Cutaneous leishmaniasis presenting with painless ulcer on the right forearm:A case report

lNTRODUCTlON

Leishmaniasis is a chronic zoonotic parasitic disease caused byprotozoa infection with the main vector being sandflies[1].There are four main types of leishmaniasis:cutaneous leishmaniasis,cutaneous mucosal leishmaniasis,visceral leishmaniasis and post-kala-azar dermal leishmaniasis.More than 90% of cutaneous leishmaniasis occurs in the Middle East and South America;the rest are found in North Africa,Central Asia and India.It is rare in East Asia(China,Japan,Korea,North Korea,)[2].Cutaneous leishmaniasis occurs in exposed skin areas susceptible to sandfly bites and presents as a papule that gradually expands into an ulcerative plaque.Because cutaneous leishmaniasis is rare in East Asia,it is more likely to be misdiagnosed and underdiagnosed and requires the attention of clinicians.

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CASE PRESENTATlON

Chief complaints

A 36-year-old male presented with a painless ulcer on the right forearm lasting for 2 mo.

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History of present illness

The patient had been working in central Asia for several years.He returned to China after noticing a soybean-sized nodule that gradually increased in size to an ulcer over a period of 2 mo on the right forearm.He was initially diagnosed with a bacterial skin infection,but the treatment of debridement and levofloxacin(0.5 g/d orally for 7 d)was unsuccessful.

History of past illness

The patient was in good health and had no history of other diseases.

Personal and family history

Shandong Provincial Natural Science Foundation,No.ZR2020QH138.

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Sodium gluconate(600 mg)topical injection into the lesions was administered once a week for 3 wk.The ulcer gradually shrank,and the subcutaneous nodules regressed significantly.

Physical examination

A 5.8 cm × 4.0 cm ulcer on the right forearm with black crust in the center and raised borders was observed.After squeezing,a clear,thin liquid secretion appeared.Three hard,peanut-sized subcutaneous nodules could be palpated around the ulcer(Figure 1A and B).No enlargement of lymph nodes was palpable in the right axilla or other areas.

Laboratory examinations

The results from routine tests,such as blood,urine,stool routine,liver and kidney function and bacterial culture,were normal.Histopathological examination revealed diffuse mixed inflammatory cell infiltration within the superficial and deep layers of the dermis.Amastigote ofexisted in the cytoplasm of the histocytes(Figure 2A).Giemsa staining showed the amastigote more clearly(Figure 2B).Leishman-Donovan bodies were seen on scrapings of the skin lesion(Figure 2C),and no Leishman-Donovan bodies were seen on bone marrow smears.Serum immunoglobulin G antibody test ofwas negative.

Imaging examinations

Computed tomography examination of the chest and abdomen showed no abnormalities.

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FlNAL DlAGNOSlS

Combined with the clinical manifestations,histopathological changes and laboratory findings,visceral leishmaniasis could be excluded,and the diagnosis of cutaneous leishmaniasis was established.Since the patient had worked abroad for a long time and China was not aepidemic area,this case was consistent with imported cutaneous leishmaniasis.

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TREATMENT

Finally, Susan decided6 that she was ready to try the trip on her own. Monday morning arrived. Before she left, she embraced her husband tightly. Her eyes filled with tears of gratitude7 for his loyalty8, his patience, and his love. She said good-bye and, for the first time, they went their separate ways. Monday, Tuesday, Wednesday, Thursday... Each day on her own went perfectly9, and a wild gaiety（） took hold of Susan. She was doing it! She was going to work all by herself!

OUTCOME AND FOLLOW-UP

Treatment selection for cutaneous leishmaniasis should be based on patients’ personal characteristics[10].Some lesions may heal on their own or by local antimonial therapy within several months.For lesions with a diameter larger than 5 cm with local enlarged lymph nodes or that involve the face,auricle or genital area,the systematic antimonial treatment is needed[11,12].Other drugs,such as amphotericin B,miltefosine,pentamidine and fluconazole,could be an option for patients who are intolerant to antimonials due to the adverse side effects[13,14].In the future,it is expected that leishmaniasis will be radically cured by vaccination[15].The lesion of our patient was solitary and confined to the forearm.Therefore,the intralesional injection of antimonials was provided,and the patient recovered well.

DlSCUSSlON

Cutaneous leishmaniasis,also known as oriental sore,occurs in the Middle East,Central Asia,India,North Africa and European countries along the Mediterranean Sea.Cases in non-endemic countries usually have a history of travel to the epidemic areas[2].A variety ofspecies can cause cutaneous leishmaniasis,including,,,andin South America and,andin Asia,Africa and Europe[1].The difference in virulence between different species is mainly due to the difference in lipophosphate glycans on the surface ofprotozoa,which play an important role in the infection of the sandfly and the invasion of human macrophages[3].

The authors declare no conflicts of interest.

The diagnosis of cutaneous leishmaniasis relies on laboratory tests.In hematoxylin and eosin sections and smears of secretions,the use of Giemsa stain clearly shows the absence of amastigote and is an important basis for confirming the diagnosis[5].Polymerase chain reaction(PCR)assays,especially sequencing of PCR products,are commonly used to determine the specific infecting species.protozoa belong to the kinetoplastida,whose kinetoplast DNA K13A and the transcribed spacer regionwithin the ribosomal DNA are highly conserved within species and differ between species[6,7].Therefore,primers are often designed in this region for PCR amplification and sequencing(Figure 4).Due to the large variation in specificity and sensitivity,serological diagnostic methods have some application in visceral leishmaniasis but are rarely used for the diagnosis of cutaneous leishmaniasis[8].In addition,some scholars are investigating the feasibility of worm antigens and specific proteins in the saliva of the vector whitefly as indicators of infection,and mouse animal models have demonstrated that host interleukin-10 levels correlate with disease severity[9].

In our case,the patient had a history of living abroad in central Asia,and the pathogenic examination revealedinfection.We excluded the mucosa and visceral involvement,so the final diagnosis of cutaneous leishmaniasis was made.

After treatment with antimony sodium gluconate,the lesion completely faded and left a scar.No recurrence was observed after 1.5 years of follow-up(Figure 3).

CONCLUSlON

The authors have obtained the patient consent form.In the form,the patient gave his consent for his images and other clinical information to be reported in the article.The patient understood that his names and initials will not be published and due efforts will be made to conceal the identity of the patient,although anonymity cannot be guaranteed.

FOOTNOTES

Tu P conceived the idea and designed the report;Zhuang L and Su J collected the patient’s clinical data;Zhuang L wrote the paper;all authors reviewed the results and approved the final version of the manuscript.

The patient had been working in central Asia for 3 years.Family history was unremarkable.

Cutaneous leishmaniasis in non-epidemic countries tends to be misdiagnosed as deep fungal infection,pyoderma gangrenosum,basal cell carcinoma or squamous cell carcinoma,which also present as ulcerative lesions and proliferative plaques.If a patient has a travel history to epidemic countries and skin lesions exist on the exposed skin areas,then leishmaniasis should be considered.Cutaneous leishmaniasis is somewhat self-healing,and treatment plans need to be tailored to the condition.

The lesions of cutaneous leishmaniasis occur on exposed areas,such as the face,neck and extremities,that are susceptible to sandfly bites.In some patients with immunodeficiency,the lesions may disseminate or distribute along lymphatic vessels[4].In most cutaneous leishmaniasis cases,the lesions may heal spontaneously with scarring within a few months,and some patients develop chronic or disseminated infections.It is important to note that leishmaniasis from South American species has a more diverse clinical presentation,is prone to disseminated and diffuse forms,and has a slightly worse prognosis than leishmaniasis from Asian,African and European species[2].

The authors have read the CARE Checklist(2016),and the manuscript was prepared and revised according to the CARE Checklist(2016)

This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers.It is distributed in accordance with the Creative Commons Attribution NonCommercial(CC BYNC 4.0)license,which permits others to distribute,remix,adapt,build upon this work non-commercially,and license their derivative works on different terms,provided the original work is properly cited and the use is noncommercial.See:https://creativecommons.org/Licenses/by-nc/4.0/

China

Le Zhuang 0000-0002-2030-4533;Jing Su 0000-0002-3946-9186;Ping Tu 0000-0002-5189-4003.

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Liu JH

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