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24 April 2023: Articles  Ecuador

Jessner’s Lymphocytic Infiltration of the Skin in a 73-Year-Old Woman: A Case Report

Challenging differential diagnosis, Unusual or unexpected effect of treatment, Rare disease

Carmen Elena Sánchez1ABDEF, María del Carmen Cabezas2ADEF*, Silvana Narváez1ABDEF, Henry Jaramillo1ABDEF, Camila Miño ORCID logo23ABDEF

DOI: 10.12659/AJCR.938969

Am J Case Rep 2023; 24:e938969

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Abstract

BACKGROUND: Jessner’s lymphocytic infiltration of the skin (JLIS) is a rare and benign process of unknown cause. This disorder affects both sexes, most commonly in the young adult population. However, the demographic characteristics remain largely unknown, due to limited information. JLIS clinical presentation is heterogeneous; lesions can be indurated papular or erythematous plaques on the upper body. Symptoms are variable, from asymptomatic to pruritus and burning sensation.

CASE REPORT: A female patient aged 73 years presented with 10-day asymptomatic dermatosis on the left malar area after an insect sting. At the beginning, the lesion was clinically classified as an abscess and antibiotic therapy was prescribed with ciprofloxacin 500 mg every 12 hours for 5 days. However, due to the lack of clinical response, a biopsy was performed, which reported a predominantly lymphocytic nodular dermatitis. JLIS diagnosis was confirmed after laboratory and imaging tests. Intralesional infiltrations of triamcinolone (0.5 ml) twice within 15 days and mineral sunscreen 3 times a day were prescribed. After the treatment, the lesion had a complete resolution without recurrence to date.

CONCLUSIONS: The present case reports JLIS in an older woman who presented a complete healing resolution of the lesion without recurrences after triamcinolone intralesional infiltrations and sunscreen protection.

Keywords: Dermatitis, Lymphoproliferative Disorders, Triamcinolone, Male, Humans, Female, Aged, Sunscreening Agents, Skin, Lymphocytes, Pseudolymphoma

Background

Jessner’ s lymphocytic infiltration of the skin (JLIS) was first identified in 1953 [1]. Since then, the disease has been described as a chronic, uncommon, benign process of unknown cause, characterized by the infiltration of T lymphocytes into the skin [1]. JLIS is also known as benign skin infiltration, Jessner-Kanof syndrome, or chronic benign CD4-positive T-cell lymphoproliferative disorder [2]. Although the disorder has been reported to affect mostly the adult population of both sexes equally, the prevalence is unestablished [3]. JLIS is clinically characterized by indurated papular lesions or erythematous plaques, commonly localized on the face, neck, and upper thorax; however, the symptoms are variable [3–5]. Some JLIS manifestations are pruritus, burning sensation, and alteration of aesthetic perception, the latter being a reason for frequent consultation [2,4]. Several therapeutic strategies for JLIS management have been described in the literature. Some successful treatments include drugs such as antimalarials (eg, hydroxychloroquine), thalidomide, topical and intralesional corticosteroids, and sun protection [2–7]. However, there were also reports of spontaneous recovery from JLIS without leaving a scar [3]. The current clinical case describes a patient diagnosed with nodular manifestation of JLIS that was resolved with corticosteroid infiltrations (triamcinolone).

Case Report

On August 18, 2020, a 73-year-old woman with a 10-year clinical history of essential arterial hypertension, hypothyroidism, and dyslipidemia was examined in dermatological consultation. Hypertension was treated with telmisartan 80 mg plus hydrochlorothiazide 12.5 mg orally every day and carvedilol 6.25 mg orally every night since diagnosis. Hypothyroidism was treated with levothyroxine 50 μg orally per day, while dyslipidemia was managed with rosuvastatin 10 mg 3 times a week. The patient’s surgical history reported an appendectomy at age 14, a cholecystectomy at age 45, and a hysterectomy at age 55. In addition, the allergy history of the patient reported an anaphylactic event from an unknown dose of penicillin due to an upper respiratory infection at an unspecified age in childhood. The patient presented with a 10-day dermatosis after an insect sting. During examination, she had an erythematous, violet, solid nodular neoformation of approximately 3 cm in diameter on the left malar area. The rest of the physical examination was normal.

At the beginning, the identified dermatological lesion was clinically classified as an abscess (Figure 1), for which antibiotic therapy with quinolones (ciprofloxacin 500 mg every 12 hours for 5 days) was prescribed. Due to the lack of response to antibiotic treatment, it was decided to perform a 4-mm diameter punch biopsy on August 22, 2020. The histopathological report of the biopsy showed macroscopically a segment of tissue of 5×4×2 mm of surface with a hemorrhagic brown, regular, and blackish base. In the microscopic description, the histological slides showed a skin fragment with a dense and diffuse inflammatory infiltrate that tended to form nodules occupying the upper dermis. The infiltrate did not extend to the epithelium or the skin annexes and was composed of numerous lymphocytes and plasmacytes cells without cell atypia. The overlying epidermis presented basket-weave orthokeratosis, flattening of the crest networks, and hyperpigmentation of the basal layer (Figure 2). The pathology test concluded that the cheek skin biopsy had predominantly lymphocytic nodular dermatitis. However, immunohistochemistry test was not performed on the biopsied tissue. Complementary tests were implemented to confirm the diagnosis on August 25, 2020. Laboratory tests registered a normal blood and metabolic profile. In addition, antinuclear antibodies were negative (less than 1: 80) and a chest radiograph reported a thickening of the bronchial walls in both hemithorax, aortic unfolding, a prominent aortic knob, and dextroscoliosis of the dorsal spine (still under clinical assessment). The abdominal echography performed in the same month reported surgical absence of a gallbladder and mild hepatic steatosis.

The only differential diagnosis considered at the time of the patient’s consultation was lupus erythematosus tumidus (LET) based in other international reports [2]. LET entity has clinical characteristics similar to JLIS, but since LET requires a positive antinuclear antibodies test and a histology reporting abundant mucin deposit, it was not reported in the patient [2,8]. On August 27, 2020, due to the clinical and histological characteristics of the disease presented by the patient, the definitive diagnosis of JLIS was established. JLIS diagnosis permitted us to establish a treatment with intralesional infiltrations of 0.5 ml of triamcinolone (presentation 10 mg in 1 ml) twice within 15 days (September and October 2020). In addition, mineral sunscreen with 50+ sun protection factor was prescribed 3 times a day.

The last control of the patient was conducted on January 24, 2021, confirming complete resolution of the lesion. At the beginning of the treatment, the patient reported post-inflammatory hyperpigmentation, which disappeared over time with sun protection (Figure 3). Currently, the patient is asymptomatic and uses sunscreen every 3 hours, without recurrence of the disease.

Discussion

The present clinical case of JLIS was an older woman, although the literature typically reports different demographic characteristics. Although the etiology of the disease has not been elucidated, this is one of the few reports of JLIS after an insect sting. The characteristics and location of our patient’s lesion was consistent with previously published clinical cases. However, unlike other described patients, the lesion was asymptomatic. The international literature described the performance of complete clinical and laboratory evaluation of patients with presumptive diagnosis of JLIS. We conducted all the currently recommended assessments to meet the diagnostic standards in our patient [4]. Considering other differential diagnosis reports, the only differential diagnosis in our patient was LET [2]. However, this pathology was ruled out in this case by antinuclear antibody titers and the histological report. JLIS does not have an established therapeutic protocol; nonetheless, some treatments are better studied than others. The administration of triamcinolone 0.1% has already been described in an adolescent who presented symptomatic JLIS. Unlike in our case, the administration of the drug in the patient was topical and per day, and after 6 weeks, clinical improvement and disappearance of pruritus were evident [4]. On the other hand, the use of antimalarials such as hydroxychloroquine has presented favorable results in several case reports [2,3,5]. However, in our patient, 0.1% triamcinolone was chosen along with photoprotection due to the size and clinical evolution of the lesion, and the absence of exacerbations.

One of the main limitations in this case report was the lack of immunohistochemical diagnosis due to economic difficulties presented by the patient. In addition, bibliographic support was scarce since most of the studies were isolated case reports and the available local updated information was limited. Finally, JLIS is a low-prevalence disease, and the international literature does not have updated guidelines or protocols for diagnosis and management of this entity.

Conclusions

The present clinical case reaffirms that JLIS is a heterogeneous and benign entity. It also shows that, despite lack of published evidence and guidelines, treatment of JLIS with triamcinolone intralesional infiltrations and sunscreen protection can provide complete healing without recurrences of the lesion.

References:

1.. Orfuss AJ, Lymphocytic infiltration of the skin: AMA Arch Derm Syphilol, 1953; 68; 447-49

2.. Wright K, Henninger C, Lupus erythematosus tumidus vs Jessner’s lymphocytic infiltrate of the skin. A systematic comparison of clinical and histologic characteristics: Residents Reports February, 2012; 42-46

3.. Nieto AI, Infiltración linfocitaria de Jessner-Kanof. Reporte de un caso: Bionatura, 2020; 5; 1250-52 [in Spanish]

4.. Petersen MP, Vestergaard V, Bygum A, Jessner lymphocytic infiltration – rare in childhood: Dermatol Online J, 2017; 23; 3

5.. ben Dhaou B, ben Dahmen F, ben Amor A, Jessner’s lymphocytic infiltration of the skin: A new case: Journal of the Saudi Society of Dermatology & Dermatologic Surgery, 2012; 16; 77-79

6.. Candiz ME, Ardanaz MF, Gómez MA, Infiltración linfocítica de Jessner-Kanof: A propósito de un caso, 1997; 428-30 [in Spanish]

7.. Saenz EM, Sánchez L, Pancorbo J, Placas infiltradas eritematosas faciales recurrentes: Dermatol Peru, 2004; 14; 61-67 [in Spanish]

8.. Rodriguez-Caruncho C, Bielsa I, Lupus erythematosus tumidus: A clinical and histological study of 25 cases: Lupus, 2015; 24; 751-55

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923