LGCmain
Indian Journal of Pathology and Microbiology
Home About us Instructions Submission Subscribe Advertise Contact e-Alerts Ahead Of Print Login 
Users Online: 2560
Print this page  Email this page Bookmark this page Small font sizeDefault font sizeIncrease font size


 
  Table of Contents    
LETTERS TO EDITOR  
Year : 2018  |  Volume : 61  |  Issue : 2  |  Page : 298-299
Fluorodeoxyglucose avid malakoplakia of the laryngohypopharynx masquerading as malignant tumor: A pathological enigma and clinical dilemma


1 Department of Pathology, Artemis Hospitals, Gurugram, Haryana, India
2 Department of Pathology, University of Pittsburgh Medical Center, Pittsburgh, PA, USA
3 Department of Nuclear Medicine, Artemis Hospitals, Gurugram, Haryana, India

Click here for correspondence address and email

Date of Web Publication20-Apr-2018
 

How to cite this article:
Goel D, Pradhan D, Tiwary A. Fluorodeoxyglucose avid malakoplakia of the laryngohypopharynx masquerading as malignant tumor: A pathological enigma and clinical dilemma. Indian J Pathol Microbiol 2018;61:298-9

How to cite this URL:
Goel D, Pradhan D, Tiwary A. Fluorodeoxyglucose avid malakoplakia of the laryngohypopharynx masquerading as malignant tumor: A pathological enigma and clinical dilemma. Indian J Pathol Microbiol [serial online] 2018 [cited 2020 Apr 2];61:298-9. Available from: http://www.ijpmonline.org/text.asp?2018/61/2/298/230534




Editor,

Malakoplakia of the laryngohypopharynx is extremely rare. We report an unusual case of malakoplakia involving the laryngohypopharynx in a 60-year-old HIV negative Nigerian female, who presented with dysphagia toward solids and hot potato voice for 6 months. On direct laryngoscopy, polypoidal growth was seen in the epiglottic region involving the vallecula. Base of the tongue was unremarkable. She had a biopsy report with a diagnosis of squamous papilloma. In view of the clinical suspicion of malignancy, a positron emission tomography-computed tomography (PET-CT) was performed which revealed a large irregular fluorodeoxyglucose (FDG) avid (SUVmax: 14.1) heterogeneously enhancing ulceroproliferative growth measuring approximately 2.0(AP) cm × 2.2(TS) cm × 3.4(CC) cm, involving the epiglottis and bilateral lateral and median glossoepiglottic folds superiorly with effacement of the valleculae, and inferiorly involving bilateral aryepiglottic folds with effacement of the left pyriform sinus [Figure 1]a. The glottis and its associated cartilages were normal. The overlying hyoid bone appeared normal. There was no significant FDG avid cervical or supraclavicular lymphadenopathy. A biopsy was performed on endoscopy which revealed a cricopharyngeal mass [Figure 1]b. Clinical diagnosis of squamous cell carcinoma was considered. Biopsy was superficial and revealed hyperplastic stratified squamous epithelium. The subepithelium had diffuse sheets of foamy granular cells admixed with a few inflammatory cells. Some of these cells showed engulfed bacteria and a few concentric calcified bodies were noted. Differentials considered were verruciform xanthoma, malakoplakia, granular cell tumor, or involvement by Whipple's disease. Malignancy was ruled out. Subsequently, complete excision of the mass was performed.
Figure 1: (a) Positron emission tomography-computed tomography reveals a large irregular fluorodeoxyglucose avid (SUVmax: 14.1) heterogeneously enhancing growth measuring approximately 2.0(AP) cm × 2.2(TS) cm × 3.4(CC) cm. (b) Upper gastrointestinal endoscopy revealed an ulceroproliferative cricopharyngeal mass. (c) Photomicrograph revealing sheets of macrophages with eosinophilic granular cytoplasm and numerous intracytoplasmic concentrically laminated calcified Michaelis– Gutmann bodies (arrow) (H and E, ×40)

Click here to view


Microscopy showed polypoidal tissue fragments lined by stratified squamous epithelium with a few intraepithelial neutrophils. No koilocytosis or dysplasia was noted. The subepithelial tissue demonstrated diffuse sheets of macrophages with eosinophilic granular cytoplasm and numerous intracytoplasmic concentrically laminated calcified Michaelis–Gutmann (MG) bodies [Figure 1]c. Numerous bacteria were seen in the histiocytes [Figure 2]a. A few plasma cells and neutrophils were also admixed. There was no evidence of malignancy. Periodic acid-Schiff, Perl's and Von Kossa stains highlighted the MG bodies [Figure 2]b, [Figure 2]c, [Figure 2]d. CD68 was strongly positive in granular cells. S100 was focally positive. Final diagnosis of malakoplakia was offered. Patient completed her antibiotic regimen. After 6 months of follow-up, the patient was asymptomatic and doing well.
Figure 2: (a) Numerous bacteria (arrow) are identified scattered in the sheets of macrophages (H and E, ×40). (b) Magenta-colored PAS-positive (arrows) Michaelis–Gutmann bodies (PAS, ×40). (c) Perl's Prussian blue staining of Michaelis–Gutmann bodies (Perl's, ×40). (d) Von Kossa stain highlighted the Michaelis–Gutmann bodies (arrow) (Von Kossa stain, ×40)

Click here to view


Malakoplakia is a chronic inflammatory disease of unknown etiology, described initially in the bladder by Michaelis and Gutmann in 1902.[1] Later, Von Hansemann coined the term “malakoplakia”. The first description of malakoplakia outside the urinary tract was in 1958.[2] Gastrointestinal tract is the second most common site. On review of literature, we found only three case reports involving larynx and none involving hypopharynx.[3] Malakoplakia has been reported in other sites as well, namely, lung, bone, trachea, skin, lymph nodes, and tongue.

The age at presentation ranges from 6 weeks to 85 years in various studies.[1] The clinical appearance varies from silent nodules to large masses mimicking cancer. It may present with vaginal bleeding, diarrhea, abdominal pain, hemorrhage, obstruction, and stridor depending on the organ involved. Our case presented with dysphagia and hot potato voice due to its location in the laryngopharynx. Nowadays, PET-CT evaluation is done in all suspected malignancies for staging purposes. Our case, for the first time, has highlighted high FDG avidity in this lesion making it a potential pitfall for malignancy. Various conditions can be associated with malakoplakia including acquired immunodeficiency syndrome (AIDS), malignancy, tuberculosis, uncontrolled diabetes, and organ transplantation.[4],[5] However, in our patient, none of these were present.

Morphologically, it should be differentiated from Whipple's disease, tuberculosis, xanthogranulomatous inflammation, and histiocytic storage diseases which can also show proliferation of macrophages. The hallmark of malakoplakia is MG bodies. In our case, numerous MG bodies were noted. These are targetoid structures which result from the initial mineralization of the matrix cores and peripherally arranged continuously accumulated phospholipids and microvesicles that represent incompletely digested debris. MG bodies can be intracellular or extracellular and are visualized by Von Kossa stain for calcium or Prussian blue stain for iron.

Several organisms have been implicated in pathogenesis, particularly  Escherichia More Details coli, Mycobacterium tuberculosis, Proteus, and Staphylococcus aureus. Coliform bacteria are seen in patients with AIDS.[3] Our case showed numerous bacteria in the histiocytes; however, unfortunately, tissue was not submitted for microbiological examination. Antibiotics are an effective treatment of malakoplakia. However, the outcomes reported are variable. Some of the reported cases showed disease recurrence or progression causing life-threatening complications.[5]

To conclude, malakoplakia of laryngohypopharynx is extremely rare. A large rapidly growing mass of malakoplakia can be misinterpreted as malignancy. A high index of suspicion is needed not only by pathologists but also by radiologists and surgeons so as to avoid any unnecessary radical surgical intervention. Pathologists should be aware that malakoplakia can coexist with malignancy and tuberculosis so thorough sampling of the tumor mass is needed. Early diagnosis and adequate treatment prevents disease recurrence.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Yousef GM, Naghibi B, Hamodat MM. Malakoplakia outside the urinary tract. Arch Pathol Lab Med 2007;131:297-300.  Back to cited text no. 1
    
2.
Alvarez Gómez GJ, Marín Botero ML, Henao Calle CA, Duque Serna FL. Malacoplakia: Case report in tongue and review of the literature. Med Oral Patol Oral Cir Bucal 2008;13:E352-4.  Back to cited text no. 2
    
3.
Akilesh S, Cross S, Kimmelshue K, Kirmani N, Dehner LP, El-Mofty SK, et al. Pseudotumor of the tracheal-laryngeal junction with unusual morphologic features caused by Rhodococcus equi infection. Head Neck Pathol 2011;5:395-400.  Back to cited text no. 3
    
4.
Dong H, Dawes S, Philip J, Chaudhri S, Subramonian K. Malakoplakia of the urogenital tract. Urol Case Rep 2015;3:6-8.  Back to cited text no. 4
    
5.
Wong SH, Yeung VH, Lee YK, Chan MT, Cheeng CH, Chu PS, et al. Malakoplakia of the urinary tract: A benign disease with a possible malignant outcome. J Case Rep 2016;6:254-8.  Back to cited text no. 5
    

Top
Correspondence Address:
Deepa Goel
Department of Pathology, Artemis Hospitals, Sector -34, Gurugram, Haryana
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_156_17

Rights and Permissions


    Figures

  [Figure 1], [Figure 2]



 

Top
 
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
    Email Alert *
    Add to My List *
* Registration required (free)  


    References
    Article Figures

 Article Access Statistics
    Viewed1140    
    Printed15    
    Emailed0    
    PDF Downloaded42    
    Comments [Add]    

Recommend this journal