lNTRODUCTlON

Tuberculosis(TB)is known to present with varied clinical features,but involvement of the adrenal glands in TB is rare[1].Isolated adrenal TB accounts for under 2% of adrenal incidentalomas[2],while 75%-100% of patients with adrenal TB have bilateral involvement[3-5].As a result,unilateral adrenal TB is considered a rare clinical entity.The computed tomography(CT)imaging characteristics of adrenal TB are significantly different from those of primary tumors,such as small size,low attenuation in the center,and peripheral rim enhancement[3,6,7].Herein,we report an unusual case of unilateral adrenal TB whose imaging characteristics were extremely atypical and suggested a high likelihood of a malignant tumor.Fine-needle aspiration biopsy(FNAB)was used to confirm TB,and prolonged anti-TB treatment was given to stabilize her condition.

CASE PRESENTATlON

Chief complaints

A 42-year-old female was admitted to our department after presenting with a half-year history of osphyalgia.

History of present illness

A 42-year-old female had low back pain for a half year,which was exaggerated when taking a deep breath or lying flat and relieved when standing.She denied frequent micturition,painful urination,fever,hematuria,or pyuria.

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History of past illness

The patient had a remote history of hypertension.She had been diagnosed with pulmonary and cerebral TB four months before presentation,and she had started a regimen of anti-TB drugs(isoniazid 0.3 g QD,rifampicin 0.45 g QD,ethambutol 0.75 g QD,and ofloxacin 0.5 g QD)upon diagnosis.

Personal and family history

Yu YR and An ZM contributed to the conceptualization;Liu H and Tang TJ collected the information;Liu H wrote the original draft;Yu YR reviewed and edited the manuscript;all authors issued final approval for the version to be submitted.

Physical examination

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Laboratory examinations

Laboratory tests yielded the following results: plasma total cortisol(8 am),594.6 nmol/L(reference,147.3-609.3 nmol/L);adrenocorticotropic hormone,48.30 ng/L(reference,5.0-78 ng/L);and aldosterone/renin ratio,16.92 ng/dL per ng/mL/h.No abnormalities were observed in the blood and urine catecholamines.Routine blood tests,routine urine tests,and biochemical tests were roughly in the normal range.CT-guided Fine needle aspiration biopsy(FNAB)of the right adrenal gland was performed,and pathological examination detected granulomas and necrosis(Figure 1A and B).

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Imaging examinations

Patients with adrenal TB are usually treated with standard quadruple antitubercular treatment(such as isoniazid,rifampicin,pyrazinamide,and ethambutol)[18-20]for nearly 12 mo or longer.Adverse reactions to anti-TB drugs and their interactions with corticosteroids that are administered for replacement therapy remain challenging[21,22].Firstly,rifampicin increases cortisol catabolism while isoniazid produces increased levels of cortisol

an opposite effect on the enzyme activity 6-Bhydroxylase;secondly,hepatitis,induced by isoniazid and worsened by rifampicin,leads to failure of 11-B-oxo-reductase,which converts cortisone to cortisol;and finally,tuberculous Addison’s disease might require increased amounts of hydrocortisone due to rifampicin administration[23].Up to 70% of patients with active TB have subclinical adrenal insufficiency[24].Anti-TB treatment might cause adrenal crisis[18],and patients should be closely monitored when starting this treatment.Most of the cases demonstrated a good response to anti-TB treatment.Early diagnosis and no delay in treatment initiation contributed to minimizing the high mortality rate[22,25].Addison’s disease usually occurs when more than 90% of adrenal tissue has been destroyed[26].Only a few patients with tuberculous Addison's disease showed recovery of adrenal function[27].

FlNAL DlAGNOSlS

Unilateral adrenal tuberculosis.

TREATMENT

Treatment with 4 anti-TB drugs was continued.

OUTCOME AND FOLLOW-UP

The authors declare that they have no conflict of interest.

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DlSCUSSlON

Extrapulmonary TB constitutes about 15%-20% of all TB patients[8].The most frequent sites of extrapulmonary TB include the lymph nodes(19%),pleural cavity(7%),gastrointestinal tract(4%),bone(6%),central nervous system(3%),and genitourinary system(1%)[9].Of the 370 reports of extrapulmonary TB in a systematic review spanning 10 years[1],only one case was shown to involve the adrenal gland,which demonstrated adrenal TB as a rare clinical entity.Bilateral involvement usually occurs because of hematogenous and lymph spread from the site of the primary mycobacterial infection to both adrenal glands,which are equally susceptible[10].In our case,adrenal TB(lesions of 1.5 cm to 5 cm)was aggravated while the anti-TB regimen was continued.It is necessary to distinguish adrenal masses from adrenal tumors.Adrenal incidentalomas,adenomas,metastases,adrenocortical carcinomas,myelolipomas,and pheochromocytomas accounted for 41%,19%,10%,9%,and 8%,respectively.The etiologies of these partly depend on the size,such that larger tumors are more likely to be malignant.Adrenal carcinomas and metastases comprise 25% and 18% of lesions and are larger than 6 cm,while adenomas account for only 18%[11].For tumors smaller than 4 cm,adrenal carcinomas comprise 2% and adenomas comprise 65%[11].Untreated TB lesions were smaller than primary tumors(2.8 cm ± 1.3 cm

3.5 cm ± 2.4 cm)[6],while the diameters of benign,malignant pheochromocytoma and adrenocortical carcinoma were 5.7 cm ± 2.3 cm,8.3 cm ± 4.1 cm[12]and 11 cm ± 4 cm[13],respectively.It is difficult to provide evidence with regard to TB based on size(6.0 cm × 4.5 cm)in this case.The CT value,attenuation measurement,and reduced central area(7 ± 4 HU)compared to the peripheral area(32 ± 14 HU)were observed between unenhanced and contrast-enhanced scans in adrenal TB[3].This characteristic of central necrosis surrounded by fibrous and granulomatous inflammatory tissue is much less common in primary adrenal tumors[6],owing to sufficient blood supply in the central area.Pheochromocytoma always has a high enhancement of ＞ 110 HU in the arterial phase[14],while adrenocortical carcinoma is less likely to show an enhancement of ＞100 HU.In the present case,evidence from images could not rule out a malignant tumor in the adrenal gland.In addition,calcification preferentially occurred in the later stages of adrenal TB than in adrenal tumors(59%

8%),which helped with a proper diagnosis[6].

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In addition to the ineffectiveness of FNAB in distinguishing adrenal adenoma from adenocarcinoma[11],it is considered advantageous due to its ease,cost-effectiveness,reduced time consumption,low complication rates,and high accuracy[15-17].It is often used for suspected nonfunctional and nonneoplastic adrenal gland lesions,but not employed for pheochromocytomas[11,17]due to the risk of hemodynamic instability.In the present case,blood pressure was well controlled and had no abnormalities in blood and urine catecholamines,indicating a lower possibility of pheochromocytoma.Therefore,FNAB was performed to obtain histological evidence,which subsequently provides clear evidence regarding targeted therapy.

Four months prior to presentation,the patient had undergone an abdominal CT scan,which revealed a 1.5 cm isointense mass in the right adrenal gland(Figure 2A).On presentation to our department,she underwent another abdominal CT scan,which revealed a 6.0 cm × 4.5 cm irregular mass with uneven density in the right adrenal gland,while the left adrenal gland was normal(Figure 2B).

CONCLUSlON

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

Physical examination showed nothing special despite percussive pain in the right kidney area.

FOOTNOTES

Her personal and family history were insignificant.

Unilateral adrenal tuberculous infection,although rare,should be considered in patients with unilateral adrenal mass but without Cushing syndrome,primary aldosteronism,or pheochromocytoma.FNAB assists in diagnosing TB,and early initiation and longer duration of anti-TB therapy are crucial to treating patients with unilateral adrenal tuberculous infection.

The patient’s low back pain was relieved,and abdominal CT(Figure 2C)demonstrated a significant reduction of the mass in the right adrenal gland(2.7 cm × 2.4 cm)after 15 mo of anti-TB therapy.Three years later,abdominal CT(Figure 2D)showed a slight enlargement of the right adrenal gland.

This year my youngest son will be a senior in high school. He, too, has now announced that he is too old for notes. But like his older brother and sister before him, he will receive those notes till the day he graduates-and in whatever lunches I pack for him afterwards.

The authors have read the CARE Checklist(2016),and the manuscript was prepared and revised according to the CARE Checklist(2016).

This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers.It is distributed in accordance with the Creative Commons Attribution NonCommercial(CC BYNC 4.0)license,which permits others to distribute,remix,adapt,build upon this work non-commercially,and license their derivative works on different terms,provided the original work is properly cited and the use is noncommercial.See: https://creativecommons.org/Licenses/by-nc/4.0/

Before this, while he was staying down there, she had told him that she was a daughter of the same troll whom the youth served, and he had shut her up there to keep her away from everyone

China

Hui Liu 0000-0002-9352-3358;Tian-Jiao Tang 0000-0002-4817-2907;Zhen-Mei An 0000-0003-1435-8209;Ye-Rong Yu 0000-0003-0103-0087.

Xing YX

A

Xing YX

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