New treatment strategy for cupulolithiasis associated with benign paroxysmal positional vertigo of the lateral canal: the head-tilt hopping exercise.

This study was performed to determine whether a novel treatment was effective against cupulolithiasis associated with benign paroxysmal positional vertigo (BPPV) of the lateral semicircular canal, which is characterized by apogeotropic direction-changing nystagmus. We herein describe our head-tilt hopping (HtH) exercise, which is designed to release otoconial debris strongly adhered to the cupula. The subjects were trained to hop while tilting their heads laterally. They completed 3 to 5 exercise sessions per day over a 4-week period. Each session ended with a 20-hop trial. The HtH exercises were performed by 27 patients with intractable lateral canal BPPV who exhibited positional vertigo and persistent nystagmus beating toward the uppermost ear for more than 4 weeks, despite performing therapeutic head shaking in the horizontal plane maneuver. All the patients were subjected to the supine roll test before and immediately after the first trial as well as after 1 and 4 weeks of the program to evaluate the effect of the treatment on their apogeotropic nystagmus. Nystagmus of 9 (33.3 %) patients disappeared immediately after the first training session. After 1 and 4 weeks of the training, the number of patients that had experienced either of these improvements had increased to 15 (55.6 %) and 19 (70.4 %) subjects, respectively. These results suggest that HtH exercises aimed at releasing otoconial debris from the cupula are feasible as a new therapy for cupulolithiasis associated with intractable lateral canal BPPV. However, further studies for comparison with control are required to confirm these preliminary results.

Gallbladder motility disorders estimated by non-invasive methods.

BACKGROUND/AIMS: The aim is to compare the radionuclide (DC) and ultrasonographic (US) method in the assessment of gallbladder (GB) motility. METHODOLOGY: The study was performed in 15 controls (C), 10 patients with acute cholecystitis (AC), 20 patients with chronic acalculous cholecystitis (CAC), 26 patients with chronic cholecystitis with calculosis (CCC) as well as in 15 patients with GB dyskinesia (D). GB emptying period (EP), ejection fraction (EF) and ejection rate (ER) were estimated with dynamic cholescintigraphy (DC) and US. RESULTS: The DC and US finding in the patients with AC was typical in all the patients, i.e. GB was not visualized at all on DC, while on US, stone was visible in the cystic duct. There were significant differences (p<0.05) between the EF and ER values obtained between C and the three groups of patients CAC, CCC and D, using both methods. However, there were no significant differences in EF, EP and ER values among CAC, CCC and D (p>0.05). There was also high correlation between the results obtained with both methods in all the groups of patients studied. CONCLUSIONS: The results obtained by both methods are valuable for the assessment of GB motility. Although there are no significant differences and there is a high correlation between the values, radionuclide method is more precise, because it can register motility continuously.

Recurrent rhinolithiasis: a case report with review of the literature.

Rhinolith is an uncommon nasal mass and a rare entity encountered in clinical practice. The typical symptoms of rhinolith are unilateral nasal obstruction, foul smelling purulent nasal discharge and epistaxis, crusting, swelling of nose or face, anosmia, epiphora and headache. In this report, we present a case of recurrent rhinolith manifesting as an incidental finding on dental radiographs. A 26-year old male patient with a history of long standing halitosis had an operation for rhinolith seven years previously. Rhinolith in the right nasal cavity was diagnosed on rigid endoscopic examination. Rhinolith was removed by using a rigid nasal endoscope under local anaesthesia. To our knowledge, recurrence of rhinolith has not been described previously in the literature.

[Classification and clinical observation of rhinoliths in the adult patients].

This paper reports 3 cases of rhinoliths 20, 14, and 45 years in duration respectively observed during the last 3 years. Rhinoliths caused difficulty of nasal breathing on the side of their localization accompanied by serous discharge from the nose and atrophy of turbinate bones; moreover, they provoked the development of maxilloethmoidal sinusitis, nasal polyps, and deviation of the nasal septum. The surgical removal of the rhinoliths promoted elimination of these complications. A new classification of rhinoliths distinguishing between 4 groups of these stone-like structures is proposed.

Canalithiasis of the anterior semicircular canal (ASC): treatment options based on the possible underlying pathogenetic mechanisms.

Benign paroxysmal positional vertigo (BPPV) of the anterior semicircular canal (ASC) is an uncommon disorder currently diagnosed with the Dix-Hallpike (D-H) examination. According to the literature, nystagmus and vertigo may be more pronounced when the affected ear is either up or down. In some patients, both right and left D-H tests can trigger nystagmus with the same direction. The proposed treatment options with the addition of a different manoeuvre applied by the authors of the present study in cases of ASC lithiasis, seem to present a respective variety regarding the position of the affected ASC during the procedure of canalith repositioning. The aim of this study is to analyse the mechanisms underlying both the proposed treatment options and the clinical findings in the D-H examination. The results of this analysis stimulate further investigation, since they probably imply that repositioning manoeuvres might vary in their effectiveness when applied to different clinical subgroups of ASC BPPV.

A new variant of posterior canal-benign paroxysmal positional vertigo-canalolithiasis.

OBJECTIVE: The characteristics of typical posterior canal-benign paroxysmal positional vertigo-canalolithiasis (P-BPPV-Can) are well known. We found 10 patients with a new variant of P-BPPV-Can: Reverse (Rev)-P-BPPV-Can. The characteristics and pathophysiology of Rev-P-BPPV-Can are discussed. METHODS: For 4 years and 9 months, 10 patients with Rev-P-BPPV-Can (9 women and 1 man; mean age: 73 years) visited our clinic. Within the same period, 268 patients with unilateral typical P-BPPV-Can were treated. During the Dix-Hallpike and Epley maneuvers, nystagmus was recorded using an infrared charge-coupled device camera mounted on a pair of spectacles. RESULTS: The patients exhibited entirely opposite direction of nystagmus from typical P-BPPV-Can in both the Dix-Hallpike head position and upright sitting position. The patients had typical characteristics of nystagmus due to canalolithiasis, such as latency, duration of <1 min, linear-torsional nystagmus, and fatigability. Rev-P-BPPV-Can appeared after the Epley maneuver for typical P-BPPV-Can; in other patients, Rev-P-BPPV-Can changed to typical P-BPPV-Can. The affected sides of Rev-P-BPPV-Can were decided by those of typical P-BPPV-Can. CONCLUSION: Canalolithiasis in the non-ampullary distal portion of the posterior semicircular canal may be a potential pathophysiology of Rev-P-BPPV-Can. The Epley maneuver has little effect for treating Rev-P-BPPV-Can, but spontaneous transition to typical P-BPPV-Can or spontaneous recovery is expected.

Pseudo-spontaneous nystagmus in lateral semicircular canal benign paroxysmal positional vertigo: Correlation with bow and lean test in a pitch plane.

OBJECTIVES: We investigated the incidence and characteristics of pseudo-spontaneous nystagmus (PSN) in benign paroxysmal positional vertigo involving the lateral semicircular canal (LC-BPPV) and evaluated the correlation between PSN and the bow and lean test. METHODS: We examined nystagmus in the sitting position using video-oculography goggles in 131 LC-BPPV patients. The positioning test and bow and lean test were also performed. Patients were divided into canalolithiasis and cupulolithiasis groups according to the character of nystagmus. In each group, the incidence and direction of PSN, correlation with the bow and lean test, and treatment outcome were analyzed. RESULTS: PSN was observed in 25 cases (19.1%) in LC-BPPV patients, 7 of which were canalolithiasis and 18 of which were cupulolithiasis (p = 0.098). Of the 25 patients with PSN, 21 (84%) exhibited nystagmus consistent with the lean test whereas 4 (16%) exhibited nystagmus consistent with the bow test. In patients with PSN, nystagmus was observed in the bow and lean test in all cases (23/23), but in patients without PSN, no nystagmus was observed in 13 cases (13/87) in the bow and lean test (p = 0.048). The number of barbecue maneuvers performed until the end of treatment was 1.4 ± 0.7 in patients with PSN and 1.4 ± 0.9 in those without PSN (p = 0.976). CONCLUSION: We identified PSN in patients with LC-BPPV irrelevant of subtype. Moreover, all patients with PSN showed nystagmus in the bow and lean test. The direction of PSN was mostly consistent with that of the lean test (21/25, 84%). The presence of PSN was not related to the treatment outcome in this study.

T-tube drainage versus choledochojejunostomy in hepatolithiasis patients with sphincter of Oddi laxity: study protocol for a randomized controlled trial.

BACKGROUND: Residual and recurrent stones remain one of the most important challenges of hepatolithiasis and are reported in 20 to 50% of patients treated for this condition. To date, the two most common surgical procedures performed for hepatolithiasis are choledochojejunostomy and T-tube drainage for biliary drainage. The goal of the present study was to evaluate the therapeutic safety and perioperative and long-term outcomes of choledochojejunostomy versus T-tube drainage for hepatolithiasis patients with sphincter of Oddi laxity (SOL). METHODS/DESIGN: In total, 210 patients who met the following eligibility criteria were included and were randomized to the choledochojejunostomy arm or T-tube drainage arm in a 1:1 ratio: (1) diagnosed with hepatolithiasis with SOL during surgery; (2) underwent foci removal, stone extraction and stricture correction during the operation; (3) provided written informed consent; (4) was willing to complete a 3-year follow-up; and (5) aged between 18 and 70 years. The primary efficacy endpoint of the trial will be the incidence of biliary complications (stone recurrence, biliary stricture, cholangitis) during the 3 years after surgery. The secondary outcomes will be the surgical, perioperative and long-term follow-up outcomes. DISCUSSION: This is a prospective, single-centre and randomized controlled two-group parallel trial designed to demonstrate which drainage method (Roux-en-Y hepaticojejunostomy or T-tube drainage) can better reduce biliary complications (stone recurrence, biliary stricture, cholangitis) in hepatolithiasis patients with SOL. TRIAL REGISTRATION: Clinical Trials.gov: NCT04218669 . Registered on 6 January 2020.

Particle motion in unsteady two-dimensional peristaltic flow with application to the ureter.

Particle motion in an unsteady peristaltic fluid flow is analyzed. The fluid is incompressible and Newtonian in a two-dimensional planar geometry. A perturbation method based on a small ratio of wave height to wavelength is used to obtain a closed-form solution for the fluid velocity field. This analytical solution is used in conjunction with an equation of motion for a small rigid sphere in nonuniform flow taking Stokes drag, virtual mass, Faxén, Basset, and gravity forces into account. Fluid streamlines and velocity profiles are calculated. Theoretical values for pumping rates are compared with available experimental data. An application to ureteral peristaltic flow is considered since fluid flow in the ureter is sometimes accompanied by particles such as stones or bacteriuria. Particle trajectories for parameters that correspond to calcium oxalates for calculosis and Escherichia coli type for bacteria are analyzed. The findings show that retrograde or reflux motion of the particles is possible and bacterial transport can occur in the upper urinary tract when there is a partial occlusion of the wave. Dilute particle mixing is also investigated, and it is found that some of the particles participate in the formation of a recirculating bolus, and some of them are delayed in transit and eventually reach the walls. This can explain the failure of clearing residuals from the upper urinary tract calculi after successful extracorporeal shock wave lithotripsy. The results may also be relevant to the transport of other physiological fluids and industrial applications in which peristaltic pumping is used.

Head accelerations during particle repositioning manoeuvres.

Benign paroxysmal positional vertigo (BPPV) due to canalithiasis can be treated with particle repositioning manoeuvres, which aim to evacuate trapped particles from the semicircular canals (SCC). The movement of particles within the SCC is affected by gravity as well as by the accelerations of the head during the manoeuvres. Moreover, as experienced by the particles, gravity is indistinguishable from an upward acceleration of the SCC in free space. We used a set of three orthogonal linear accelerometers to measure the net three-dimensional linear acceleration vector acting on the head during the Hallpike manoeuvre and Epley and Semont particle repositioning manoeuvres (which are used to treat posterior canal BPPV). The projection of the net acceleration vector onto the SCC planes showed that both the Epley and Semont manoeuvres approximated to stepwise, 360 degrees , backward rotations in the plane of the targeted posterior canal. Angular velocity measurements however showed that the rotational component during the central stages of these two manoeuvres is opposite in direction. A simple model of head rotations during particle repositioning manoeuvres was created which showed good agreement to the linear acceleration measurements. Analysis of modelled and measured data identified that speed of movement during the Semont manoeuvre should be critical to its clinical success.

Benign paroxysmal positional vertigo of the anterior semicircular canal: atypical clinical findings and possible underlying mechanisms.

Anterior semicircular canal (ASC) lithiasis is uncommon and usually self-treated. In the rare cases when such patients seek medical advice, diagnosis requires careful consideration of the patient's symptoms and the clinical characteristics of the nystagmus triggered by the Dix-Hallpike (D-H) examination. In this study, two atypical cases of ASC benign paroxysmal positional vertigo (BPPV) are presented and the relevant literature is reviewed. Regardless of their unique symptoms, both participants in this study were diagnosed with lithiasis of the left ASC. The clinical manifestations of ASC BPPV may differ significantly from typical symptoms seen in the more common posterior semicircular canal BPPV. The possible mechanisms responsible for this intriguing variance, as well as guidelines for diagnosing the affected side, are discussed.

Simulation of cupulolithiasis and canalolithiasis by an animal model.

The physical mechanisms responsible for cupulolithiasis and canalolithiasis have been investigated by two groups of experiments in isolated posterior semicircular canal (SCC) of frog (Rana esculenta L.). First, clouds of 10-30 isolated otoconia were let to fall (snowfall of otoconia) either through the ampulla onto the cupula, or inside the long arm of the canal, opposite to the cupula. Second, microspheres ranging 30 to 350 microm in diameter were gently moved to and fro inside the long arm of the canal by a micromanipulator. The effects were evaluated by recording the firing rate (Nfr) of the ampullary nerve. Snowfall of otoconia produced detectable changes of Nfr only when otoconia got in contact with the cupula, but not when falling through the endolymph. Movement of the microspheres in the canal long arm induced Nfr changes only if the microsphere diameter exceeded about 50 microm. Although the exact microsphere size needed for receptor stimulation may depend on the experimental conditions, these results strongly suggest that debris moving inside a SCC (canalolithiasis) can produce transcupular pressures able to stimulate ampullar receptors only if they have suitable size, whereas isolated otoconia cannot, except when lying on the cupula (cupulolithiasis).

Phenotypic testicular abnormalities and pubertal development in boys with McCune-Albright syndrome.

Aim of this survey is to review the few available literature data on pathophysiologic and clinical aspects of pubertal development in boys with McCune-Albright syndrome (MAS). On the basis of such analysis, we concluded that:1) peripheral precocious puberty (PPP) is significantly more infrequent in boys than in girls; 2) the most common testicular abnormality at MAS presentation is macroorchidism, that may be either monolateral or bilateral; 3) macroorchidism is not always associated with clinical and biochemical evidence of PPP; 4) testicular microlothiasis is distinctly more frequent in boys with MAS than in those without MAS; 5) the available therapeutic schedules have to be adopted already at MAS presentation only in the cases with PPP.

Anterior canal benign paroxysmal positional vertigo: an underappreciated entity.

OBJECTIVE: Evaluate the frequency and characteristics of benign paroxysmal positional vertigo (BPPV) arising from involvement of the anterior semicircular canal (AC) as compared with the posterior canal (PC) and horizontal canal (HC). STUDY DESIGN: Prospective review of patients with BPPV. SETTING: Tertiary referral center. PATIENTS: A total of 260 patients who were evaluated for vertigo were identified as experiencing BPPV. INTERVENTIONS: Standard vestibular assessment including the use of electrooculography (EOG) or video-oculography (VOG) was completed on all patients. Based on EOG/VOG findings, the BPPV origin was attributed to AC, PC, or HC involvement secondary to canalithiasis versus cupulolithiasis. Treatment was performed with canalith repositioning maneuvers (CRMs) appropriate for type of canal involvement. RESULTS: For the 260 patients, the positionally induced nystagmus patterns suggested the canal of origin to be AC in 21.2%, PC in 66.9%, and HC in 11.9%. Cupulolithiasis was observed in 27.3% of the AC, 6.3% of the PC, and 41.9% of the HC patients. Head trauma was confirmed in the history preceding the onset of vertigo in 36.4% of the AC, versus 9.2% of the PC and 9.7% of the HC patients (p < 0.001). The number of CRMs completed to treat the BPPV did not differ between canals involved (1.32 for AC, 1.49 for PC, and 1.34 for HC). CONCLUSION: The direction of subtle vertical-beating nystagmus underlying the torsional component is critical in differentiating AC versus PC origin; EOG/VOG aids in accurate assessment of the vertical component for the diagnosis of canal involvement. AC involvement may be more prevalent than previously appreciated, particularly if the examiner does not appreciate the vertical component of the nystagmus or the diagnosis is made without the assistance of EOG/VOG. Head trauma history is significantly more frequent in AC versus other forms of BPPV, and patients with a history of head trauma should be examined closely for AC involvement. CRM is as successful for treatment of AC BPPV as for other types of BPPV.

Dynamics of canal response to head-shaking test in benign paroxysmal positional vertigo.

CONCLUSIONS: Time constant and maximum slow phase velocity (SPV) of head-shaking nystagmus (HSN) demonstrated a differential canal response to head shaking in 24% of patients with posterior canal benign paroxysmal positional vertigo (BPPV). We suggest that vestibular lithiasis has a limited contribution to the mechanism that generates HSN. OBJECTIVE: To determine the canal response to head shaking in BPPV. PATIENTS AND METHODS: This was a case-control study including 104 individuals with BPPV. The diagnosis was based on the presence of vertigo and nystagmus during the positional test. Subjects were examined by the horizontal and vertical head-shaking test. Eye movements were recorded on a video camera to analyze the nystagmus. The head was shaken passively in the horizontal and sagittal planes, respectively, for horizontal and vertical HSN at a frequency of 2 Hz. HSN was considered when six consecutive beats of nystagmus with an SPV of at least 2 degrees/s were detected. Main outcome measures were the presence of horizontal and vertical HSN, maximum SPV of HSN, time constant of HSN, and canal paresis. RESULTS: Maximum SPV of vertical HSN was higher in BPPV patients with posterior canal BPPV (n = 10) than in controls (p = 0.04). Moreover, the time constant of vertical HSN was significantly lower for posterior canal BPPV when compared with controls (p < 0.02).

Spontaneous nystagmus and canalithiasis preceding a loss of vestibular function.

The patient, a 45-year-old female with Ménière's disease, was submitted to endolymphatic sac surgery because of frequent spells of vertigo. The day after surgery she became dizzy and lost the hearing in the operated ear. She presented with a brisk nystagmus towards the healthy ear; however, a remaining vestibular function on the operated side was still present. The head impulse test was normal and in the positioning test, when turning to the operated ear, the spontaneous nystagmus was replaced by a transient horizontal nystagmus in the opposite direction. It is likely that this geotropic nystagmus was elicited from the operated ear because of debris that had accumulated in the lateral semicircular canal. Due to a suspicion of post surgical edema, the patient was treated with prednisolone, but the progression of the lesion could not be arrested. The signs of canalithiasis disappeared, but the head impulse test became pathologic as did the caloric reaction. The patient remained deaf in the operated ear.

'Bow and lean test' to determine the affected ear of horizontal canal benign paroxysmal positional vertigo.

OBJECTIVE: One of the problems in the management of horizontal semicircular canal benign paroxysmal positional vertigo (HSC-BPPV) is the difficulty in determining the affected ear using Ewald's second law. The purpose of this study is to develop the new "bow and lean test (BLT)" to easily determine the affected ear of HSC-BPPV and evaluate its efficiency. METHODS: We compared the efficiency between the classic method and BLT in 26 patients with HSC-BPPV. The classic method is based on Ewald's second law comparing the intensity of nystagmus or symptoms in the head roll test. BLT is based on the direction of both "bowing nystagmus" and "leaning nystagmus" at the head's bowing and leaning state in a sitting position. The affected ear is the same direction of bowing nystagmus in canalolithiasis and the same direction of leaning nystagmus in cupulolithiasis. RESULTS: In 26 patents (15 canalolithiasis, 11 cupulolithiasis), 3 (11.5%) patients did not show a prominent affected ear in the classic method, and 7 (26.9%) patients showed the different affected ear between the two methods. All 10 patients were successfully treated with just one trial of barbecue rotation based on the affected ear in BLT. Three patients did not show any bowing or leaning nystagmus. The side with canal paresis in all four patients, who showed significant canal paresis in bithermal caloric tests, was equal to the affected ear based on BLT. CONCLUSIONS: The "bow and lean test" (also called "Choung's test") is a new method that can easily determine the affected ear of HC-BPPV.

Laparoscopic Cholecystectomy for Gallbladder Calculosis in Fibromyalgia Patients: Impact on Musculoskeletal Pain, Somatic Hyperalgesia and Central Sensitization.

Fibromyalgia, a chronic syndrome of diffuse musculoskeletal pain and somatic hyperalgesia from central sensitization, is very often comorbid with visceral pain conditions. In fibromyalgia patients with gallbladder calculosis, this study assessed the short and long-term impact of laparoscopic cholecystectomy on fibromyalgia pain symptoms. Fibromyalgia pain (VAS scale) and pain thresholds in tender points and control areas (skin, subcutis and muscle) were evaluated 1week before (basis) and 1week, 1,3,6 and 12months after laparoscopic cholecystectomy in fibromyalgia patients with symptomatic calculosis (n = 31) vs calculosis patients without fibromyalgia (n. 26) and at comparable time points in fibromyalgia patients not undergoing cholecystectomy, with symptomatic (n = 27) and asymptomatic (n = 28) calculosis, and no calculosis (n = 30). At basis, fibromyalgia+symptomatic calculosis patients presented a significant linear correlation between the number of previously experienced biliary colics and fibromyalgia pain (direct) and muscle thresholds (inverse)(p<0.0001). After cholecystectomy, fibromyalgia pain significantly increased and all thresholds significantly decreased at 1week and 1month (1-way ANOVA, p<0.01-p<0.001), the decrease in muscle thresholds correlating linearly with the peak postoperative pain at surgery site (p<0.003-p<0.0001). Fibromyalgia pain and thresholds returned to preoperative values at 3months, then pain significantly decreased and thresholds significantly increased at 6 and 12months (p<0.05-p<0.0001). Over the same 12-month period: in non-fibromyalgia patients undergoing cholecystectomy thresholds did not change; in all other fibromyalgia groups not undergoing cholecystectomy fibromyalgia pain and thresholds remained stable, except in fibromyalgia+symptomatic calculosis at 12months when pain significantly increased and muscle thresholds significantly decreased (p<0.05-p<0.0001). The results of the study show that biliary colics from gallbladder calculosis represent an exacerbating factor for fibromyalgia symptoms and that laparoscopic cholecystectomy produces only a transitory worsening of these symptoms, largely compensated by the long-term improvement/desensitization due to gallbladder removal. This study provides new insights into the role of visceral pain comorbidities and the effects of their treatment on fibromyalgia pain/hypersensitivity.

[Horizontal canal benign paroxysmal positional vertigo--its affected side and horizontal semicircular canal function].

We report horizontal canal BPPV (HC-BPPV) targeting its pathophysiology, the affected side, and the function of the horizontal semicircular canal, together with a review of the literature. Subjects were 13 patients with HC-BPPV visiting our vertigo outpatient clinic at Nara medical university hospital and a related hospital in the 2.5 years from January 2000 to June 2002. Subjects were classified into 7 with canalolithiasis and 6 patients of cupulolithiasis after a neurotological examination. CP was positive in 54% of all patients, 71% of those with canalolithiasis, and 33% of those with cupulolithiasis. To determine the affected side in HC-BPPV, we used the affected side by using the law of Ewald in canalolithaisis patients and the detection of a neutral diminishing nystagmus in cupulolithiasis patients. CP positive in caloric testing indicated insignificant dysfunction of the horizontal semicircular canal in canalolithiasis patients compared to that in cupulolithasis patients. The mechanism behind caloric nystagmus was thought to be a convection of endolymphatic fluid interrupted consequently by an otolith in the semicircular canal in canalolithiasis patients. In contrast, CP was positive in cupulolithiasis patients regarded as having no convection of endolymphatic fluid. The mechanism causing a difference in caloric test results between canalolithiasis and cupulolithiasis patients thus requires a larger patient population and further examination to be conclusive.

Oddi sphincter preserved cholangioplasty with hepatico-subcutaneous stoma for hepatolithiasis.

AIM: To evaluate the long-term outcomes of Oddi sphincter preserved cholangioplasty with hepatico-subcutaneous stoma (OSPCHS) and risk factors for recurrence in hepatolithiasis. METHODS: From March 1993 to December 2012, 202 consecutive patients with hepatolithiasis underwent OSPCHS at our department. The Oddi sphincter preserved procedure consisted of common hepatic duct exploration, stone extraction, hilar bile duct plasty, establishment of subcutaneous stoma to the bile duct. Patients with recurrent stones can undergo stone extraction and/or biliary drainage via the subcutaneous stoma which can be incised under local anesthesia. The long-term results were reviewed. Cox regression model was employed to analyze the risk factors for stone recurrence. RESULTS: Ninety-seven (48.0%) OSPCHS patients underwent hepatic resection concomitantly. The rate of surgical complications was 10.4%. There was no perioperative death. The immediate stone clearance rate was 72.8%. Postoperative cholangioscopic lithotomy raised the clearance rate to 97.0%. With a median follow-up period of 78.5 mo (range: 2-233 mo), 24.8% of patients had recurrent stones, 2.5% had late development of cholangiocarcinoma, and the mortality rate was 5.4%. Removal of recurrent stones and/or drainage of inflammatory bile via subcutaneous stoma were conducted in 44 (21.8%) patients. The clearance rate of recurrent stones was 84.0% after subsequent choledochoscopic lithotripsy via subcutaneous stoma. Cox regression analysis showed that residual stone was an independent prognostic factor for stone recurrence. CONCLUSION: In selected patients with hepatolithiasis, OSPCHS achieves excellent long-term outcomes, and residual stone is an independent prognostic factor for stone recurrence.