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\title{Ulcerative Sarcoidosis: An Atypical Cause of Leg Ulcers.}



\author[1]{Kazuki Miyaue}%
\author[2]{Testuya Hoshi}%
\author[3]{Hiroki Isono}%
\affil[1]{HITO Medical Center}%
\affil[2]{Teine Keijinkai Hospital}%
\affil[3]{HITO medal center}%


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  \date{May 20, 2024}


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CASE

An 83-year-old Japanese woman presented with multiple chronic painful
pretibial ulcers on both extremities that first appeared 3 years
earlier, as diagnosed by a dermatologist. She reported generalized edema
and blurred vision, without fever, night sweats, weight loss, or
arthralgia. Four years prior to presentation, she had bilateral hilar
adenopathy, which was diagnosed as an epithelioid cell granuloma through
biopsy. Upon examination, the ulcers displayed asymmetric features with
violaceous borders and necrotic bases, while the surrounding tissues
appeared erythematous and edematous (Figure 1). The patient also
exhibited erythematous plaques on both arms and bilateral uveitis.
Laboratory tests revealed an elevated eosinophil count of 2630 \selectlanguage{greek}μ\selectlanguage{english}L
(reference range: 70-440) and an elevated serum angiotensin-converting
enzyme (ACE) concentration of 25.5 U/L (reference range: 8.3--21.4).
Antinuclear antibody test, antineutrophil cytoplasmic antibody test,
interferon gamma release assays, and serological tests for syphilis
yielded negative results. Deep vein thrombosis, vascular insufficiency
and cardiac abnormalities were ruled out upon ultrasonography. Plaque
biopsy of the right arm revealed perivascular dermatitis with giant
cells. As further evaluation ruled out other possibilities, the patient
was diagnosed with sarcoidosis and initiated on systemic prednisolone
(0.7 mg/kg/day). Subsequently, the prednisolone dose was gradually
tapered to 5 mg/day, leading to complete healing of the ulcers within 12
months (Figure 2).

DISCUSSION

In this case, the ulcers exhibited atypical characteristics. Clinical
signs of atypical ulcers include a necrotic wound bed, a purple border,
surrounding inflammation, an unusual site such as the proximal calf,
asymmetry sites and severe pain\textsuperscript{1}. The differential
diagnosis of atypical ulcers includes external, neoplastic,
vasculopathic, hematologic, infectious, drug-induced, and inflammatory
etiologies, including sarcoidosis.\textsuperscript{1} Atypical ulcers
are differentiated from typical ulcers caused by venous insufficiency,
diabetes, ischemia, or pressure\textsuperscript{1}.

Sarcoidosis is a granulomatous disease affecting multiple organs.
Cutaneous involvement occurs in approximately 20\%--35\% of patients
with sarcoidosis\textsuperscript{2}. Common skin manifestations include
maculopapules, nodules, plaques, infiltrative scars, lupus
perio(specific lesions), and erythema nodosum (nonspecific
lesion)\textsuperscript{2}. Only 4.8\% of patients with skin lesions
develop skin ulcers. The diagnosis of sarcoidosis is established based
on clinical and histopathological findings after excluding other
potential causes. Differential diagnoses for ulcerative sarcoidosis
include necrobiosis lipoidica, atypical mycobacterial infection,
malignancy, and trauma\textsuperscript{2,3}. Although a biopsy of the
ulcerative lesions was not performed due to concerns about delayed skin
healing, we suspected sarcoidosis based on the ulcer features, such as
necrotic ulcers with violaceous rolled borders in the pretibial
erea\textsuperscript{2}. Additionally, the patient's history of hilar
lymphadenopathy, bilateral uveitis, elevated ACE concentration, and
histopathological analysis of the lymph nodes supported the diagnosis.
The patients was finally diagnosed with ulcerative sarcoidosis based on
her rapid response to corticosteroids.

References

1. Nickles MA, Tsoukas MM, Sweiss N, Ennis W, Altman IA. Atypical
ulcers: a stepwise approach for clinicians. Wounds. 2022
Oct;34(10):236-244.

2. Fernandez-Faith E, McDonnell J. Cutaneous sarcoidosis: differential
diagnosis. Clin Dermatol. 2007;25(3):276-287.

3. Wanat KA, Rosenbach M. Cutaneous Sarcoidosis. Clin Chest Med. 2015
Dec;36(4):685-702.\selectlanguage{english}
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