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Misdiagnosis of cutaneous leishmaniasis and recurrence after surgical excision
  1. Mark S Bailey1,2 and
  2. G Langman3
  1. 1Department of Infection & Tropical Medicine, Birmingham Heartlands Hospital, Birmingham, UK
  2. 2Department of Military Medicine, Royal Centre for Defence Medicine, Birmingham, UK
  3. 3Department of Histopathology, Birmingham Heartlands Hospital, Birmingham, UK
  1. Correspondence to Lt Col Mark S Bailey RAMC, Department of Infection & Tropical Medicine, Birmingham Heartlands Hospital, Bordesley Green East, Birmingham, England B9 5ST, UK; markbailey{at}nhs.net

Abstract

Cutaneous Leishmaniasis (CL) occurs in British troops deployed to Belize, Afghanistan, Iraq and elsewhere. From 1998 to 2009, 156 (45%) of 343 confirmed cases seen in the UK were in military personnel. CL is a rare disease and requires specialist clinical management because numerous pitfalls exist during diagnosis and treatment. A 19-year-old soldier developed CL on his neck 6 weeks after taking part in jungle warfare training in Belize. However, this was not suspected and the diagnosis was not made from either a skin biopsy or following surgical excision. The travel history and the patient's own photograph prompted retrospective investigations that confirmed this was CL due to Leishmania mexicana. Three months after surgery, the disease recurred locally and was treated appropriately with a good outcome. British military personnel with suspected CL should be referred to the UK Role 4 Military Infectious Diseases & Tropical Medicine Service.

  • Leishmaniasis, Cutaneous
  • Skin Diseases, Infectious
  • Military Personnel
  • Belize

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Key messages

  • Cutaneous leishmaniasis (CL) may present with various skin lesions that develop weeks or months after exposure.

  • Suspected military cases must be referred as per the MedAlert card and Joint Service Publication 950 Leaflet 7-2-5.

  • Histopathology specimens alone are usually insufficient for diagnosis.

  • Surgical excision is unnecessary and ineffective for most forms of CL.

  • Telemedicine could improve the management of military personnel with suspected CL.

Introduction

Skin complaints are responsible for up to 25% of medical consultations by military personnel during overseas deployments.1 A wide range of diseases may be involved2 and they are the commonest cause of referral to the UK Role 4 Military Infectious Diseases & Tropical Medicine Service.3 ,4 Cutaneous Leishmaniasis (CL) is an infection caused by various species of Leishmania protozoa, which are transmitted by the bite of various species of phlebotomine sandflies.5 In recent years, cases have occurred in troops deployed to Belize,6 ,7 Afghanistan,8 ,9 Iraq10 and elsewhere. From 1998 to 2009, 156 (45%) of the 343 confirmed cases seen in the UK were in military personnel.7

CL skin lesions usually develop weeks or months after inoculation and typically form ulcers with raised edges, but may also have a hyperkeratotic or nodular appearance. They are usually non-tender unless a secondary infection occurs and do not respond to anti-bacterial treatment. CL is usually diagnosed from a skin biopsy that is sent for histology and parasitology tests, including PCR tests, to identify the exact species involved.5 The choice of treatment is currently dependent on the site, size and number of lesions, whether there are features of dissemination and the species involved.11 Local treatments such as intralesional injection of sodium stibogluconate (SSG) and cryotherapy may be effective for some lesions,12 but systemic treatments such as intravenous SSG and oral miltefosine are sometimes required.11 For CL cases from Latin America, local treatments are usually sufficient for infections due to species from the Leishmania mexicana complex, whereas systemic treatments are normally used for those due to Leishmania (Viannia) braziliensis complex species because of concerns regarding subsequent mucosal leishmaniasis.13 Overall, CL is a rare disease that requires specialist clinical management because numerous pitfalls exist during diagnosis and treatment.

Case report

A soldier took part in jungle warfare training in Belize and 6 weeks later noticed a small ‘pimple’ on the right side of his neck. By then, he had been posted to Cyprus and the lesion enlarged to form an indurated ulcer that was painless and associated with an enlarged lymph node. The soldier took a photograph of the lesion at this stage with his mobile phone camera (Figure 1). He visited his medical centre in Cyprus and was prescribed topical and oral anti-bacterial treatments, which were of no benefit and so he was referred to a local hospital in Cyprus. A biopsy of the lesion was performed, but no diagnosis was made and he was then referred to the Princess Mary Hospital in Akrotiri where the ulcer and associated lymph node were excised. The tissues removed were sent to an NHS hospital in the UK for histopathology investigations. The skin tissue was reported to show necrosis with multiple granulomata including multinucleate giant cells and although Ziehl–Neelsen (ZN) staining was negative, the findings were said to be highly suspicious for tuberculosis. The lymph node showed reactive follicular hyperplasia with similar granulomata and ZN staining was negative again.

Figure 1

Patient's own mobile phone image of the initial cutaneous leishmaniasis lesion.

The patient was then referred to the Military Infectious Diseases & Tropical Medicine Service at Birmingham Heartlands Hospital (BHH), which was 5 months after the lesion had first appeared. On examination, he had 40 and 120 mm surgical scars on his neck that appeared healthy. However, the travel history and the patient's own photographs were suggestive of CL and so the histopathology specimens were retrieved for review at an infection-histopathology meeting at BHH and for PCR tests to look for Leishmania parasites at the Hospital for Tropical Diseases in London. Repeat examination of the histopathology specimens at 600 times magnification showed obvious Leishmania amastigotes (Figure 2) and the PCR results identified these as L mexicana. The patient was informed of the diagnosis and warned to beware of local recurrence, which can occur with this species.14

Figure 2

Excised skin lesion showing intracellular Leishmania amastigotes (H&E; ×600).

Unfortunately, 2 months later (3 months after surgery), the patient was referred again with inflammation, induration and hyperkeratotic scaling at the antero-inferior margin of the scar where the ulcer had been excised (Figure 3). A punch biopsy was sent for histopathology and parasitology investigations, which confirmed reactivation of CL due to L mexicana. Therefore, the patient was treated with intravenous SSG 20 mg/kg for 20 days on an outpatient basis at BHH. He tolerated this treatment well and his monitoring investigations showed only minor changes in serum amylase and liver transaminases and no significant haematological or electrocardiographic changes. However, he did develop phlebitis associated with the intravenous ‘mid-line’ inserted at the ante-cubital fossa and so this had to be changed once. Two years later, he remained well with no further recurrence and no additional scarring.

Figure 3

Recurrence of cutaneous leishmaniasis due to Leishmania mexicana within the previous surgical scar.

Discussion

This case could have been suspected, referred and diagnosed earlier, which would have saved the patient time off work and surgery. The prolonged incubation period of CL does mean that military patients will sometimes present long after the deployment when they were exposed and possibly during another overseas deployment, which can lead to the disease not being suspected. Therefore, all British military personnel who deploy to leishmaniasis endemic areas should receive a health brief and be issued with a MedAlert card that reminds them (and their medical officers) of the risk of CL occurring up to 2 years later. This card also provides contact details of the Military Infectious Diseases & Tropical Medicine Service at BHH and all suspected cases should be referred in accordance with Joint Service Publication 950 Leaflet 7-2-5,15 which replaces Surgeon General's Policy Letter 09/04. Appropriate referral will avoid problems in the clinical management of patients with CL, which include long delays in diagnosis, inappropriate skin biopsies leading to complications (eg, on the external ear), use of ineffective treatments (eg, fluconazole), both under-dosing and over-use of intravenous SSG (depending on the species involved), early termination of treatment due to only minor adverse effects and a failure to carry out monitoring investigations when necessary.

Histopathology samples can be used to diagnose CL, but the findings vary between different types of CL lesion,16 are non-specific unless amastigotes are seen and are less sensitive than PCR tests overall. Leishmania amastigotes are intracellular organisms, measuring only 2–4 µm in diameter and their identification requires high power magnification. Physical treatments such as cryotherapy and thermotherapy are accepted treatments for many types of CL12 and curettage has also been used in certain countries for CL due to certain species (eg, Pakistan where Leishmania tropica predominates).17 However, the recurrence after surgical excision in this case illustrates why research findings for CL cannot automatically be applied more globally. For the two species present in Belize, L mexicana has a spontaneous cure rate of 88% at 3 months, which falls to 68% at 6 months due to recurrences, while for L braziliensis the equivalent figures are 22% falling to 6%.14 Therefore, this patient had a reasonable chance of not having recurrent disease after surgery and further treatment was not justified in the absence of clinical disease. Intravenous SSG was used subsequently because of the recurrence.

The patient's own photograph (Figure 1) taken with a mobile phone camera would not meet the standard required for a telemedicine consultation. However, the ulcer with raised edges on an exposed body surface was sufficient to prompt retrospective investigations for CL. Military patients often take photographs of their own skin lesions or rashes and the value of these has been reported previously.18 As the quality of digital cameras improves, these images could be increasingly useful so long as patients resist the temptation of getting too close when taking the photograph, which often leads to images that are out of focus. However, a formal military telemedicine service would be preferable for clinical governance reasons and could lead to efficiency improvements in the clinical management of military personnel with suspected CL, especially since many will have an alternative diagnosis, such as a bacterial skin infection or dermatofibroma (unpublished data, MSB).

Overall, this case illustrates why military personnel with suspected CL should be referred to the UK Role 4 Military Infectious Diseases & Tropical Medicine Service at BHH,4 which has unrivalled experience of treating military personnel with this disease.

References

Footnotes

  • Correction notice This article has been corrected since it was published Online First. The word Leishmaniasis in the title has been amended to read leishmaniasis.

  • Contributors MSB managed the case, drafted the manuscript and is the guarantor. GL retrieved and re-examined the histopathology specimens and reviewed the manuscript.

  • Competing interests None.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.