CASE REPORT
Nagoya J. Med. Sci. 85. 123–126, 2023
doi:10.18999/nagjms.85.1.123

Prominent dermal accumulation of Russell bodies underlying
pseudocarcinomatous hyperplasia with fungal infection
Tatsuhiro Noda1, Norika Akashi1, Maiko Shimomura1, Haruka Koizumi1,
Miyuki Mizuta1, Kosei Nakajima2, Takuya Takeichi3,
Teruyuki Mitsuma1 and Masashi Akiyama3
Department of Dermatology, Ichinomiya Municipal Hospital, Ichinomiya, Japan2
Department of Pathology, Ichinomiya Municipal Hospital, Ichinomiya, Japan
Department of Dermatology, Nagoya University Graduate School of Medicine, Nagoya, Japan
13ABSTRACT
Blockade of the secretion of immunoglobulins leads to their accumulation in plasma cells, resulting incondensed immunoglobulins in the rough endoplasmic reticulum of plasma cells, termed Russell bodies.
They are sometimes found in lymphoplasmacellular inflammation of the intestinal mucosa and in lymphoidcell malignancies, but only very rarely in skin diseases. Here, we report an 86-year-old female who
presented with a lesion with the prominent accumulation of Russell bodies underlying pseudocarcinomatous
hyperplasia with fungal infection in the face. Immunohistochemical staining showed the cells containing
Russell bodies to be positive for CD138 and the Russell bodies to be positive for immunoglobulin k andl light chains. The present case suggests that when inflammatory cell infiltration with abundant roundintracellular eosinophilic materials is observed in the dermis, the dermal accumulation of Russell bodies
should be considered in cases with reactive pseudocarcinomatous hyperplasia with fungal infection.
Keywords: fungus, immunoglobulin, plasma cell, Russell bodyThis is an Open Access article distributed under the Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International
License. To view the details of this license, please visit (http://creativecommons.org/licenses/by-nc-nd/4.0/).

INTRODUCTION
When immunoglobulin secretion becomes blockaded, immunoglobulins can accumulate inplasma cells, resulting in condensed immunoglobulins in the rough endoplasmic reticulum ofplasma cells. These are called Russell bodies, and while they are sometimes found in lymphoplasmacellular inflammation in the intestinal mucosa and lymphoid cell malignancies, they
are only very rarely found in skin diseases.1,2 Here, we report a lesion of pseudocarcinomatous
hyperplasia with superficial fungal infection in the face, showing prominent intracellular andextracellular Russell bodies in the underlying dermis.

Received: January 12, 2022; accepted: February 10, 2022
Corresponding Author: Masashi Akiyama, MD, PhD
Department of Dermatology, Nagoya University Graduate School of Medicine,
65 Tsurumai-cho, Showa-ku, Nagoya 466-8550, Japan
Tel: +81-52-744-2314, Fax: +81-52-744-2318, E-mail: makiyama@med.nagoya-u.ac.jp
123Tatsuhiro Noda et al

CASE PRESENTATION
An 86-year-old woman presented with a 3-month history of scaly erythema on the face.
Physical examination showed a reddish, irregularly shaped plaque with scales on the right cheek(Figure 1A). Potassium hydroxide examination of the scales revealed numerous hyphae. She had

Fig. 1 The clinical appearance and histopathology of hyperkeratotic erythema on the patient’s faceFig. 1A: Scaling erythema is seen on the right cheek.
Fig. 1B: The hyperkeratotic erythema improved significantly after topical treatment with an antifungal agent.
Fig. 1C: Microscopy reveals acanthosis and hyperkeratosis with parakeratosis in the epidermis. Inflammatory
cell infiltration with large numbers of round eosinophilic bodies is seen in the superficial and mid
dermis. The areas bordered by the red rectangle and the yellow rectangle are enlarged in (D) and (E),respectively (hematoxylin and eosin staining, scale bar: 200 μm).
Fig. 1D: Pseudocarcinomatous hyperplasia is seen mainly in the basal layer (hematoxylin and eosin staining,
scale bar: 100 μm).
Fig. 1E: Intracellular eosinophilic bodies are seen predominantly in the inflammatory cell infiltration in the dermis(hematoxylin and eosin staining, scale bar: 50 μm).
Fig. 1F: Cells containing the eosinophilic bodies are positive for anti-CD138 antibodies and are thought to beplasma cells (anti-CD138 staining, scale bar: 50 μm).
Fig. 1G–1H: The round eosinophilic bodies are positive for immunoglobulin k light chains (G) and immunoglobulin l light chains (H), indicating that the eosinophilic bodies are Russell bodies (G, anti-klight chains; H, anti-l light chains: scale bars: 50 μm).
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Dermal Russell bodies

been under hemodialysis due to renal failure. She had no history of blood disorders. A skinbiopsy histopathologically showed irregular acanthosis, hyperkeratosis, and parakeratosis in thelesional epidermis, and basal cells in the epidermis showed enlarged, irregular, hyperchromatic
nuclei (Figure 1C and D). There were no dermal granulomatous lesions such as those seen inMajocchi granulomas. From these findings, pseudocarcinomatous hyperplasia with superficial
fungal infection was diagnosed. In addition, inflammatory cell infiltration, along with abundantround eosinophilic materials, was apparent in the dermis (Figure 1C and E). Immunohistochemical staining revealed the cells with cytoplasmic eosinophilic materials to be positive for CD138(Figure 1F) and the round eosinophilic bodies to be positive for immunoglobulin k and llight chains (Figure 1G and H). From these findings, the prominent eosinophilic bodies in theunderlying dermis were considered to be Russell bodies. The infiltrating cells with cytoplasmic
eosinophilic bodies were considered to be polyclonal plasma cells with conspicuous Russellbodies. The plasma cells showed neither nuclear atypia nor mitosis. Blood tests ruled out syphilis
and multiple myeloma. We treated the fungal infection with a topical antifungal agent, and thelesion improved significantly (Figure 1B).

DISCUSSION
Gastritis with the infiltration of Russell body-containing plasma cells in the gastric mucosa
is known as Russell body gastritis.3 A number of cases of Russell body gastritis associatedwith Helicobacter pylori infection have been reported.4 A chronic bacterial infection can be theantigenic stimulus for the formation or accumulation of Russell bodies in the gastric mucosa.4
Concerning skin diseases, a case with actinic keratosis showing conspicuous Russell bodyaccumulation in the dermis was reported.5 In that case, no fungal infection was described in theactinic keratosis lesion.5 In the present patient, the skin lesion was successfully treated with a
topical antifungal agent, and the lesion largely disappeared in 3 months. Thus, we consider that
actinic keratosis can be ruled out in the present case. In addition, a patient with chronic syphilis
was reported to show dermal Russell body accumulation in a phimosis lesion.2
To our knowledge, the present patient is the first reported case of prominent dermal Russel
body accumulation associated with pseudocarcinomatous hyperplasia with superficial fungal
infection. In the present case, the patient had a facial lesion with the prominent accumulationof Russell bodies in the dermis beneath pseudocarcinomatous hyperplasia with superficial fungalinfection. We speculate that the Russell bodies could have been produced by the chronic antigenic
stimulus from the superficial fungal infection, similarly to the Russell body production in Russell
body gastritis associated with Helicobacter pylori infection. We searched the literature for reportsof patients with cutaneous superficial fungal infection and Russell body accumulation. However,no such reports were found. In addition, in the present skin lesion, no granulomatous lesions
caused by the fungal infection were observed in the dermis. Thus, we were unable to obtain
direct evidence of any association between the superficial fungal infection and the Russell body
production. Furthermore, we speculate that inflammatory reactions to the pseudocarcinomatous
hyperplasia in the lesional epidermis might be associated with the Russell body production. We
have to accumulate more cases with cutaneous Russell body accumulation and to study their skinlesions in detail to elucidate the exact mechanisms of Russell body accumulation in the skin.
Eosinophilic bodies morphologically similar to Russell bodies include amyloids, colloid bodies,eosinophilic bodies seen in cryptococcosis and other fungal infections, and Michaelis–Gutmannbodies found in histiocytes in malakoplakia. These similar structures can be ruled out by specificstains.5
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CONCLUSION
The present case suggests that when inflammatory cell infiltration with abundant roundintracellular eosinophilic materials is observed in the dermis, the dermal accumulation of Russell
bodies should be considered in cases with pseudocarcinomatous hyperplasia with superficial
fungal infection. The further accumulation of similar cases is needed to elucidate the molecularmechanisms behind Russel body formation associated with pseudocarcinomatous hyperplasia withcutaneous superficial fungal infection.

FUNDING SOURCES
None.

CONFLICTS OF INTEREST
The authors have no conflicts of interest to declare.

REFERENCES
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