- Email: [email protected]
Pediatric and Adolescent Surgical Breast Clinic: Preliminary Experience
Original Study Pediatric and Adolescent Surgical Breast Clinic: Preliminary Experience Osher Cohen MD 1,2,*, Efrat Avinadav MD 1,2, Eran Sharon MD 2,3, Avinoam Pirogovsky MD 2,4, Enrique Freud MD 1,2 1
Departments of Pediatric and Adolescent Surgery, Schneider Children's Medical Center of Israel, Petach Tikva, Israel Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel 3 Department of Surgery, Rabin Medical Center e Beilinson Hospital, Petach Tikva, Israel 4 Medical Management, Schneider Children's Medical Center of Israel, Petach Tikva, Israel 2
a b s t r a c t Study Objective: The diagnostic and treatment tools used in breast centers are largely geared to adults, and there is little consideration of the unique characteristics of breast diseases in younger age groups. Herein we report on the preliminary experience of a specialized breast clinic for children and adolescents. Design: Retrospective, observational. Setting: Pediatric Surgical Breast Clinic of Schneider Children's Medical Center of Israel. Participants: Patients referred to the clinic during the first 18 months of its establishment. Interventions and Main Outcome Measures: Rate of breast masses, rate of malignancy, and types of evaluation and treatment. Results: Forty-seven patients aged 0-19 years were referred. Breast masses were suspected in 23/47 (48.9%) and confirmed using ultrasound in 14/47 (29.7%; mean age, 16.4 years), followed by needle core biopsy in 7. Seven patients had multiple masses. Breast Imaging and Reporting Data System scores ranged from 3 to 4b. All cases were treated as fibroadenomas. Four patients underwent surgery because of a large mass (O5 cm; n 5 3) or patient's preference to remove the lump rather than follow-up (n 5 1). There were no malignancies. The median time from patient identification of the mass until seeking medical help was 5 months. Conclusion: Several findings distinct to this age group were highlighted: high rate of multiple masses, lack of malignancy, and high risk of delayed diagnosis. Care should be taken when using Breast Imaging and Reporting Data System categorization because it was formulated for adults and might be very limited in the adolescent population. Longer-term studies of larger cohorts are planned. Key Words: Breast mass, Breast clinic, Fibroadenoma, Adolescent, BI-RADS
Introduction
The awareness of the lay and professional populations of breast diseases has increased considerably in recent years owing to the widespread dissemination of information by the electronic and print media and nationwide educational programs. Breast disease clinics are a major arm of general surgery practice, but the diagnostic tools, the treatments offered, and even the environment are geared to adults. Little attention has been addressed to the natural history and specific characteristics of breast diseases in the pediatric age group. This is important because children and adolescents do not routinely undergo breast examination by a surgeon or using imaging, and mammography is not efficacious because of the dense breast tissue in young patients.1 Ionizing radiation also poses a potential risk to the developing breast.2 Thus, it is the patients themselves who usually notice abnormalities and sets the timing of seeking medical help. Furthermore, breast masses in children and adolescent are usually benign, mainly fibroadenomas.3,4 The authors indicate no conflicts of interest. * Address correspondence to: Osher Cohen, MD, Department of Pediatric and Adolescent Surgery, Schneider Children's Medical Center of Israel, 14 Kaplan Street, Petach Tikva 4920235, Israel; Phone: (972) 545879954 E-mail address: [email protected] (O. Cohen).
Breast malignancies are rare in adolescents, and when a malignancy is found, it is usually a nonbreast metastatic disease such as Hodgkin and non-Hodgkin lymphoma, neuroblastoma, hepatocellular carcinoma, and rhabdomyosarcoma.5,6 Primary breast cancer is extremely rare.5 The most common primary cancer in adolescents is juvenile secretory carcinoma, followed by intraductal carcinoma, rhabdomyosarcoma, lymphoma,5e8 and rarely, ductal carcinoma in situ.9 At present, there are no established guidelines or algorithms for the investigation and treatment of breast disease in the pediatric population. Nevertheless, it is currently well recognized that ultrasonography is the optimal imaging tool in this age group for confirming the diagnosis and characterizing the mass.10,11 More invasive methods such as core needle biopsy might also be used when a tissue diagnosis in warranted. Additionally, children and adolescents with suspected or confirmed breast disease are evaluated or followed in general pediatric clinics or adult breast clinics or by various medical professionals including general surgeons, gynecologists, or primary pediatricians, all with different management approaches. To offer children and adolescents a more uniform, comprehensive solution, in 2017 the Department of Pediatric and Adolescent Surgery at Schneider Children's
1083-3188/$ - see front matter Ó 2019 North American Society for Pediatric and Adolescent Gynecology. Published by Elsevier Inc. https://doi.org/10.1016/j.jpag.2019.08.010
2
O. Cohen et al. / J Pediatr Adolesc Gynecol xxx (2019) 1e4
Medical Center of Israel (SCMCI) opened a dedicated pediatric surgical breast clinic run by 2 pediatric surgeons who are also board certified general surgeons. The aim of this report was to present the preliminary experience of the clinic. Materials and Methods
The study was conducted at the Pediatric Surgical Breast Clinic of SCMCI, which works in cooperation with the Breast Center of Rabin Medical Center on the same campus. Included were patients who attended the clinic during the first 18 months of its establishment. All patients were referred by their primary physician or a specialist for evaluation of suspected breast disease. Referral criteria were all patients younger than the age of 18 years with suspected breast disease other than cosmetic issues. The patients were further evaluated in clinic by 1 of the 2 surgeons who staffed the clinic. Sonography was used as the imaging modality when needed. The sonographic evaluation was concluded with the Breast Imaging Reporting and Data System (BI-RADS) score (the BI-RADS is a standardized, imaging-based risk assessment tool for breast cancer developed by the American College of Radiology; the score rages from 0 to 6 where 0 represents incomplete study, 3 is “probably benign,” 4 is suspected malignancy, and 6 is a biopsy proven malignancy). Data were prospectively entered into the electronic files and collected retrospectively for the study, with a focus on patients who presented with breast masses. Data collected included age, gender, reason for referral, time elapsed since first self-identifying a concerning finding, relevant family history, preliminary inquiry findings and the findings on physical exam, sonography, and pathology. Additionally data on all invasive interventions before and after the first visit were recorded. The study was approved by the local institutional review board (0064-19-RMC). Descriptive statistics were used to analyze the data. Results
In the first 18 months of clinic activity (August 2017 to January 2019), 47 patients were referred for evaluation, 44 female and 3 male. The cohort included 42 adolescents (age 10-19 years12), 3 children (age 1-10 years), and 2 infants (01 year). One or more suspected breast masses were the reason for referral in 23/47 patients (48.9%). They included 20 adolescents, 2 children, and 1 infant. The remaining patients were referred because of an infection or abscess (8 patients), breast pain (4 patients), and other reasons (1-3 patients each): breast cyst, nipple discharge, skin lesions, breast asymmetry, excess nipple or breast, and gynecomastia. The patients with a suspected breast mass underwent further evaluation and treatment as necessary under the auspices of the clinic. Some of the patients with a presentation other than a breast mass were referred to the appropriate discipline: plastic surgery, endocrinology, and gynecology. On evaluation, the presence of at least 1 mass was confirmed in 14 adolescents of the 23 patients (60.8%; 29.7%
of the cohort), including 7/14 (50%) who had several masses (up to 15). Findings were unilateral in 3 patients and bilateral in 4. Mean age of the patients with masses was 16.4 years (Table 1). The other 9 patients included 1 neonate with neonatal breast hypertrophy and 2 young patients with retroalveolar mass diagnosed as prepubertal breast bud. The other 6 patients with self-palpated “masses” were adolescents with no defined mass detected by the surgeon and were not identified using sonography. The suspected masses were found to be normal breast tissue or fibrocystic changes. All 20 adolescents underwent sonography to verify clinical findings, either in the community by referral of their primary physician, or at the breast clinic. The size of the masses on sonography ranged from 0.6 to 7 cm. Scores on the BI-RADS ranged from 3 to 4b; nevertheless, all patients were assumed to have a fibroadenoma on the basis of the clinical presentation, the sonographic findings, and it was confirmed on pathology in the cases that biopsy was taken. The attending physicians reported particularly high anxiety and stress in the patients with the higher BI-RADS scores and their parents. Seven patients were further evaluated using core needle biopsy. The BI-RADS score for 6 of them was 4A or higher and it was 3 in 1 patient. The diagnosis was fibroadenoma in all cases. Four patients eventually received surgery and the others were followed clinically. The reason for surgery was the large size of the mass (O5 cm in diameter) in 3 patients (Fig. 1) and patient's preference to remove the lump rather than follow-up in the fourth patient (Fig. 2). Fibroadenoma was confirmed on final pathological study in all 4 patients. In no case was a malignancy detected. The time elapsed from the patients’ initial self-palpation of the mass until they voiced their concerns to their parent(s) ranged from 2 weeks to 3 years (median, 5 months). When the parents were informed, they sought medical advice immediately. Discussion
In the present report we describe the first 18 months’ experience of a novel pediatric breast clinic that was established to provide comprehensive care to children and adolescents with breast masses. The clinic is staffed by medical personnel who are alert to the unique aspects of breast disease in the young population. It is located in SCMCI, where the environment is oriented to the younger age groups, and it operates in cooperation with the adult Breast Center of Rabin Medical Center on the same campus. Our clinic is distinct from other adolescent breast clinics that focus on cosmetic treatment of macromastia, gynecomastia, and breast asymmetry; this probably explains the high percentage of patients with breast masses in the study cohort. Some of the patients with other findings were referred to the relevant disciplines. Our review revealed several important features of pediatric breast disease. The patients with suspected breast masses accounted for 48.9% of all referrals (23/47). Of them, the diagnosis of breast mass was made clinically and radiologically in 14/47 patients (29.7%) and verified in subsequent biopsy in 7; 7 patients had more than 1 mass.
O. Cohen et al. / J Pediatr Adolesc Gynecol xxx (2019) 1e4
3
Table 1 Patients With Confirmed Breast Mass or Masses Patient Age, years Months since Self-Palpation* Number 1 2 3 4 5 6 7 8 9 10 11 12 13 14
17.4 14.9 16.7 16.5 17.4 17.7 15.5 16.11 15 17.6 16.2 16.5 16.11 17
6 18 5 1 4 27 0.5 12 2 0.5 36 1.5 6 Was detected by physician
Clinical Findings
Sonographyy
Right breast UOQ Upper right breast Multiple, bilateral Multiple, bilateral Right breast UOQ Right breast UOQ Multiple, right breast Multiple, right breast Right breast LOQ Multiple, bilateral Multiple, right breast Multiple, bilateral Right breast, retroalveolar Right breast UOQ
3 cm solid mass 7 cm 15 masses, 3 cm for the largest 5 masses, 4.5 cm for the largest 1.9 cm 5.7 cm 4 masses, 2 cm for the largest 3 masses, 1.4 cm for the largest 4.2 cm mass enlarged to 5.8 cm 2 masses, 3.6 cm for the largest 2 masses, 4.6 cm for the largest 8 masses, 3.7 cm for the largest 3.3 cm 3.7 cm
Largest BI-RADS Score 3 4A 3 4A 4A 4B 3 3 4A 4A 4A 3 3 3
Intervention
Pathology
CNB FA CNB and excision FA F/U CNB FA CNB FA Excision FA F/U F/U CNB and excision FA CNB FA CNB and excision FA F/U F/U F/U
BI-RADS, Breast Imaging and Reporting Data System; CNB, core needle biopsy; FA, fibroadenoma; F/U, clinical follow-up; LOQ, lower outer quadrant; UOQ, upper outer quadrant. * Time elapsed since the patients first palpated the mass to the time for seeking medical advice. y Sonographic description of the mass or masses including the largest diameter.
All masses were diagnosed and treated as fibroadenomas. Four masses were surgically excised. There were no cases of phyllodes or cancer; in agreement with the literature showing a low rate of breast malignancy in the pediatric age group.5e9 Further studies of a larger cohort followed for a longer term are planned. On the basis of the patients’ interviews and clinical encounters we assume that anxiety and shame played a major role in the medical experience of the patients and often led to a delay in their seeking treatment. A protective and welcoming medical environment that is adapted to children and adolescents is important to gain patient trust and compliance for further follow-up. All caregivers in the breast clinic are made aware of these special needs and ways of providing suitable care. From a clinical point of view, surgeons working in the clinic need to be familiar with the normal breast development in the pediatric population and the diseases and lesions that are likely to occur. Accordingly, the evaluation of breast disease in children and adolescents, in addition to clinical judgements and decisions regarding imaging, tissue diagnosis, surgical intervention, and follow-up, should not
be derived from practices in adults. Furthermore, the medical staff should be particularly mindful of patient and parental concerns and anxiety and the effect of the disease or treatment on body image and self-esteem. The physical examination and imaging studies should be performed in an environment conducive to the well-being of the young patients and meet their physical and emotional needs. Considering the emotional state of the patients and parents is crucial to making decisions about imaging, tissue diagnosis, or surgery. It is also important to discuss the differences in adults with the patients and their parents to dispel their concerns and help them to make the right treatment decisions.13 Radiologists also need to be alert to the unique characteristics of pediatric breast disease. According to the American College of Radiology, category 4A of the BI-RADS reporting system represents a 2%-10% likelihood of cancer and mandates tissue diagnosis.14 However, in our cohort, a relatively large number of patients (7/14; 50%) had a BI-RADS score of 4A or higher, but none had a malignancy. This finding suggests that the use of this reporting system in adolescents might lead to inaccurate categorization and consequently, unnecessary stress to
Fig. 1. Macroscopic appearance of a large fibroadenoma (patient 2).Ă
Fig. 2. Fibroadenoma in an anxious patient who had sought medical help only 3 years after self-palpating the mass (patient 11).
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O. Cohen et al. / J Pediatr Adolesc Gynecol xxx (2019) 1e4
patient and parents. In addition, it can present a dilemma for the surgeon and sometimes lead to mistreatment. Some authors recommend clinical follow-up alone or with sonographic scans in most cases15e17; others recommend biopsy studies of any mass suggestive of malignancy and long-term follow-up for nonsuggestive masses3,17; still others recommend excision of any mass suggestive of malignancy,18,19 or masses that persist on long-term follow-up until age 35 years.19 The significance of the different categorization of breast masses in the pediatric and adult populations has hardly been discussed in the literature.20 Recently Sanders et al21 proposed that any palpable mass in a young patient should undergo ultrasound imaging, which should be followed, if findings are indicative of malignancy, with core biopsy. However, they emphasized that the reason for the biopsy is to rule out phyllodes tumor rather than breast cancer, further emphasizing the irrelevance of the BI-RADS that was developed for the adult population in which the probability of cancer is considerably higher. Further studies are needed to develop an ageadjusted system for the categorization of breast masses. This report presents the preliminary experience for a surgical breast clinic dedicated to the pediatric and adolescent patients. The relatively small cohort and short follow-up is a limiting factor for drawing clear recommendations regarding decision-making while evaluating these patients for either the invasive intervention needed or categorizing the risk for breast masses. Longer-term studies of larger cohorts are needed and planned. Another limit is that probably not all primary physicians are aware of our clinic and patients who are older than 16 years might be referred to an adult clinic according to definitions different to those for adolescents. That might cause selection bias in our clinic toward a younger age. Conclusions
Breast diseases in children and adolescents have several distinct features compared with adults and warrant a different type of management with greater consideration of emotional factors such as body image and shame. We suggest that many of the practices adopted from adults, particular those on the basis of the use of the BI-RADS scores, are inappropriate for this age group. This study highlights the need for tertiary clinics dedicated to breast
diseases in children and adolescents that offer comprehensive, age-adjusted care in a stress-free environment. Acknowledgments
The authors thank our research coordinator Naama Tirosh for her exceptional contribution to the current and other reports. References 1. De Silva NK, Brandt ML: Disorders of the breast in children and adolescents, part 2: breast masses. J Pediatr Adolesc Gynecol 2006; 19:415 2. García CJ, Espinoza A, Dinamarca V, et al: Breast US in children and adolescents. Radiographics 2000; 20:1605 3. West KW, Rescorla FJ, Scherer LR 3rd, et al: Diagnosis and treatment of symptomatic breast masses in the pediatric population. J Pediatr Surg 1995; 30:182 4. Jayasinghe Y, Simmons PS: Fibroadenomas in adolescence. Curr Opin Obstet Gynecol 2009; 21:402 5. Rogers DA, Lobe TE, Rao BN, et al: Breast malignancy in children. J Pediatr Surg 1994; 29:48 6. Corpron CA, Black CT, Singletary SE, et al: Breast cancer in adolescent females. J Pediatr Surg 1995; 30:322 7. NIH: National Cancer Institute. Surveillance, Epidemiology, and End Results Program: SEER Cancer Statistics Review, 1975-2014. Available: https://seer. cancer.gov/csr/1975_2014/. Accessed August 3, 2019. 8. Longo OA, Mosto A, Moran JC, et al: Breast carcinoma in childhood and adolescence: case report and review of the literature. Breast J 1999; 5:65 9. Tea MK, Asseryanis E, Kroiss R, et al: Surgical breast lesions in adolescent females. Pediatr Surg Int 2009; 25:73 10. Kaneda HJ, Mack J, Kasales CJ, et al: Pediatric and adolescent breast masses: a review of pathophysiology, imaging, diagnosis, and treatment. AJR Am J Roentgenol 2013; 200:W204 11. Bock K, Duda VF, Hadji P, et al: Pathologic breast conditions in childhood and adolescence: evaluation by sonographic diagnosis. J Ultrasound Med 2005; 24:1347. [quiz: 1356]. 12. Canadian Paediatric Society: Age limits and adolescents. Paediatr Child Health 2003; 8:577 13. Gao Y, Saksena MA, Brachtel EF, et al: How to approach breast lesions in children and adolescents. Eur J Radiol 2015; 84:1350 €hm-Ve lez M, Berg WA, et al: ACR BI-RADS ultrasound. In: 14. Mendelson EB, Bo D’Orsi CJ, Sickles EA, Mendelson EB, et al, editors. ACR BI-RADS Atlas, Breast Imaging Reporting and Data System, (5th ed.).. Reston, VA, American College of Radiology, 2013, pp 128 15. McLaughlin CM, Gonzalez-Hernandez J, Bennett M, et al: Pediatric breast masses: an argument for observation. J Surg Res 2018; 228:247 16. Smith GE, Burrows P: Ultrasound diagnosis of fibroadenoma - is biopsy always necessary? Clin Radiol 2008; 63:511 17. Santen RJ, Mansel R: Benign breast disorders. N Engl J Med 2005; 353:275 18. Ezer SS, Oguzkurt P, Ince E, et al: Surgical treatment of the solid breast masses in female adolescents. J Pediatr Adolesc Gynecol 2013; 26:31 19. Greenberg R, Skornick Y, Kaplan O: Management of breast fibroadenomas. J Gen Intern Med 1998; 13:640 20. Fu CY, Hsu HH, Yu JC, et al: Influence of age on PPV of sonographic BI-RADS categories 3, 4, and 5. Ultraschall Med 2011; 32(suppl 1):S8 21. Sanders LM, Sharma P, Madany M, et al: Clinical breast concerns in low-risk pediatric patients: practice review with proposed recommendations. Pediatr Radiol 2018; 48:186
Departments of Pediatric and Adolescent Surgery, Schneider Children's Medical Center of Israel, Petach Tikva, Israel Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel 3 Department of Surgery, Rabin Medical Center e Beilinson Hospital, Petach Tikva, Israel 4 Medical Management, Schneider Children's Medical Center of Israel, Petach Tikva, Israel 2
a b s t r a c t Study Objective: The diagnostic and treatment tools used in breast centers are largely geared to adults, and there is little consideration of the unique characteristics of breast diseases in younger age groups. Herein we report on the preliminary experience of a specialized breast clinic for children and adolescents. Design: Retrospective, observational. Setting: Pediatric Surgical Breast Clinic of Schneider Children's Medical Center of Israel. Participants: Patients referred to the clinic during the first 18 months of its establishment. Interventions and Main Outcome Measures: Rate of breast masses, rate of malignancy, and types of evaluation and treatment. Results: Forty-seven patients aged 0-19 years were referred. Breast masses were suspected in 23/47 (48.9%) and confirmed using ultrasound in 14/47 (29.7%; mean age, 16.4 years), followed by needle core biopsy in 7. Seven patients had multiple masses. Breast Imaging and Reporting Data System scores ranged from 3 to 4b. All cases were treated as fibroadenomas. Four patients underwent surgery because of a large mass (O5 cm; n 5 3) or patient's preference to remove the lump rather than follow-up (n 5 1). There were no malignancies. The median time from patient identification of the mass until seeking medical help was 5 months. Conclusion: Several findings distinct to this age group were highlighted: high rate of multiple masses, lack of malignancy, and high risk of delayed diagnosis. Care should be taken when using Breast Imaging and Reporting Data System categorization because it was formulated for adults and might be very limited in the adolescent population. Longer-term studies of larger cohorts are planned. Key Words: Breast mass, Breast clinic, Fibroadenoma, Adolescent, BI-RADS
Introduction
The awareness of the lay and professional populations of breast diseases has increased considerably in recent years owing to the widespread dissemination of information by the electronic and print media and nationwide educational programs. Breast disease clinics are a major arm of general surgery practice, but the diagnostic tools, the treatments offered, and even the environment are geared to adults. Little attention has been addressed to the natural history and specific characteristics of breast diseases in the pediatric age group. This is important because children and adolescents do not routinely undergo breast examination by a surgeon or using imaging, and mammography is not efficacious because of the dense breast tissue in young patients.1 Ionizing radiation also poses a potential risk to the developing breast.2 Thus, it is the patients themselves who usually notice abnormalities and sets the timing of seeking medical help. Furthermore, breast masses in children and adolescent are usually benign, mainly fibroadenomas.3,4 The authors indicate no conflicts of interest. * Address correspondence to: Osher Cohen, MD, Department of Pediatric and Adolescent Surgery, Schneider Children's Medical Center of Israel, 14 Kaplan Street, Petach Tikva 4920235, Israel; Phone: (972) 545879954 E-mail address: [email protected] (O. Cohen).
Breast malignancies are rare in adolescents, and when a malignancy is found, it is usually a nonbreast metastatic disease such as Hodgkin and non-Hodgkin lymphoma, neuroblastoma, hepatocellular carcinoma, and rhabdomyosarcoma.5,6 Primary breast cancer is extremely rare.5 The most common primary cancer in adolescents is juvenile secretory carcinoma, followed by intraductal carcinoma, rhabdomyosarcoma, lymphoma,5e8 and rarely, ductal carcinoma in situ.9 At present, there are no established guidelines or algorithms for the investigation and treatment of breast disease in the pediatric population. Nevertheless, it is currently well recognized that ultrasonography is the optimal imaging tool in this age group for confirming the diagnosis and characterizing the mass.10,11 More invasive methods such as core needle biopsy might also be used when a tissue diagnosis in warranted. Additionally, children and adolescents with suspected or confirmed breast disease are evaluated or followed in general pediatric clinics or adult breast clinics or by various medical professionals including general surgeons, gynecologists, or primary pediatricians, all with different management approaches. To offer children and adolescents a more uniform, comprehensive solution, in 2017 the Department of Pediatric and Adolescent Surgery at Schneider Children's
1083-3188/$ - see front matter Ó 2019 North American Society for Pediatric and Adolescent Gynecology. Published by Elsevier Inc. https://doi.org/10.1016/j.jpag.2019.08.010
2
O. Cohen et al. / J Pediatr Adolesc Gynecol xxx (2019) 1e4
Medical Center of Israel (SCMCI) opened a dedicated pediatric surgical breast clinic run by 2 pediatric surgeons who are also board certified general surgeons. The aim of this report was to present the preliminary experience of the clinic. Materials and Methods
The study was conducted at the Pediatric Surgical Breast Clinic of SCMCI, which works in cooperation with the Breast Center of Rabin Medical Center on the same campus. Included were patients who attended the clinic during the first 18 months of its establishment. All patients were referred by their primary physician or a specialist for evaluation of suspected breast disease. Referral criteria were all patients younger than the age of 18 years with suspected breast disease other than cosmetic issues. The patients were further evaluated in clinic by 1 of the 2 surgeons who staffed the clinic. Sonography was used as the imaging modality when needed. The sonographic evaluation was concluded with the Breast Imaging Reporting and Data System (BI-RADS) score (the BI-RADS is a standardized, imaging-based risk assessment tool for breast cancer developed by the American College of Radiology; the score rages from 0 to 6 where 0 represents incomplete study, 3 is “probably benign,” 4 is suspected malignancy, and 6 is a biopsy proven malignancy). Data were prospectively entered into the electronic files and collected retrospectively for the study, with a focus on patients who presented with breast masses. Data collected included age, gender, reason for referral, time elapsed since first self-identifying a concerning finding, relevant family history, preliminary inquiry findings and the findings on physical exam, sonography, and pathology. Additionally data on all invasive interventions before and after the first visit were recorded. The study was approved by the local institutional review board (0064-19-RMC). Descriptive statistics were used to analyze the data. Results
In the first 18 months of clinic activity (August 2017 to January 2019), 47 patients were referred for evaluation, 44 female and 3 male. The cohort included 42 adolescents (age 10-19 years12), 3 children (age 1-10 years), and 2 infants (01 year). One or more suspected breast masses were the reason for referral in 23/47 patients (48.9%). They included 20 adolescents, 2 children, and 1 infant. The remaining patients were referred because of an infection or abscess (8 patients), breast pain (4 patients), and other reasons (1-3 patients each): breast cyst, nipple discharge, skin lesions, breast asymmetry, excess nipple or breast, and gynecomastia. The patients with a suspected breast mass underwent further evaluation and treatment as necessary under the auspices of the clinic. Some of the patients with a presentation other than a breast mass were referred to the appropriate discipline: plastic surgery, endocrinology, and gynecology. On evaluation, the presence of at least 1 mass was confirmed in 14 adolescents of the 23 patients (60.8%; 29.7%
of the cohort), including 7/14 (50%) who had several masses (up to 15). Findings were unilateral in 3 patients and bilateral in 4. Mean age of the patients with masses was 16.4 years (Table 1). The other 9 patients included 1 neonate with neonatal breast hypertrophy and 2 young patients with retroalveolar mass diagnosed as prepubertal breast bud. The other 6 patients with self-palpated “masses” were adolescents with no defined mass detected by the surgeon and were not identified using sonography. The suspected masses were found to be normal breast tissue or fibrocystic changes. All 20 adolescents underwent sonography to verify clinical findings, either in the community by referral of their primary physician, or at the breast clinic. The size of the masses on sonography ranged from 0.6 to 7 cm. Scores on the BI-RADS ranged from 3 to 4b; nevertheless, all patients were assumed to have a fibroadenoma on the basis of the clinical presentation, the sonographic findings, and it was confirmed on pathology in the cases that biopsy was taken. The attending physicians reported particularly high anxiety and stress in the patients with the higher BI-RADS scores and their parents. Seven patients were further evaluated using core needle biopsy. The BI-RADS score for 6 of them was 4A or higher and it was 3 in 1 patient. The diagnosis was fibroadenoma in all cases. Four patients eventually received surgery and the others were followed clinically. The reason for surgery was the large size of the mass (O5 cm in diameter) in 3 patients (Fig. 1) and patient's preference to remove the lump rather than follow-up in the fourth patient (Fig. 2). Fibroadenoma was confirmed on final pathological study in all 4 patients. In no case was a malignancy detected. The time elapsed from the patients’ initial self-palpation of the mass until they voiced their concerns to their parent(s) ranged from 2 weeks to 3 years (median, 5 months). When the parents were informed, they sought medical advice immediately. Discussion
In the present report we describe the first 18 months’ experience of a novel pediatric breast clinic that was established to provide comprehensive care to children and adolescents with breast masses. The clinic is staffed by medical personnel who are alert to the unique aspects of breast disease in the young population. It is located in SCMCI, where the environment is oriented to the younger age groups, and it operates in cooperation with the adult Breast Center of Rabin Medical Center on the same campus. Our clinic is distinct from other adolescent breast clinics that focus on cosmetic treatment of macromastia, gynecomastia, and breast asymmetry; this probably explains the high percentage of patients with breast masses in the study cohort. Some of the patients with other findings were referred to the relevant disciplines. Our review revealed several important features of pediatric breast disease. The patients with suspected breast masses accounted for 48.9% of all referrals (23/47). Of them, the diagnosis of breast mass was made clinically and radiologically in 14/47 patients (29.7%) and verified in subsequent biopsy in 7; 7 patients had more than 1 mass.
O. Cohen et al. / J Pediatr Adolesc Gynecol xxx (2019) 1e4
3
Table 1 Patients With Confirmed Breast Mass or Masses Patient Age, years Months since Self-Palpation* Number 1 2 3 4 5 6 7 8 9 10 11 12 13 14
17.4 14.9 16.7 16.5 17.4 17.7 15.5 16.11 15 17.6 16.2 16.5 16.11 17
6 18 5 1 4 27 0.5 12 2 0.5 36 1.5 6 Was detected by physician
Clinical Findings
Sonographyy
Right breast UOQ Upper right breast Multiple, bilateral Multiple, bilateral Right breast UOQ Right breast UOQ Multiple, right breast Multiple, right breast Right breast LOQ Multiple, bilateral Multiple, right breast Multiple, bilateral Right breast, retroalveolar Right breast UOQ
3 cm solid mass 7 cm 15 masses, 3 cm for the largest 5 masses, 4.5 cm for the largest 1.9 cm 5.7 cm 4 masses, 2 cm for the largest 3 masses, 1.4 cm for the largest 4.2 cm mass enlarged to 5.8 cm 2 masses, 3.6 cm for the largest 2 masses, 4.6 cm for the largest 8 masses, 3.7 cm for the largest 3.3 cm 3.7 cm
Largest BI-RADS Score 3 4A 3 4A 4A 4B 3 3 4A 4A 4A 3 3 3
Intervention
Pathology
CNB FA CNB and excision FA F/U CNB FA CNB FA Excision FA F/U F/U CNB and excision FA CNB FA CNB and excision FA F/U F/U F/U
BI-RADS, Breast Imaging and Reporting Data System; CNB, core needle biopsy; FA, fibroadenoma; F/U, clinical follow-up; LOQ, lower outer quadrant; UOQ, upper outer quadrant. * Time elapsed since the patients first palpated the mass to the time for seeking medical advice. y Sonographic description of the mass or masses including the largest diameter.
All masses were diagnosed and treated as fibroadenomas. Four masses were surgically excised. There were no cases of phyllodes or cancer; in agreement with the literature showing a low rate of breast malignancy in the pediatric age group.5e9 Further studies of a larger cohort followed for a longer term are planned. On the basis of the patients’ interviews and clinical encounters we assume that anxiety and shame played a major role in the medical experience of the patients and often led to a delay in their seeking treatment. A protective and welcoming medical environment that is adapted to children and adolescents is important to gain patient trust and compliance for further follow-up. All caregivers in the breast clinic are made aware of these special needs and ways of providing suitable care. From a clinical point of view, surgeons working in the clinic need to be familiar with the normal breast development in the pediatric population and the diseases and lesions that are likely to occur. Accordingly, the evaluation of breast disease in children and adolescents, in addition to clinical judgements and decisions regarding imaging, tissue diagnosis, surgical intervention, and follow-up, should not
be derived from practices in adults. Furthermore, the medical staff should be particularly mindful of patient and parental concerns and anxiety and the effect of the disease or treatment on body image and self-esteem. The physical examination and imaging studies should be performed in an environment conducive to the well-being of the young patients and meet their physical and emotional needs. Considering the emotional state of the patients and parents is crucial to making decisions about imaging, tissue diagnosis, or surgery. It is also important to discuss the differences in adults with the patients and their parents to dispel their concerns and help them to make the right treatment decisions.13 Radiologists also need to be alert to the unique characteristics of pediatric breast disease. According to the American College of Radiology, category 4A of the BI-RADS reporting system represents a 2%-10% likelihood of cancer and mandates tissue diagnosis.14 However, in our cohort, a relatively large number of patients (7/14; 50%) had a BI-RADS score of 4A or higher, but none had a malignancy. This finding suggests that the use of this reporting system in adolescents might lead to inaccurate categorization and consequently, unnecessary stress to
Fig. 1. Macroscopic appearance of a large fibroadenoma (patient 2).Ă
Fig. 2. Fibroadenoma in an anxious patient who had sought medical help only 3 years after self-palpating the mass (patient 11).
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patient and parents. In addition, it can present a dilemma for the surgeon and sometimes lead to mistreatment. Some authors recommend clinical follow-up alone or with sonographic scans in most cases15e17; others recommend biopsy studies of any mass suggestive of malignancy and long-term follow-up for nonsuggestive masses3,17; still others recommend excision of any mass suggestive of malignancy,18,19 or masses that persist on long-term follow-up until age 35 years.19 The significance of the different categorization of breast masses in the pediatric and adult populations has hardly been discussed in the literature.20 Recently Sanders et al21 proposed that any palpable mass in a young patient should undergo ultrasound imaging, which should be followed, if findings are indicative of malignancy, with core biopsy. However, they emphasized that the reason for the biopsy is to rule out phyllodes tumor rather than breast cancer, further emphasizing the irrelevance of the BI-RADS that was developed for the adult population in which the probability of cancer is considerably higher. Further studies are needed to develop an ageadjusted system for the categorization of breast masses. This report presents the preliminary experience for a surgical breast clinic dedicated to the pediatric and adolescent patients. The relatively small cohort and short follow-up is a limiting factor for drawing clear recommendations regarding decision-making while evaluating these patients for either the invasive intervention needed or categorizing the risk for breast masses. Longer-term studies of larger cohorts are needed and planned. Another limit is that probably not all primary physicians are aware of our clinic and patients who are older than 16 years might be referred to an adult clinic according to definitions different to those for adolescents. That might cause selection bias in our clinic toward a younger age. Conclusions
Breast diseases in children and adolescents have several distinct features compared with adults and warrant a different type of management with greater consideration of emotional factors such as body image and shame. We suggest that many of the practices adopted from adults, particular those on the basis of the use of the BI-RADS scores, are inappropriate for this age group. This study highlights the need for tertiary clinics dedicated to breast
diseases in children and adolescents that offer comprehensive, age-adjusted care in a stress-free environment. Acknowledgments
The authors thank our research coordinator Naama Tirosh for her exceptional contribution to the current and other reports. References 1. De Silva NK, Brandt ML: Disorders of the breast in children and adolescents, part 2: breast masses. J Pediatr Adolesc Gynecol 2006; 19:415 2. García CJ, Espinoza A, Dinamarca V, et al: Breast US in children and adolescents. Radiographics 2000; 20:1605 3. West KW, Rescorla FJ, Scherer LR 3rd, et al: Diagnosis and treatment of symptomatic breast masses in the pediatric population. J Pediatr Surg 1995; 30:182 4. Jayasinghe Y, Simmons PS: Fibroadenomas in adolescence. Curr Opin Obstet Gynecol 2009; 21:402 5. Rogers DA, Lobe TE, Rao BN, et al: Breast malignancy in children. J Pediatr Surg 1994; 29:48 6. Corpron CA, Black CT, Singletary SE, et al: Breast cancer in adolescent females. J Pediatr Surg 1995; 30:322 7. NIH: National Cancer Institute. Surveillance, Epidemiology, and End Results Program: SEER Cancer Statistics Review, 1975-2014. Available: https://seer. cancer.gov/csr/1975_2014/. Accessed August 3, 2019. 8. Longo OA, Mosto A, Moran JC, et al: Breast carcinoma in childhood and adolescence: case report and review of the literature. Breast J 1999; 5:65 9. Tea MK, Asseryanis E, Kroiss R, et al: Surgical breast lesions in adolescent females. Pediatr Surg Int 2009; 25:73 10. Kaneda HJ, Mack J, Kasales CJ, et al: Pediatric and adolescent breast masses: a review of pathophysiology, imaging, diagnosis, and treatment. AJR Am J Roentgenol 2013; 200:W204 11. Bock K, Duda VF, Hadji P, et al: Pathologic breast conditions in childhood and adolescence: evaluation by sonographic diagnosis. J Ultrasound Med 2005; 24:1347. [quiz: 1356]. 12. Canadian Paediatric Society: Age limits and adolescents. Paediatr Child Health 2003; 8:577 13. Gao Y, Saksena MA, Brachtel EF, et al: How to approach breast lesions in children and adolescents. Eur J Radiol 2015; 84:1350 €hm-Ve lez M, Berg WA, et al: ACR BI-RADS ultrasound. In: 14. Mendelson EB, Bo D’Orsi CJ, Sickles EA, Mendelson EB, et al, editors. ACR BI-RADS Atlas, Breast Imaging Reporting and Data System, (5th ed.).. Reston, VA, American College of Radiology, 2013, pp 128 15. McLaughlin CM, Gonzalez-Hernandez J, Bennett M, et al: Pediatric breast masses: an argument for observation. J Surg Res 2018; 228:247 16. Smith GE, Burrows P: Ultrasound diagnosis of fibroadenoma - is biopsy always necessary? Clin Radiol 2008; 63:511 17. Santen RJ, Mansel R: Benign breast disorders. N Engl J Med 2005; 353:275 18. Ezer SS, Oguzkurt P, Ince E, et al: Surgical treatment of the solid breast masses in female adolescents. J Pediatr Adolesc Gynecol 2013; 26:31 19. Greenberg R, Skornick Y, Kaplan O: Management of breast fibroadenomas. J Gen Intern Med 1998; 13:640 20. Fu CY, Hsu HH, Yu JC, et al: Influence of age on PPV of sonographic BI-RADS categories 3, 4, and 5. Ultraschall Med 2011; 32(suppl 1):S8 21. Sanders LM, Sharma P, Madany M, et al: Clinical breast concerns in low-risk pediatric patients: practice review with proposed recommendations. Pediatr Radiol 2018; 48:186