medicines Case Report Adrenal Cortical Rests in the Fallopian Tube: Report of a Case and Review of the Literature Theodoros Tzigkalidis 1, Eleni Skandalou 2, Maria Eleni Manthou 3,*, Nikolaos Kolovogiannis 4 and Soultana Meditskou 3 1 Pathology Department, General Hospital “Agios Pavlos”, 55134 Thessaloniki, Greece; [email protected] 2 Internal Medicine Department, General Hospital “Papanikolaou”, 57010 Thessaloniki, Greece; [email protected] 3 Laboratory of Histology-Embryology, School of Medicine, Faculty of Health Sciences, Aristotle University of Thessaloniki, 54124 Thessaloniki, Greece; [email protected] 4 Obstetrics and Gynecologic Department, General Hospital of Polygyros, 63100 Chalkidiki, Greece; [email protected] * Correspondence: [email protected]; Tel.: +30-2310999070 Abstract: We report an extremely unusual finding discovered incidentally during a routine pathologic evaluation of a fallopian tube, surgically removed on the grounds of ectopic pregnancy. We came across a minute yellowish nodule situated within the wall of the salpinx, which corresponded to ectopic adrenal cortex, as verified by microscopical examination, and coexisted with salpingitis isthmica nodosa. A research of the available English literature on this subject confirmed the rareness of the entity we encountered. This case is presented because of its uniqueness, aiming to raise awareness of a rare condition which is discussed along with potential diagnostic dilemmas, its prognostic significance, and possible complications. Citation: Tzigkalidis, T.; Skandalou, Keywords: adrenocortical rest; ectopic adrenal; fallopian tube; adrenal ectopy; adult E.; Manthou, M.E.; Kolovogiannis, N.; Meditskou, S. Adrenal Cortical Rests in the Fallopian Tube: Report of a Case and Review of the Literature. 1. Introduction Medicines 2021, 8, 14. https:// Adrenal glands are normally situated on the kidneys and have a double embryological doi.org/10.3390/medicines8030014 origin [1]. Ectopias of the adrenal gland, and especially of its cortex, are mostly reported during childhood and may be encountered in association with pelvic structures, usually Academic Editor: M Bernal-López those of the genitourinary system, and usually in males [2,3]. Adrenal cortical rests occur in 50 per cent of new-borns and usually regress and disappear within a few years [3], but they Received: 17 February 2021 may remain and persist as functioning tissue throughout adulthood [1]. Ectopic adrenal Accepted: 9 March 2021 Published: 12 March 2021 tissue is generally rarely observed and is usually a random finding [2]. We report an extremely unusual finding of adrenal cortical rest within the wall of salpinx, which is an extremely rare site [4,5], in an older female, which is even more Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in unusual. The lesion was discovered incidentally during a routine pathologic evaluation published maps and institutional affil- of a fallopian tube, surgically removed on the grounds of ectopic pregnancy. This case is iations. presented because of its uniqueness, aiming to raise awareness of a rare condition which is discussed along with the pathogenetic mechanism responsible for the lesion’s occurrence, potential diagnostic dilemmas, its prognostic significance and possible complications. 2. Case Presentation Copyright: © 2021 by the authors. Licensee MDPI, Basel, Switzerland. A 37-year-old female patient visited the local hospital unit complaining of pelvic pain This article is an open access article and discomfort, combined with vaginal bleeding. She reported that symptoms began a distributed under the terms and few days earlier and that her last menstruation was documented 7 weeks prior to the conditions of the Creative Commons symptoms. Her personal medical history only included a surgically removed right ovarian Attribution (CC BY) license (https:// cyst 5 years before, which was diagnosed as a benign serous cystadenoma. creativecommons.org/licenses/by/ A blood count was performed, revealing normal values. CEA (carcinoembryonic 4.0/). antigen), aFP (alpha fetoprotein), CA19-9 (carbohydrate antigen 19-9) values were within Medicines 2021, 8, 14. https://doi.org/10.3390/medicines8030014 https://www.mdpi.com/journal/medicines Medicines 2021, 8, x FOR PEER REVIEW 2 of 6 symptoms. Her personal medical history only included a surgically removed right ovar- Medicines 2021, 8, 14 ian cyst 5 years before, which was diagnosed as a benign serous cystadenoma. 2 of 6 A blood count was performed, revealing normal values. CEA (carcinoembryonic an- tigen), aFP (alpha fetoprotein), CA19-9 (carbohydrate antigen 19-9) values were within normalnormal ranges. ranges. Serum Serum CA-125 CA-125 (cancer (cancer antigen antigen 125) was found at 18.6 IU/mLIU/mL (the (the upper normalnormal value value is is 35 35 IU/mL). IU/mL). βHCGβHCG (beta (beta human human chorionic chorionic gonadotropin) gonadotropin) was was measured measured at 11,564at 11,564 mIU/mL, mIU/mL, suggesting suggesting pregnancy. pregnancy. An An endometrial endometrial curettage curettage was was performed performed and and ββHCGHCG dropped dropped to to 9541.92 mIU/mL. Thorough Thorough ultrasonographic ultrasonographic examination examination was was per- per- formed,formed, which which indicated indicated an an ectopic ectopic pregnancy pregnancy in in the the right right fallopian fallopian tube. tube. It It was was therefore therefore immediatelyimmediately decided decided to to have have it it su surgicallyrgically removed. removed. After After surgery, surgery, βHCGHCG dropped to 2418.742418.74 mIU/mL. mIU/mL. All All specimens specimens were were sent sent to th toe thelaboratory laboratory for histol for histologicalogical evaluation evaluation and diagnosis.and diagnosis. Macroscopically,Macroscopically, the salpinx measured 8 × 2.52.5 cm cm and and appeared appeared intact intact but but was was obvi- obvi- ouslyously dilated dilated in in the the middle middle of of its its ampullary region. region. The The lumen lumen seemed seemed to to be be filled filled with clotsclots of of blood. blood. The The organ organ was was cut cut perpendicularl perpendicularlyy to to its its long long axis axis at at sequential sequential sections, sections, measuringmeasuring approximately approximately 0.5 0.5 cm cm each. each. During During sectioning, sectioning, a a minute, minute, yellowish yellowish and and firm firm nodulenodule was found located withinwithin thethe wallwall ofof thethe salpinx, salpinx, measuring measuring no no more more than than 2 mm.2 mm. It Itwas was prepared prepared for for microscopic microscopic investigation. investigation. MicroscopicallyMicroscopically with with H&E H&E (hematoxylin (hematoxylin eo eosin)sin) staining, staining, many many chorionic chorionic villi villi and trophoblastictrophoblastic cellcell islandsislands were were observed observed between between fibrin fibrin thrombi thrombi and and the luminalthe luminal blood blood clots clotsin the in salpinx the salpinx (Figure (Figure1a). The 1a). endometrial The endometrial samplings samplings exhibited exhibited morphological morphological features fea- of turesa hypersecretory of a hypersecretory or gestational or gestational endometrium, endometrium, obviously obviously caused by caused increased by increased progesterone pro- gesteroneeffect, confirming effect, confirming the initial the clinical initial diagnosis clinical diagnosis of ectopic of tubal ectopic pregnancy. tubal pregnancy. Meanwhile, Mean- the while,very small the very nodule small discovered nodule discovered was located wa betweens located bundles between of bundles smooth muscleof smooth cells muscle of the cellsouter of longitudinal the outer longitudinal muscularis muscularis layer, with layer, interspersed with interspersed loose connective loose connective tissue of serosatissue ofpresent. serosa Thepresent. nodule The appeared nodule appeared well demarcated, well demarcated, it was surrounded it was surrounded by a thin fibrous by a thin rim fibrousand was rim composed and was composed of two intermingled of two intermingl but distincted but cell distinct populations, cell populations, arranged arranged in small inparallel small cordsparallel and cords trabeculae and trabeculae (Figure1 (Figureb). 1b). (a) (b) FigureFigure 1. 1. FallopianFallopian tube: tube: ( (aa)) fibrotic fibrotic chorionic chorionic villi villi and trophoblast in the lumen ofof thethe salpinxsalpinx (H(H && E,E, ××40);40); ( (bb)) demarcated demarcated nodulenodule in the fallopian fibromuscular fibromuscular layer (blue arrow) (H & E, ××20).20). AllAll cells cells were clearlyclearly outlined,outlined, withwith small, small, uniform uniform and and pycnotic pycnotic nuclei, nuclei, which which had had no nosigns signs of atypiaof atypia or mitoses.or mitoses. Many Many cells cells appeared appeared with with clear clear and and abundant, abundant, microvesicular microvesic- ular(lipid-rich) (lipid-rich) cytoplasm, cytoplasm, while while some some had amphophilichad amphophilic to slightly to slightly eosinophilic eosinophilic cytoplasm cyto- plasm(Figure (Figure2). 2). Medicines 2021, 8, x FOR PEER REVIEW 3 of 6 Medicines 2021, 8, 14x FOR PEER REVIEW 3 of 6 (a) (b) (a) (b) Figure 2. (a,b). Nodule: vacuolated lipid-rich cell population (zona fasciculata) admixed with a lesser population of eosin- ophilicFigure or2. 2. (amphophilica(a,b,b).). Nodule: Nodule: cells vacuolated vacuolated (zona glomerulosa) lipid-rich lipid-rich cell (H