Journal of Medical Cases, ISSN 1923-4155 print, 1923-4163 online, Open Access
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Case Report

Volume 12, Number 4, April 2021, pages 164-171

Pulmonary Adenosquamous Cell Carcinoma With Systemic Lymphadenopathy due to Immunoglobulin G4-Related Disease: A Case Report


Figure 1.
Figure 1. Chest computed tomography (CT) scan showing (a) the primary tumor in the left lower lobe (shown by black arrow) and bilateral sub-pleural reticular shadows in the bilateral lower lobes, (b) the first acute exacerbation of interstitial pneumonia, remarkable shrinkage of (c, d) mediastinal and (e, f) peri-gastric lymphadenopathy (c, e) before steroid therapy (shown by white arrows, in the middle of September) and (d, f) during steroid tapering (in the late October).
Figure 2.
Figure 2. Fludeoxyglucose-positron emission tomography (FDG-PET) scan at the initial staging before steroid therapy showed uptake in (a) the left supraclavicular, (b) mediastinal and (c) peri-gastric lymph nodes, and (d) the primary tumor in the left lower lobe (shown by white arrows).
Figure 3.
Figure 3. Histological findings (hematoxylin and eosin (H&E) stain, × 10) of (a) main component of squamous cell carcinoma and (b) partial component of adenocarcinoma in the primary tumor, (c) interstitial pneumonia (favor for non-specific interstitial pneumonia), (d) lymphoplasmacytic cell infiltration into interstitium of interlobular septal walls, (e) coexistence of epithelioid granulomas and primary squamous cell carcinoma in the left lower lobe, and (f) coexistence of epithelioid granulomas and metastatic lesions in the mediastinal lymph nodes. (e, f) These findings suggest complication of pulmonary tuberculosis and lung cancer.
Figure 4.
Figure 4. Immunohistochemical findings (× 20): (a, b) mediastinal lymph node, (c, d) lung tissue, (a, c) anti-IgG antibody and (b, d) anti-IgG4 antibody. IgG4: immunoglobulin G4.


Table 1. A Review of English or Japanese Case Reports of Coexistence of Lung Cancer and IgG4-Related Disease
Authors (year, country)Agea, sexIgG4-related diseaseCancer
Radiological featuresDiagnostic techniqueAdditional featuresSerum IgG4 (mg/dL)IHC IgG4/IgG ratioHistology (TMN) (treatment)Outcomes (survivalb)
aAge at the time of lung cancer diagnosis. bAfter cancer diagnosis or surgery. IgG: immunoglobulin G; TMN: tumor, metastasis, node; Ad, adenocarcinoma; AdSq: adenosquamous carcinoma; IgE: immunoglobulin E; CRT: chemoradiotherapy; CTGB: computed tomography guided biopsy; F: female; GGO: ground-glass opacity; GPA: granulomatosis with polyangiitis; IHC: immunohistochemistry; IPF: idiopathic pulmonary fibrosis; M: male; ND: not described; NSIP: non-specific interstitial pneumonia; PR3-ANCA: anti-proteinase 3-antineutrophil cytoplasmic antibody; SCLC: small cell lung cancer; SQ: squamous cell carcinoma; VATS: video-assisted thoracoscopic surgery.
Ikari (2010, Japan) [11]67, MIrregular fibrosis, lymphadenopathyLobectomyCastleman’s disease2,280NDSQ (pT1N0M0) (lobectomy)Dead (> 15 months)
Inoue (2014, Japan) [12]78, MGGO, consolidation, lymphadenopathyLobectomy and partial resection (for biopsy)Gastric cancer, autoimmune pancreatitis983NDAd (pT1bN0M0) (lobectomy)No recurrence (18 months)
Tashiro (2016, Japan) [14]72, MA small noduleLobectomyStoriform fibrosis, retroperitoneal fibrosis346> 40%Ad (cT1bN0M0) (lobectomy)No recurrence (5 years)
Gomez-Hernandez (2018, Spain) [10]70, FA mass with mediastinal lymphadenopathyLobectomyPericarditisNDNDAd (pT1aN2M0) (lobectomy)ND
Tokuda (2018, Japan) [16]71, MBilateral lung nodules, hilar and mediastinal lymphadenopathyBronchoscopyNoneNDNDAd (cT3N3M1b) (chemotherapy)Dead (1 year and 5 months)
Abbass (2019, USA) [8]64, MMultiple pulmonary nodulesCTGB, VATSPR3-ANCA(+)GPA153> 10%SCLC (ND) (chemotherapy)Dead (10 months)
Choi (2019, South Korea) [9]66, MA subpleural nodule in reticular and honeycomb fibrosisLobectomyAutoimmune cholangitis, IPF232.4> 40%AdSq (pT2aN2M0) (lobectomy)ND
Terashima (2020, Japan) [15]64, MBilateral pleural effusionPleural biopsyEosinophilia, serum IgE↑2,750> 50%Ad (stage IIIB) (CRT → durvalumab)ND
Ito (2020, Japan) [13]80, MSystemic lymphadenopathyAxillary lymph node biopsyNone137> 40%Ad (cT3N3M0) (CRT → osimertinib)ND
Ours (Japan)75, MA tumor, interstitial pneumonia, and systemic lymphadenopathyLobectomyNSIP, tuberculosis, pneumothorax38554%AdSq (pT2aN2M0) (lobectomy)Dead (24 days)