A 50-year-old non-diabetic male patient from Manikganj presented to Sir Salimullah Medical College Mitford Hospital in 2021 with fever, skin rash, and heaviness of the left upper abdomen for the previous three months. The fever was high grade (Highest recorded temp was 102°F), intermittent in nature, was not associated with chills and rigor, and did not subside by sweating. The patients also complained of substantial weight loss despite having a good appetite.
On query, he gave a previous history of Visceral Leishmaniasis 20 years back and 2 histories of relapse of VL (Reappearance of symptoms and sign of Kalazar within 6 months after treatment)10, one is 10 years, and another is 2 years back. He was treated with Sodium Stibogluconate the first two times and with Amphotericin B the last time. He had no other known illness.
CLINICAL COURSE:
Examination revealed multiple maculopapular non-scaly skin lesions (Fig. 1) and a few erythematous lesions (Fig. 2) on the face, limbs, and back of the chest, ranging from 1–2 centimeters in diameter. Skin rashes developed gradually but remained non-itchy and non-tender,non-ulcerative, with no loss of sensation in those regions.
The left hypochondrium showed the most obvious signs of distention on abdominal examination. Significant splenomegaly (7 cm from the left coastal margin to the right iliac fossa) and hepatomegaly (2 cm from the right coastal margin) were present. Hemodynamically, the patient was stable. Hematological study revealed pancytopenia, including normocytic normochromic anemia (Table 1). Routine biochemistry revealed no abnormality (Table 2). But certain investigations showed specific findings conclusive of active kalazar (Table 3).
Table 1
Hematological study findings:
| Parameters | Findings | Reference |
| Hemoglobin | 9 g/dL | 13–17 g/dL |
| ESR | 130 mm/hr | 0–12 mm/hr |
| White blood cells | 1.53 K/uL | 4–10 K/uL |
| Differential Count: |
| Neutrophil | 44% | 40–80% |
| Lymphocyte | 46% | 20–40% |
| Platelet | 61 K/uL | 150–410 K/uL |
Table 2
Routine investigation findings:
| Parameters | Findings | Reference |
| Urinary Routine Examination | Normal | |
| Serum creatinine | 0.99 mg/dL | 0.70–1.20 mm/dL |
| RBS | 6.35 mmol/L | < 11.1 mmol/L |
| ALT | 29 U/L | 15–61 UL |
| ALP | 120 U/L | 38–126 U/L |
| Albumin | 4.16 g/dL | 3.50–5.20 g/dL |
| USG of whole abdomen | - Grade I prostate enlargement - No abdominal lymphadenopathy - No ascites | |
| Prothrombin time | 16 secs | 12–17 secs |
| Bleeding Time | 3 mins | 2–7 mins |
| Clotting Time | 6 mins | 6–9 mins |
| APTT | 49.4 secs | 28–36 secs |
| Uric Acid | 6.25 mg/ dL | 3.50–7.20 mg/dL |
| Calcium | 10.15 mg/dL | 8.80–10.60 mg/dL |
Table 3
Significant findings in different investigations:
| Parameters | Findings |
| ICT for Kala-azar (Both from serum sample and splenic smear) | Positive |
| Bone Marrow Biopsy | Hyperactive erythropoiesis, active granulopoiesis and megakaryopoiesis. LD body was virtually absent. |
| Splenic Biopsy | LD bodies were present (Fig. 3) |
| Slit Skin Smear | Dense infiltration of histocytes and lymphocytes, many of the histocytes were packed with LD bodies (Fig. 4) |
All investigation reports and clinical examinations indicated the presence of active visceral leishmaniasis and PKDL, ultimately leading to the confirmed diagnosis of Para kala-azar dermal leishmaniasis (Para-KDL).
Multiple cases of relapsing due to immunosuppression have been documented. Yet, despite being a relapsing case, our patient did not have any additional immunocompromised condition or infection (Table 4).
Table 4
Investigation results for any co-infection:
| Parameters | Result |
| HBs Ag | Non-reactive |
| Anti- HCV | Non-reactive |
| HIV 1,2 | Non-reactive |
TREATMENT MODULE:
The patient had both Relapse visceral leishmaniasis and PKDL. The treatment protocol for these two conditions is different. However, there was no specific guideline for managing Para-kala-azar dermal leishmaniasis. So, according to previous case-based data, we followed the following treatment regimen. The patient was first treated with a single dosage of Liposomal Amphotericin B (10mg/kg body weight) for relapse visceral leishmaniasis, followed by Miltefosine (2.5 mg/kg/daily) 50 mg twice a day for 12 weeks for PKDL (Fig. 5).
During a 3-month follow-up, the PKDL had improved, as seen by the disappearance of the skin rashes and the absence of LD bodies in the skin smear from the Slit incision. However, several systemic features, such as splenomegaly and failure to gain weight, were still evident. Thus, the patient was treated again with three doses of Liposomal Amphotericin B (5mg /kg) on alternate days.
One month following the last dose of liposomal amphotericin B, we followed up with the patient again. This time, the patient’s overall clinical condition was Improved. The patient had gained weight. There was no fever or splenomegaly. We followed up with the patient again at 6 months, and there was no fever no splenomegaly, and the patient was feeling of general wellbeing. The patient was definitively cured10. The patient was also in good health during his follow-up after 2 years in 2023.