsy of the left flank skin revealed panniculitis, and oral steroids were started, which led to signs of improvement.

Clinical discussion:

Since orbital cellulitis and orbital panniculitis have similar features, it is challenging for clinicians to differentiate between these two conditions. Early detection of orbital panniculitis can help clinicians prevent unnecessary antibiotics, interventions, or surgery, and direct them to definitive management.

Conclusion:

A diagnostic dilemma can occur when patients present with inflammatory conditions such as panniculitis. Non-infectious causes, such as panniculitis, should be considered in the context of patients presenting with subacute symptoms, a poor response to antibiotics, and associated systemic inflammatory lesions. A multidisciplinary approach involving clinicians from internal medicine and specialists in otolaryngology (ENT), rheumatology, and radiology can help with timely diagnosis and appropriate management in such cases.

Introduction

Orbital cellulitis, an infectious inflammatory process involving fat and muscles located posterior to the orbital septum within the orbital cavity, originates from structures surrounding the eye or from distant infectious sites via the hematogenous route^[1,2^]. It is more common in pediatric and young adult populations and is very rare in older adults ^[1,2^]. The causes include foreign bodies in the eye, trauma, dental procedures, and caries^[3^].

HIGHLIGHTS

• Orbital cellulitis is an infectious, inflammatory condition of the structures of the eye, and panniculitis is an inflammatory disorder of the subcutaneous fat.

• As orbital cellulitis and orbital panniculitis share similar features, it can be challenging for clinicians to distinguish between the two conditions.

• Early detection of orbital panniculitis can help clinicians prevent unnecessary antibiotics, interventions, or surgery and direct patients to definitive management.

Patients usually present to the emergency department with complaints of redness and swelling of the eyelid, vision loss, headache, fever, chemosis, diplopia, difficulty moving the eyeball, and outward bulging of the eyeball^[1,4^]. Diagnosis is usually based on clinical presentation, but further confirmation of the diagnosis; the extent and route of spread; response to intravenous (IV) antibiotics; and complications are assessed by CT and MRI^[2^]. It is managed with IV antibiotics, depending on the causative organisms, along with surgical procedures, while the role of IV steroids is controversial^[2,5^].

Orbital cellulitis can be mimicked by various conditions – such as orbital lymphoma, choroidal melanoma, rhabdomyosarcoma, sebaceous carcinoma, and panniculitis – which can present with symptoms similar to those of orbital cellulitis, including pain, swelling, restricted eye movement, bulging of the eyeball, and fever, making it very important to differentiate these conditions from true orbital cellulitis^[6–8^].

Panniculitis is a diverse group of inflammatory disorders that primarily involve the subcutaneous fat and can be classified as septal or lobular based on whether they are centered on fibrovascular septa or adipose lobules, which may or may not be associated with vasculitis^[9,10^]. Although panniculitis primarily affects subcutaneous fat, resulting in subcutaneous nodules and systemic features^[11^], inflammation involving periorbital or orbital adipose tissue, though rare, may produce edema, pain, erythema, and proptosis, similar to orbital cellulitis^[11^]. Since infectious and inflammatory orbital conditions may overlap clinically, it is very difficult to distinguish between inflammatory and infectious orbital conditions, particularly in atypical cases lacking classic infectious features. Here, we describe the rare case of a 65-year-old woman with systemic panniculitis presenting with orbital inflammatory signs resembling orbital cellulitis.

Case presentation

A 65-year-old woman, married, from Nuwakot, was brought to the emergency department with the chief complaint of pain and swelling in the left eye for 1.5 months, which worsened 1 day prior to admission. The onset was insidious and progressive (swelling followed by pain). It was associated with redness and tearing, with no discharge.

She also had multiple localized swellings distributed on various parts of her body (right lateral flank, both arms, right thigh, and chest) for the past 2 months. They were of insidious onset and gradually increased in size. The first lesion appeared on the right lateral flank, followed by the anterior aspects of both arms, the anterior aspect of the right thigh, and the chest. The largest lesion was located on the right lateral flank. It was associated with constant pain but no ulceration. She also had fever for 5-7 days, documented up to 102°F, with no chills or rigors.

There was no history of weight loss, night sweats, joint pain, abdominal pain, chest pain, or palpitations. There was no contact with a TB patient. There was no significant family history of medical or surgical illnesses.

On examination, she was alert, thin-built, conscious, cooperative, and well-oriented to time, place, and person. She did not have pallor, icterus, cyanosis, dehydration, or edema. Her blood pressure was 130/80 mmHg on the right hand in the sitting position, the pulse rate was 88 beats/min, regular, and the respiratory rate was 18 breaths/min. On local examination, approximately six lumps were found, with the largest located on the right lateral flank, measuring 15 × 7 cm, oval, firm, and smooth, with well-defined margins, and the overlying skin was erythematous with excoriation and biopsy marks. Fluctuation, slip sign, and transillumination were negative. On examination of the left eye, the conjunctiva was congested and edematous, and the periorbital area had watery discharge. Proptosis measurement with Hertel’s exophthalmometer revealed forward displacement of 24 mm. There was no ophthalmoplegia, restricted eyeball movement, adenopathy, or visual disturbance. There was no RAPD, and the pupillary reflexes were normal. The slit-lamp examination was normal. The rest of the physical examination findings were unremarkable.

Her blood counts, including eosinophil and platelet counts, were within the normal range. Her renal and liver function tests were normal. The ESR was normal, but the CRP level was elevated. The laboratory reports are shown in Figures 1 and 2.

Figure 1.

(A and B) Features of left orbital cellulitis with possible extension into the left cavernous sinus and no definite evidence of cavernous sinus thrombosis.

Figure 2.

(A) Scattered histiocyte-like cells with eosinophilic cytoplasm and an eccentric, oval, hyperchromatic nucleus (low power). (B) The subcutis shows lobular infiltration by histiocytes and lymphocytes. (C) The dermis shows edema and moderate perivascular lymphocytic infiltration. (D) Scattered histiocyte-like cells with eosinophilic cytoplasm and an eccentric, oval, hyperchromatic nucleus (high power).

In immunohistochemistry, epithelial membrane antigen (EMA) and pan-cytokeratin (Pan-CK) were negative, excluding carcinoma.

Her random blood sugar level was normal. The electrocardiogram findings were normal.

Diagnostic assessment

Imaging

CT head

CT of the head showed an ill-defined, heterogeneously enhancing, soft-tissue–density mass in the left orbit and pre-septal space, with marked proptosis and soft-tissue stranding in the left infraorbital fat, along with bulky lateral and inferior rectus muscles.

MRI of the brain and orbits

Biopsy

She was diagnosed with orbital cellulitis, hypertension, and hyperlipidemia, for which she was given ofloxacin and dexamethasone eye drops; carboxymethylcellulose sodium eye drops; ciprofloxacin and fusidic acid eye cream; and IV antibiotics (vancomycin, ceftriaxone, and metronidazole). Later, a biopsy of the left flank skin revealed panniculitis, and prednisolone 40 mg tablets were given on a tapering dose, with improvement in signs and symptoms. Amlodipine and losartan potassium (5 mg + 50 mg) were used to control her BP.

She was discharged after a 24-day hospital stay and was stable at the time of discharge.

Discussion

Orbital cellulitis, one of the most common types of orbital inflammatory disease, usually progresses as a result of the spread of disease from nearby sinuses and is more common in children than in adults^[12^]. Trauma, dermatological manifestations, panophthalmitis, and dental caries are often causative factors in adults^[12^].

Orbital cellulitis has also been reported to be mimicked by various conditions, such as orbital lymphoma^[6^], choroidal melanoma^[7^], rhabdomyosarcoma^[8^], and sebaceous carcinoma^[13^]. To the best of our knowledge, panniculitis of the orbital fat has not yet been reported in the existing literature as a mimicker of orbital cellulitis, although panniculitis is well documented in systemic or subcutaneous forms^[14^].

Fundamentally, in panniculitis, although it is non-infectious, there is an inflammatory reaction in the subcutaneous tissue, leading to the breakdown of adipocytes and cellular infiltration, including neutrophils, lymphocytes, histiocytes, or mixed infiltrates, further causing the release of a cascade of inflammatory mediators, and when panniculitis involves the orbital fat, it most likely triggers a surge of orbital inflammation^[7^].

This increased interstitial edema and the inflammatory expansion within the rigid orbital compartment increase the pressure within the limited space of the orbit, thereby compressing the extraocular muscles and compromising venous outflow. Further, due to the pressure effect, inflammation of the periorbital soft tissues occurs, producing erythema, pain, proptosis, and restricted extraocular motility, which raises stronger clinical suspicion of orbital cellulitis^[7^].

Although well-described in other orbital masses such as lymphomas, a similar mechanism of mass effect can be considered in panniculitis, where inflammatory enlargement of orbital fat may compress the optic nerve and thereby reduce visual acuity^[15^]. Based on the site and vascularity, dermatologists classify panniculitis into (1) septal with vascularity, (2) septal without vascularity, (3) lobular with vascularity, and (4) lobular without vascularity; however, the microscopic pictures usually overlap, and so does the clinical picture^[16^]. Compared with classic orbital cellulitis, several features in our patient were not typical for orbital cellulitis. Our patient had a subacute presentation over 1.5 months, no ophthalmoplegia, preserved visual function, and multiple painful subcutaneous nodules elsewhere in the body, in contrast to the acute, rapidly progressive course typically observed in orbital cellulitis, including ophthalmoplegia and impaired visual function^[17^]. Multiple painful subcutaneous nodules were among the observations that raised suspicion for the possibility of a systemic inflammatory process^[18^] rather than isolated infection. A predominantly inflammatory etiology was further confirmed by the significant clinical improvement following corticosteroid therapy and the poor response to antibiotic therapy.

Further investigation with a computed tomography scan of the head, orbit, and brain showed an ill-defined, heterogeneously enhancing, soft-tissue–density mass in the left orbit and pre septal space, with marked proptosis and soft-tissue stranding in the left infraorbital fat, with bulky lateral and inferior recti, suggesting preseptal or orbital cellulitis. MRI of the brain and orbit showed left orbital cellulitis with possible left cavernous sinus extension and no cavernous sinus thrombosis.

As the patient had multiple nodular swellings, T-cell lymphoma was clinically suspected, and a portion of the skin biopsy from the largest swelling on the right lower back was taken for T-cell clonal expansion studies and histopathological examination. The histopathology report noted increased dermal interstitial mucin with perivascular lymphocytic infiltrates and scattered histiocyte-like cells. Immunohistochemistry was negative for EMA and PanCK, which excluded carcinoma, and the findings were also not diagnostic of lymphoma. These features on histopathology suggest an inflammatory process involving subcutaneous fat. Taken together, correlation of the clinical presentation with the histopathological findings confirmed the diagnosis of panniculitis.

Orbital cellulitis is a medical emergency requiring urgent and aggressive intervention ^[17^]. Because orbital panniculitis and orbital cellulitis have similar clinical courses, it might be challenging for both clinicians and pathologists to differentiate orbital panniculitis from orbital cellulitis at first glance. Early detection of orbital panniculitis can prevent unnecessary antibiotics, interventions, or surgery, and lead to definitive management. The current case study demonstrates that an insidious onset, gradually progressing mild proptosis in a painful red eye with multiple other swellings must be aggressively investigated, and panniculitis should be considered a close differential diagnosis in cases mimicking orbital cellulitis.

Strengths and limitations

Strength

The patient's symptoms improved. This case report highlights the need for physicians to broaden their thinking beyond infectious etiologies when a patient presents with features suggestive of orbital cellulitis.

Limitations

This is a case report, so it cannot be generalized to a large population.

Patient perspective

The patient was very happy during follow-up due to clinical improvement and improved quality of life.

Conclusion

Inflammatory conditions like panniculitis masquerading as orbital cellulitis can pose a diagnostic dilemma. Our case illustrates that not all presentations that resemble orbital cellulitis are infectious in origin. Non-infectious etiologies, such as panniculitis, should be considered in the context of a patient presenting with subacute symptoms, a poor response to antibiotics, and related systemic inflammatory lesions. Accurate diagnosis and suitable treatment depend on multidisciplinary assessment involving consultation with internal medicine, neurology, ENT, and rheumatology, as well as histopathological assessment.

Ethical approval

Ethical approval not required.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying images, A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Sources of funding

None.

Conflicts of interest disclosure

The authors declare no conflicts of interest.

Author contributions

S.P., S.T., S.J., and S.S. wrote the original manuscript and reviewed and edited it. A.A. and S.J. reviewed and edited the manuscript. All authors have read and agreed to the final version of the manuscript.

Research registration unique identifying number (UIN)

Research registration not required.

Guarantor

Shreeram Paudel.

Provenance and peer review

Double anonymized.

Data availability statement

The data regarding the findings of this case report is available from the corresponding author upon request, subject to patient confidentiality and ethical restrictions.