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CLINICAL IMAGE
Year : 2020  |  Volume : 152  |  Issue : 7  |  Page : 167-168

A unique case of overlap syndrome with strange immunological findings


Department of Immunopathology, Postgraduate Institute of Medical Education & Research, Chandigarh 160 012, India

Date of Submission20-Nov-2019
Date of Web Publication25-May-2021

Correspondence Address:
Yashwant Kumar
Department of Immunopathology, Postgraduate Institute of Medical Education & Research, Chandigarh 160 012
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmr.IJMR_2260_19

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How to cite this article:
Kumar Y, Das P. A unique case of overlap syndrome with strange immunological findings. Indian J Med Res 2020;152, Suppl S1:167-8

How to cite this URL:
Kumar Y, Das P. A unique case of overlap syndrome with strange immunological findings. Indian J Med Res [serial online] 2020 [cited 2021 Aug 4];152, Suppl S1:167-8. Available from: https://www.ijmr.org.in/text.asp?2020/152/7/167/316800

Patient's consent obtained to publish clinical information and images.


A 46 yr old female presented to the outpatient department of Postgraduate Institute of Medical Education & Research, Chandigarh, India, in July 2019, with weakness and arthralgia for a duration of 2-3 months. Her peripheral blood sample was received in the department of Immunopathology for antinuclear antibody (ANA) testing. Indirect immunofluorescence ANA test (IIF-ANA) on human epithelial type 2 (HEp-2) cells showed a peculiar pattern in the form of haphazardly dispersed granules in both nucleus and cytoplasm [Figure 1]A and forming a zipper pattern within the mitosis (inset). IIF-ANA on composite section (rat liver, kidney tubules and gastric mucosa) showed classical anti-mitochondrial antibody (AMA) pattern in the form of granular positivity in the above tissues ([Figure 1]B, [Figure 1]C, [Figure 1]D, respectively). ANA and liver immunoblot thereafter were performed and found to be positive for CENP, AMA-M2 [Figure 1]E and [Figure 1]F and SP100 autoantibodies [Figure 1]F. The patient also had raised serum IgG, weak-positive p-ANCA and positive AMA-M2 on ELISA (110.0 U), whereas liver-kidney microsomes (LKM) and soluble liver antigen (SLA) were negative. On the basis of these immunoassay findings, the patient was given corticosteroid and ursodeoxycholic acid combination therapy. At three months follow up she remained asymptomatic.
Figure 1: (A) Indirect immunofluorescence antinuclear antibody (IIF-ANA) test on Hep-2 images showing a peculiar pattern in the form of haphazardly dispersed granules in both nucleus and cytoplasm and forming a zipper pattern within the mitosis (inset). (B-D) IIF-ANA test on composite section (rat liver, kidney tubules and gastric mucosa) showed classical anti-mitochondrial pattern in the form of granular positivity. (E) Antinuclear antibody and liver immunoblot are positive for CENPB (Centromere protein B), anti-mitochondrial-M2 (E and F, white arrow; red arrows are control bands) and (F) SP100 autoantibodies.

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The above findings led to revised histopathological diagnosis of autoimmune hepatitis to overlap syndrome [autoimmune hepatitis - primary sclerosing cholangitis (AIH-PSC)]. This case is an example where a careful analysis of IIF-ANA on Hep-2 cells helped in correct diagnosis of the patient.

Conflicts of Interest: None.


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