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CASE REPORT  
Year : 2018  |  Volume : 11  |  Issue : 3  |  Page : 100-102
Does this sarcoidosis case triggered by typhoid fever? A rare case report


1 Universiti Tunku Abdul Rahman, FMHS, Selangor, Malaysia
2 Baghdad University, College of Medicine, Baghdad, Iraq

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Date of Web Publication10-Dec-2019
 

   Abstract 


Sarcoidosis is a multisystemic disease that characterized by the presence of nonspecific noncaseating granuloma of undetermined etiology. Its peak incidence was found in patients ᠆ years old. Globally, the disease is more common among Scandinavians and African Americans. In Asia, it is rare and the highest incidence was found in Japan and India. In Iraq, sarcoidosis is a rare disease, but there are no accurate records of its incidence. Mycobacterium spp. and Propionibacterium acnes are the most common triggering infectious agent, and corticosteroids are the standard treatment of choice for symptomatic patients for not ɣ year. In particular, this report presents a rare case of sarcoidosis that developed in an elderly Iraqi female with an unusual presentation. In addition, it has been heralded exceptionally by an acute typhoid fever with a possible correlation. She treated effectively with prednisolone for only 2 months without any complaint for 1½-year duration.

Keywords: Iraq, noncaseating granuloma, sarcoidosis, typhoid fever

How to cite this article:
Yassin WA, Abed NF, Ghafour KH, Jabbar MA. Does this sarcoidosis case triggered by typhoid fever? A rare case report. Ann Trop Med Public Health 2018;11:100-2

How to cite this URL:
Yassin WA, Abed NF, Ghafour KH, Jabbar MA. Does this sarcoidosis case triggered by typhoid fever? A rare case report. Ann Trop Med Public Health [serial online] 2018 [cited 2020 Aug 10];11:100-2. Available from: http://www.atmph.org/text.asp?2018/11/3/100/272546



   Introduction Top


Sarcoidosis is a chronic multisystemic inflammatory condition of undetermined etiology, commonly affecting young adults (20–40 years).[1] It is characterized by the presence of noncaseating granuloma in the affected organs, which most commonly involve lungs, eyes, and skin.[2] Higher incidences have been reported among Northern Europeans and African Americans.[3] It is rare in Asia with highest rates found in Japan[3] and India.[4] The current case is interesting because sarcoidosis has been reported to be very rare among Arabians.[5] Furthermore, the patient presented with an unusual presentation, in an uncommon geographic area, probably correlated with typhoid fever and successfully treated for a limited duration.


   Case Report Top


A 69-year-old Iraqi woman, presented in July 2014 complaining of fever and loss of appetite for 3 weeks, associated with night sweating, easy fatigability, nausea, and vomiting with vague abdominal pain, but no respiratory complaints. She had a controlled diabetes mellitus for 5 years. On clinical examination, the patient looked ill, pale and feverish with 39°C body temperature. Her pulse rate was 77 beats / min. She had normal breathing sounds, normal heart sounds with soft abdomen. No hepatomegaly or splenomegaly was detected. There was no axillary or inguinal lymphadenopathy. Her laboratory investigations showed; hemoglobin (11.8 g/L), white blood cell (4000 × 109/L), erythrocyte sedimentation rate (ESR) (50 mm/h), C-reactive protein (CRP) was increased, fasting blood sugar (120 mg/dl), Widal test; Salmonella typhi O (1/320) and S. typhi H (1/160). All S. paratyphi groups were found negative, but blood culture for S. typhi was found positive. Rose Bengal test was negative with normal renal and liver function tests. She was diagnosed as typhoid fever and treated with ciprofloxacin 500 mg twice daily for a total of 14 days. Five months later, the patient presented again and claimed persistence of her previous symptoms but less severe. She had a mild nocturnal fever and sweating with significant loss of appetite. Her body weight was progressively dropped from 93 kg to 58 kg during her whole complaint period. In addition, she developed dyspnea, fatigability, generalized aches with burning sensation, and red discoloration of her tongue [Figure 1]. Clinically, she looked ill, cachexic, and pale but afebrile. She had impalpable axillary and inguinal lymph nodes (LNs) with normal thyroid and breast examinations. There was poor air entry in chest examination with mild upper abdominal tenderness and splenomegaly. She was sent for new investigations and the results revealed anemia, elevated ESR and CRP. There was reduction of both of S. typhi O and H titers to 1/80. Antinuclear Antibody Test, Tuberculin Test and Rose Bengal Test were all negative. Imaging tests showed hilar lymphadenopathy with interstitial infiltrates by chest X-ray [Figure 2] and splenomegaly and para-aortic lymphadenopathy by both abdominal ultrasound and computed tomography scan [Figure 3]a and [Figure 3]b. Later, after taking an informed consent, the patient was sent for excisional biopsy. During the surgery, an excisional biopsy of the two most enlarged para-aortic lymph nodes were taken. In addition, there were hyper-pigmented patches found on the liver where biopsy was also taken. All biopsies were sent for histopathological analysis. The histopathological result showed numerous noncaseating granulomas and multiple multinucleated giant cells with many asteroid bodies in all biopsies and suggested as sarcoidosis [Figure 4]a, [Figure 4]b, [Figure 4]c. Then, on further evaluation, patient's serum Ca++ was 12.0 mg/dl, urinary Ca++ was 290 mg/24 h, and serum angiotensin-converting enzyme was 26.4 U/L. Treatment was started immediately with prednisolone tablet orally started with 40 mg daily in divided doses and tapered course for 2 months duration. The patient showed prompt and dramatic improvement of clinical as well as laboratory investigations that carried out on 3 and 6 months later (data not shown). Early during steroid treatment, the patient retained her full daily activities and gained weight gradually. After few months, her body weight fixed on 76 kg and no any complaint for 1½-year follow-up.
Figure 1: Patient's tongue shows red discoloration

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Figure 2: Chest X-ray shows hilar lymphadenopathy with interstitial infiltrates

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Figure 3: Abdominal computed tomography scan shows (a) Homogenous splenomegaly (b) Para-aortic lymphadenopathy

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Figure 4: Histopathological result. (a) Para-aortic lymph node section. It shows loss of its texture (effacement of lymph node architecture) with multiple noncaseating granulomas. There is plenty of lymphocytes (white arrows) and multinucleated giant cells (blue arrow) (b) Liver section. It shows normal hepatocytes arranged in cord-like pattern (green arrow), multiple noncaseating granulomas (white arrow), and multinucleated giant cell (blue arrow) (c) It shows an asteroid body inside a multinucleated giant cell (blue arrow)

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   Discussion Top


Sarcoidosis developed when a genetically susceptible person exposed to an environmental agent, either chemical or micro-organism.[6]Mycobacterium spp. and Propionibacterium acnes are the most commonly implicated infectious agent[7],[8] while the current case was preceded by typhoid fever. At the best of our knowledge, this is the first case of sarcoidosis which has a possible association with typhoid fever. The patient was treated with ciprofloxacin for 2 weeks, and all her complaints were addressed to be improved but not cured. After 5 months, she presented again with same but mild, nonspecific symptoms with significant weight loss. At that time, our differential diagnosis was chronic typhoid fever, tuberculosis, brucellosis, and malignancy. Both tuberculosis and brucellosis were excluded by investigations. Chronic typhoid fever was still possible, but there were pulmonary involvement, considerable loss of weight, and the presence of hilar and para-aortic lymphadenopathy; hence, lymphoma was highly suspected. Histopathological examination revealed sarcoidosis picture. The diagnoses of sarcoidosis was confirmed depending on histopathological findings as well as the presence of chronic nonspecific constitutional symptoms, and progressive weight loss. In addition, it was depending on laboratory results which showed the presence of chronic inflammatory process, hypercalcemia and negative infectious screening results.

In multisystemic sarcoidosis, despite most literature recommended corticosteroid to be continued for not ០ months,[9] prednisolone alone was given for 2 months only due to the concern about patient's glycemic control. It cured all her symptoms for 1½-year follow-up.

Now, this case raised the following questions; Does typhoid fever trigger this case of sarcoidosis or it activates an already existed latent sarcoidosis? Does sarcoidosis develop after typhoid fever independently? All of these probabilities are possible, but it was difficult to determine which one was the scenario, and we reported it here for documentation and for further evaluation in the future.


   Conclusion Top


We concluded that sarcoidosis needs to be included in the differential diagnosis of any case of typhoid fever which does not respond fully to treatment and in any case of nonspecific constitutional symptoms with negative tuberculin test. Equally important, the monotreatment with prednisolone tablet 40 mg/day then tapered gradually for a total duration of 2 months can be sufficient. Finally, we found that typhoid fever has a possible correlation with sarcoidosis by activating or triggering it. This conclusion must be interpreted with caution and considered tentative till further cases reported in the future.

Acknowledgment

The authors are much grateful to Dr. Thant Zin for his assistance and to the patient for her agreement to publish her case.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Weisinger HS, Steinfort D, Zimmet AD, Hall AJ, Pesudovs K. Sarcoidosis: Case report and review. Clin Exp Optom 2006;89:361-7.  Back to cited text no. 1
    
2.
Nunes H, Bouvry D, Soler P, Valeyre D. Sarcoidosis. Orphanet J Rare Dis 2007;2:46.  Back to cited text no. 2
    
3.
Rybicki BA, Major M, Popovich J Jr., Maliarik MJ, Iannuzzi MC. Racial differences in sarcoidosis incidence: A 5-year study in a health maintenance organization. Am J Epidemiol 1997;145:234-41.  Back to cited text no. 3
    
4.
Sharma SK, Mohan A. Sarcoidosis in India: Not so rare! JIACM 2004;5:12-21.  Back to cited text no. 4
    
5.
Alhamad EH, Idrees MM, Alanezi MO, Alboukai AA, Shaik SA. Sarcoidosis-associated pulmonary hypertension: Clinical features and outcomes in Arab patients. Ann Thorac Med 2010;5:86-91.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Costabel U. Sarcoidosis: Clinical update. Eur Respir J Suppl 2001;32:56s-68s.  Back to cited text no. 6
    
7.
Saidha S, Sotirchos ES, Eckstein C. Etiology of sarcoidosis: Does infection play a role? Yale J Biol Med 2012;85:133-41.  Back to cited text no. 7
    
8.
Eishi Y, Suga M, Ishige I, Kobayashi D, Yamada T, Takemura T, et al. Quantitative analysis of mycobacterial and propionibacterial DNA in lymph nodes of Japanese and European patients with sarcoidosis. J Clin Microbiol 2002;40:198-204.  Back to cited text no. 8
    
9.
Judson MA. The treatment of pulmonary sarcoidosis. Respir Med 2012;106:1351-61.  Back to cited text no. 9
    

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Correspondence Address:
Naghem Farouk Abed
Universiti Tunku Abdul Rahman, FMHS, Selangor
Malaysia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ATMPH.ATMPH_3_17

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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